Lippen-Kiefer-Gaumen Spalten

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Lippen-Kiefer-Gaumen Spalten
Aus der Universitätsklinik für
Hals-, Nasen-, Ohrenheilkunde
der Albert-Ludwigs-Universität Freiburg i. Br.
Lippen-Kiefer-Gaumen Spalten:
Objektivierung der Sprachanalyse durch ein
automatisches Spracherkennungsprogramm
Inaugural- Dissertation
zur
Erlangung des Medizinischen Doktorgrades
der Medizinischen Fakultät
der Albert-Ludwigs-Universität
Freiburg i. Br.
Vorgelegt: 2012
von: Julia Gottges
geboren in: West Palm Beach, USA.
i
Dekan: Professor Dr. Dr. h.c. mult. Hubert E. Blum
Referent: Professor Dr. med. E. Löhle
Koreferent: Professor Dr. med Dr. med. dent. P. Stoll
Jahr der Promotion: 2012
ii
Acknowledgements:
A special thanks goes to Prof. Dr. Erwin Löhle. I could not have wished for a better counselor
and supervisor. Prof. Löhle was always available and helpful, sacrificing many hours to
discuss problems and offering stimulating suggestions throughout my work on this thesis. His
continual advice and encouragement were invaluable.
I would also like to thank Dr. Yael Gilgen and Nina Ospelt for their corroboration in this
project. They supervised most of the PEAKS recordings and also participated in the
perceptive assessment. Due to their efforts, enough data could be acquired. Thanks to Prof.
Dr. rer. Nat. Dieter Hauschke for his support in the statistical analysis of the data.
The support and encouragement of the Clad family was also greatly appreciated. Marius Clad
and PD Dr. Andreas Clad played a central role in correcting English style and grammar,
formatting the final version of this thesis and offering suggestions for improvement.
Lastly, I would like to thank my parents, my brother and Marius Clad for their constant love,
patience and support.
iii
Table of Contents
1
INTRODUCTION ...............................................................................................................................1
2
BACKGROUND .................................................................................................................................2
2.1
EPIDEMIOLOGY
OF
CRANIOFACIAL
CLEFTS ....................................................................................................2
2.2
ETIOLOGY
OF
CRANIOFACIAL
CLEFTS............................................................................................................2
2.3
EMBRYOLOGY
OF
CRANIOFACIAL
CLEFTS: .....................................................................................................3
2.4
ANATOMY
OF
THE
SOFT
PALATE .................................................................................................................6
2.4.1
The Muscles of the Soft Palate ..................................................................................................6
2.4.2
The Significance of the “Muscular Rings” of the Soft Palate .....................................................6
2.5
CONSEQUENCES
OF
A
CRANIOFACIAL
CLEFT ...................................................................................................7
2.6
LINGUISTIC
ASPECTS
OF
CLP
PATIENTS ........................................................................................................8
2.6.1
Velopharyngeal Inadequacy ......................................................................................................8
2.6.2
Rhinolalia ...................................................................................................................................9
2.6.3
Articulation disorders ................................................................................................................9
2.6.4
Speech Therapy........................................................................................................................10
2.7
HEARING
IMPAIRMENT
OF
CLP
PATIENTS...................................................................................................10
2.7.1
Middle Ear Disease and the Eustachian Tube ..........................................................................10
2.7.2
Hearing as a Prerequisite for Speech Acquisition ....................................................................12
2.7.3
Treatment of Middle Ear Disease ............................................................................................13
2.8
INTERDISCIPLINARY
THERAPY
OF
CLP
PATIENTS ...........................................................................................13
2.8.1
Outline of the Treatment Concept of CLP patients: .................................................................14
2.8.2
Treatment Concept in Freiburg:...............................................................................................15
2.8.2.1
2.8.2.2
2.8.2.3
Lip
Operation......................................................................................................................................................16
Middle
Ear
Treatment
Indication .......................................................................................................................19
Palate
Operation ................................................................................................................................................20
2.8.3.1
2.8.3.2
2.8.3.3
2.8.3.4
Palatal
Obturator................................................................................................................................................22
Lip
Operation......................................................................................................................................................23
Middle
Ear
Treatment
Indication .......................................................................................................................24
Palate
Operation ................................................................................................................................................24
2.8.3
Treatment Concept in Erlangen ...............................................................................................22
2.8.4
Myringotomy and Grommet Insertion.....................................................................................26
2.9
DIAGNOSTIC
METHODS
FOR
PATHOLOGICAL
SPEECH ....................................................................................28
2.9.1
Perceptive Evaluation ..............................................................................................................28
2.9.2
Objective Evaluation ................................................................................................................29
2.9.3
PEAKS.......................................................................................................................................30
2.10
THE
HYPOTHESES
OF
THIS
STUDY .........................................................................................................33
2.11
THE
OBJECTIVES
OF
THIS
STUDY ..........................................................................................................34
3
MATERIALS AND METHODS ...........................................................................................................35
3.1
PATIENTS..............................................................................................................................................35
3.1.1
Data Source .............................................................................................................................35
3.1.2
Data Processing .......................................................................................................................35
3.2
PROCEDURE ..........................................................................................................................................36
3.2.1
Data Collection ........................................................................................................................36
3.2.2
PEAKS Recordings ....................................................................................................................36
3.2.3
Speech Evaluation of the CLP Patients.....................................................................................37
3.2.4
Statistical Analysis ...................................................................................................................37
3.3
MATERIALS
AND
METHODS
ANALYSIS........................................................................................................38
3.3.1
Database..................................................................................................................................38
3.3.2
PEAKS Recording......................................................................................................................38
3.3.3
Subjective Speech Intelligibility Assessment ............................................................................38
3.3.4
Improving the Procedure .........................................................................................................39
iv
4
RESULTS ........................................................................................................................................40
4.1
OVERVIEW
OF
THE
PATIENTS....................................................................................................................40
4.1.1
Total Number of Patients (126 Patients) .................................................................................40
4.1.2
Patients with Additional Illnesses ............................................................................................40
4.1.3
Timing of the Cleft Operations in Freiburg ..............................................................................41
4.2
PEAKS
ASSESSMENT ..............................................................................................................................42
4.2.1
Patients Included in the PEAKS Assessment.............................................................................42
4.2.2
Comparison of the objective and subjective Speech Intelligibility Assessment........................42
4.2.3
Comparison of the subjective Intelligibility Assessments of the different Raters‐ Linguist, ENT Doctor and Medical Student..........................................................................................................44
4.2.4
Word Recognition Rate considering Group, Gender and Age..................................................45
4.2.5
Word Recognition Rate considering Operation Method..........................................................47
4.3
EVALUATION
OF
GROMMET
INSERTION
IN
FREIBURG ...................................................................................48
4.3.1
Overview of the Grommets Inserted in the 126 patients evaluated: Were they placed during or after the Palate Operation? ......................................................................................................48
4.3.2
Word Recognition Rate considering the number of Grommets Inserted .................................49
4.3.3
Word Recognition Rate considering the Timing of the Grommet Insertion.............................51
4.4
SUMMARY:
PEAKS
RESULTS
CONSIDERING
THE
OBJECTIVES
OF
THE
STUDY .....................................................52
5
DISCUSSION...................................................................................................................................54
5.1
SPEECH
ANALYSIS
AND
THE
PEAKS
PROGRAM ............................................................................................54
5.2
EFFECT
OF
CLEFT
TYPE
ON
THE
DEGREE
OF
SPEECH
INTELLIGIBILITY .................................................................62
5.3
EFFECT
OF
GENDER
ON
THE
DEGREE
OF
SPEECH
INTELLIGIBILITY .....................................................................63
5.4
EFFECT
OF
AGE
ON
THE
DEGREE
OF
SPEECH
INTELLIGIBILITY ..........................................................................64
5.5
INTER‐CENTER
STUDY:
EFFECT
OF
DIFFERENT
TREATMENT
STRATEGIES
ON
THE
DEGREE
OF
SPEECH
INTELLIGIBILITY 65
5.6
EFFECT
OF
GROMMET
INSERTION
ON
THE
DEGREE
OF
SPEECH
INTELLIGIBILITY ..................................................68
5.7
IMPLICATIONS .......................................................................................................................................72
5.8
CONCLUSION.........................................................................................................................................74
6
SUMMARY.....................................................................................................................................75
7
ZUSAMMENFASSUNG ....................................................................................................................76
8
BIBLIOGRAPHY ..............................................................................................................................77
9
APPENDIX......................................................................................................................................84
9.1
DATA
ENTRY
FORM ................................................................................................................................84
9.2
PEAKS
QUESTIONNAIRE .........................................................................................................................85
9.3
VOCABULARY
OF
THE
PLAKSS
TEST ..........................................................................................................86
v
Tables: Table
2‐1
Embryogenesis
of
the
Palate
‐
Chronological
Overview..........................................................3
Table
2‐2
Chronological
overview
of
the
Concept
of
Erlangen.
Adapted
from
(Wohlleben,
2004) ......22
Table
4‐1
Number
of
Patients
according
to
Group
and
Cleft
Typ.
(Group
1:
Patients
without
a
palate
cleft.
Group
2:
Patients
with
a
palate
cleft.)..................................................................................40
Table
4‐2
Number
of
Patients
with
Additional
Illnesses .......................................................................40
Table
4‐3
Number
of
Patients
according
to
Group
and
Cleft
Type .......................................................42
Table
4‐4
Table
comparing
the
Speech
Intelligibility
Evaluation
(of
a
medical
student,
an
ENT
Doctor
and
a
linguist)
with
the
estimated
WR
Values
of
the
PEAKS
Program ..........................................42
Table
4‐5
Comparing
the
Speech
Intelligibility
Evaluation
of
different
Raters .....................................44
Table
4‐6
WR
Values
according
to
Group..............................................................................................45
Table
4‐7
WR
Value
according
to
Gender .............................................................................................46
Table
4‐8
WR
Values
according
to
Age..................................................................................................46
Table
4‐9
WR
values
considering
Operation
Method ...........................................................................47
Table
4‐10
Grommet
insertions
in
Freiburg:
A
conservative
approach ................................................48
Table
4‐11
Number
of
Grommets
Inserted
according
to
the
Group
of
the
Cleft
Patient .....................48
Table
4‐12
WR
Values
according
to
the
Number
of
Grommets
inserted..............................................49
Table
4‐13
The
effect
of
the
early
or
late
grommet
insertion
on
the
CLP
patients’
degree
of
intelligibility:
according
to
the
achieved
WR
Value.......................................................................51
Table
9‐1
Original
Target
words
of
the
PLAKSS
test
(Fox,
2002).
For
the
extended
vocabulary
see
(Maier,
2009).................................................................................................................................86
Figures: Figure
2‐1
Embryonic
Development
in
the
4th
Gestation
Week
(Aronson,
1990)
p.
217 ........................4
Figure
2‐2
Embryonic
Development
in
the
6th
Gestation
Week:
Intermaxillary
Segment
is
formed.
(Aronson,
1990)...............................................................................................................................4
Figure
2‐3
Embryonic
Development
from
the
7th
until
the
9th
Gestation
Week
(Aronson,
1990)
p.
218
.........................................................................................................................................................5
Figure
2‐4
Muscles
of
the
Soft
Palate.
(A.
Kummer,
2000)(p.
14) ...........................................................7
Figure
2‐5
Opening
of
the
Eustachian
Tube:
the
tensor
veli
palatine
muscle
pulls
the
lateral
hooked
cartilage
away
from
the
membranous
wall
of
the
tube.
(Peterson‐Falzone,
et
al.,
2010),
p.208.12
Figure
2‐6
Millard
Procedure,
just
before
the
closing
procedure
(Van
de
Ven,
Defrancq,
Defrancq,
&
Rooljen,
2008) ...............................................................................................................................16
Figure
2‐7
Chronological
Overview:
Millard
Procedure
for
Cleft
lip
Repair
(Bitter,
2000) ....................17
Figure
2‐8
Veau
III
Procedure:
A‐
Lines
of
Incision,
B‐
Sutured
Bilateral
Cleft
(Cronin
&
Penoff,
1971)
.......................................................................................................................................................18
Figure
2‐9
Schematic
drawing
of
the
three
facial
muscular
rings
(perinasal,
perioral,
perimental)
which
are
all
disturbed
by
the
cleft
lip.
Note
the
attachment
of
the
muscles
to
the
primary
growth
centre
(nasal
cartilage)
and
the
secondary
growth
centre
(zygomaticomaxillary
suture).
(Hemprich,
et
al.,
2006).................................................................................................................19
Figure
2‐10
Muscles
of
the
soft
Palate:
Left=
cleft,
Right=
non‐cleft
(Markus,
et
al.,
1993) ..............20
Figure
2‐11
Delaire
Operation
Method.
(Anastassov
&
Joos,
2001)
p.
106 ..........................................21
Figure
2‐12
Tennison
Randall
Procedure
(Van
de
Ven,
et
al.,
2008) .....................................................23
vi
Figure
2‐13
Lip
closure
combined
with
a
vomer
flap
closure
of
the
hard
palate.
(Lehner,
et
al.,
2003)
.......................................................................................................................................................23
Figure
2‐14
Forked
Flap
Procedure
(Kastenbauer
&
Tardy,
2005) ........................................................24
Figure
2‐15
Widmaier
Operation
Method
(Wiedmaier,
1991)..............................................................25
Figure
2‐16
Langenbeck
Operation
Method
(Patel,
et
al.,
2012) .........................................................26
Figure
2‐17
Grommet
Insertion.
(Donaldson,
1966) .............................................................................27
Figure
2‐18
Otitis
media
with
effusion:
multiple
air/fluid
levels.
Same
after
treatment
with
grommet.
(R.
Shaw,
et
al.,
2003)....................................................................................................................27
Figure
2‐19
The
architecture
of
the
PEAKSlocal
System.
The
normal
PEAKS
client
is
connected
to
a
simulated
PEAKS
server,
which
has
just
the
ability
to
store
new
recordings.
Later
on,
the
recorded
data
can
be
committed
to
the
real
PEAKS
server.
(Maier,
2009) ..................................30
Figure
2‐20
Example
of
a
Pictogram
Slide
used
in
the
PEAKS
Program
(Maier,
et
al.,
2006) ................32
Figure
4‐1
Timing
of
Lip
Operation
in
Freiburg:
126
patients
assessed.
(Age
of
3
Patients
unknown) .41
Figure
4‐2
Timing
of
Palate
Operation
in
Freiburg:
126
patients
assessed.
(Age
of
5
Patients
unknown)
.......................................................................................................................................................41
Figure
4‐3
Regression
Analysis
comparing
the
WR
Values
and
the
Linguist’s
Speech
Intelligibility
Rating ............................................................................................................................................43
Figure
4‐4
Regression
Analysis
comparing
the
WR
Values
and
the
Medical
Student’s
Speech
Intelligibility
Rating........................................................................................................................43
Figure
4‐5
Regression
Analysis
comparing
the
medical
Student’s
and
Linguist’s
Speech
Intelligibility
Rating. ...........................................................................................................................................45
Figure
4‐6
WR
values
according
to
the
Age
of
the
Cleft
Patient ...........................................................47
Figure
4‐7
left:
Pie
Graph
showing
the
Number
of
Grommets
Inserted
in
the
Cleft
Patients
of
Group
1
right:
Pie
Graph
showing
the
Number
of
Grommets
Inserted
in
the
Cleft
patients
of
Group
2 ...49
Figure
4‐8:
Box
Plot
showing
the
Influence
of
the
Number
of
Grommet
Insertions
on
the
WR
Value.50
Figure
4‐9:
Bar
Chart
showing
the
Influence
of
the
number
of
Grommets
inserted
on
the
mean
WR
value ..............................................................................................................................................51
Figure
5‐1:
Type
of
Assessed
Speech
Variables
in
Reviewed
Articles
(n=88)
(Lohmander
&
Olsson,
2004) .............................................................................................................................................58
1
1 Introduction
In this study, the speech intelligibility of children with craniofacial clefts is evaluated
objectively and subjectively using a newly developed speech recognition program named
PEAKS. PEAKS is an abbreviation for “Program to Evaluate and Analyze all Kinds of
Speech disorders” and was developed in Erlangen in 2006. It shows potential in the clinical
application of speech diagnostics in various fields of medicine. In this study the validity and
clinical value of the PEAKS program is investigated in examining speech deficits of cleft
patients.
Sixty cleft patients of the Oral and Maxillofacial Surgery Department in Freiburg were
recorded using PEAKS software. To quantify intelligibility objectively, a word recognition
rate (WR) was calculated as the percentage of correctly recognized words of a standardized
speech test. These patients were subsequently also assessed subjectively through trained and
untrained raters. A high correlation between the objective PEAKS results and the subjective
ratings of the trained staff, would demonstrate the validity of the program.
Background information of the patients concerning cleft type, gender and age were extracted
from the archives of the Phoniatrics and Pedaudilogy Department of Freiburg. This data
enabled us to investigate the influence of the mentioned variables on the WR value, and hence
compare the results with other studies. The patients’ medical files also gave an overview of
the current status of the cleft patient collective in Freiburg and of the present operative and
diagnostic procedures. The clinical value of the program was investigated by evaluating the
speech outcome of the CLP1 patients regarding the different treatments applied. The WR
results were compared regarding the number of grommets inserted and also on the operative
method implemented (comparing the methods of Erlangen and Freiburg).
1
CLP:
The
term
“CLP
patient”
refers
to
any
patient
with
a
cleft:
ranging
from
a
slight
notch
in
the
lip,
to
a
bilateral
lip,
alveolus
and
palate
cleft.
2
2 Background
2.1 Epidemiology of Craniofacial Clefts
Craniofacial clefts rank as among the most frequent innate malformation (11-15%)
(Hemprich, 2011) following heart defects (Wohlleben, 2004). Clefts of the lip, alveolus and
palate are regarded as one of the most important and common craniofacial malformations.
The incidence is estimated around 1-2.2:1000 in Europe (Derijcke, 1996), with characteristic
regional variations (Mossey, 2007) and differences in race and gender. About 1,200 children
are born with a CLP every year in Germany. A general, continual increase in frequency has
been observed and is considered due to industrialization and the hence changed
environmental and living conditions (Wohlleben, 2004).
Clefts of the lip, alveolus and palate exist in various forms, either isolated or combined with
other developmental abnormalities. Approximately 20% of the clefts involve only lip or lip
and alveolus, 50-70% are complete lip, alveolus and palate clefts and the remaining 30% are
isolated clefts of the hard or soft palate (Hausamen J. E., 2003).
2.2 Etiology of Craniofacial Clefts
The cause of a craniofacial cleft is not yet fully understood in detail. The development of the
face is an exceptionally complicated process, coordinated by complex morphogenetic events
and rapid proliferative expansion. It is therefore especially susceptible to environmental and
genetic factors, explaining the high incidence of these facial malformations. The scientific
theory which is most commonly accepted today, is that the majority of malformations arise
due to a complex interaction between genetic factors and exogenous influences (Honigmann,
1998).
Exclusively genetic factors are nonetheless held responsible for approximately 10%
(Fanghänel, 1991) of all cleft deformities. The polygenic controlled defects are caused by an
addition of several defective genes. Once a certain threshold is crossed, it can result in the
predisposition to a cleft development. A mendelian inheritance is seldom (20-30%)
(Wohlleben, 2004) and only a few genes which may contribute to clefting have been
identified (Mossey, 2007).
3
Exogenous factors are also believed to be exclusively accountable for up to 10% of the cleft
developments (Fanghänel, 1991). These environmental factors include for example the use
of certain medications (e.g. Steroids, anticonvulsants) and pollutants (e.g. Alcohol, Nicotine)
or the exposure to X-ray radiation. The list of risk factors is continually growing and also
includes viral infectious diseases, systemic diseases, psychological/emotional strains and
even noise exposition and high altitude (Wohlleben, 2004). Exogenous factors are
considered particularly harmful in the first weeks of pregnancy, and should be avoided
(Neumann, 2000); the possible consequence being that the fetus lacks oxygen in the crucial
hours during the fusion of the nasal prominences and palatal shelve, thus increasing the
inclination for a deformity.
2.3 Embryology of Craniofacial Clefts:
A CLP results from an error in the normal embryological development between the 4th and
12th gestation week. The fusion of the 5 major facial prominences occur between the 4th and
10th week of gestation (Larrabee, 2004), with lip development between the 4th and 7th week,
and palate development between the 5th and 12th week (Aronson, 1990) (see Table 2-1). The
spectrum and severity of the clefting is extremely variable and depends on the type, intensity
and time period of interference in the embryologic development.
Gestation
Week
4th
5th
6th
7th
8th
9th
10th
11th
12th
Fusion
of
the
5
major
facial
prominences
Lip
development
Development
of
the
primary
Merging
of
the
palatal
shelves
from
anterior
to
palate
posterior:
forming
the
secondary
palate2
Table 2-1 Embryogenesis of the Palate - Chronological Overview
Lip and alveolar clefts arise due to a disturbance in the development of the lip and primary
palate in the 4th - 7th week of pregnancy. In this time period the frontonasal prominence and
maxillary prominences fuse to form the primary palate (Aronson, 1990). If one imagines the
upper lip to be an archway, then the frontonasal prominence is the keystone and the two
maxillary processes are the arms of the archway (see Figure 2-1). An inadequate fusion
causes a malformation of the intermaxillary segment (see Figure 2-2), and can occur on
either side of the keystone. Cleft lip defects can therefore be unilateral or bilateral.
2
Secondary
Palate:
forms
the
soft
and
hard
palate
posterior
to
the
incisive
foramen.
4
Figure 2-1 Embryonic Development in the 4th Gestation Week (Aronson, 1990) p. 217
Figure 2-2 Embryonic Development in the 6th Gestation Week: Intermaxillary Segment is
formed. (Aronson, 1990)
A cleft of the lip, alveolus and palate also initially involves the incorrect development of the
intermaxillary segment. The secondary palate is formed by the two horizontal projections of
mesoderm from the inner surfaces of maxillary prominences called lateral palatine processes
(Aronson, 1990) (see Figure 2-3). The malformation of the inter-maxillary segment can
subsequently cause an interrupted fusion of these palatal shelves in the 8-10th gestation
5
week, resulting additionally in a defective secondary palate. The complete lip, alveolus and
palate clefts are the most common (50-70%).
Figure 2-3 Embryonic Development from the 7th until the 9th Gestation Week (Aronson, 1990)
p. 218
Even without the malformation of the intermaxillary segment, an inadequate fusion of the
palatal shelves can take place. The development of the isolated cleft palate is often due to a
post fusion disturbance of the secondary palate (Goss, 1977). Previously merged ectoderm
tissue can tear and a secondary cleft is the result. However, an isolated palate cleft can also
be due to a disturbed fusion of the mesoderm (lateral palatine processes), causing a primary
cleft (Hemprich, 2011). The later the merging is inhibited, the less severe is the defect. The
merging progresses from anterior to posterior and is complete in the 12th gestation week. The
anterior part forms the hard palate while the posterior part develops into the soft palate and
uvula (Stanier, 2004). Depending on the location of the disorder, the soft and/or hard palate
can be affected. The involvement of the soft palate plays a crucial role in the speech and
hearing impairments of the CLP patients, therefore the anatomy will be discussed in more
detail below.
6
2.4 Anatomy of the Soft Palate
Anatomical knowledge of the soft palate is necessary to understand the cause of various
disabilities of the cleft patients and also to gain insight on the current operation techniques
applied. The soft palate is movable and consists of muscle fibers sheathed in a mucous
membrane. It forms a dynamic boundary between the oral cavity and the nasal cavity,
therefore responsible for closing off the nasal passages during the act of swallowing and for
closing off the airway. Consequently, functioning muscles of the soft palate are essential for
normal articulation, hearing and feeding. The soft palate consists of five paired muscles and
a central aponeurosis. The five muscles, which are involved in the movement of the soft
palate, are: m. levator veli palatini, m. tensor veli palatini, m. palatoglossus, m.
palatopharyngeus and m. constrictor pharyngeus superior.
2.4.1 The Muscles of the Soft Palate
All five muscles are important in effectively sealing off the pharynx thereby preventing a
velopharyngeal inadequacy and resulting speech disorders (see Chapter 2.6.1). The
intravelar termination of the m. palatopharyngeus und levator veli palati forms the posterior
part of the soft palate. Their simultaneous contraction raises and lengthens the soft palate
and therefore the normal functioning of these muscles are essential for velo-pharyngeal
competence. M. tensor veli palatine tenses the soft palate whilst m. palatoglossus
simultaneously lowers the velum and elevates the tongue upwards and backwards. The
tensor veli palatine muscle is also thought to have an additional very important function.
This muscle is held responsible for the opening of the Eustachian tube and is therefore
necessary for middle ear ventilation (Hartzell, 2010; Wohlleben, 2004). (see Chapter 2.7.1)
2.4.2 The Significance of the “Muscular Rings” of the Soft Palate
The levator and tensor veli palatine muscles meet in the middle of the soft palate, forming a
muscle sling. They are also connected at the soft palate with the palatoglossal and
palatopharyngeal muscles. In forming these connecting “muscular rings or sphincters”
(Anastassov, 2001) from one side to the other and from cranial to caudal (see Figure 2-4),
the muscles provide a firm hold for each other during action. A cleft palate can disrupt this
functional equilibrium. If the described muscular rings are sectioned, as is often the case in
cleft palate patients, this important support mechanism breaks down. The importance of the
correct anatomical reconstruction of these muscular rings, coined by Kriens (1970) as an
7
intravelar veloplasty, has been highlighted in recent studies (Hassan, 2007). The surgeon
has to utilize very subtle operation techniques to recreate the disrupted functional
equilibrium.
Figure 2-4 Muscles of the Soft Palate. (Kummer A., 2000)(p. 14)
2.5
Consequences of a craniofacial cleft
Craniofacial clefts should not only be perceived as a problem to be solved simply through a
surgical closure of the cleft. A wide variety of complications accompany these morphologic
anomalies, resulting in aesthetic and functional disorders. These can be differentiated into
primary and secondary disorders (Rosanowski, 2002). Primary disorders include for example
misalignment of teeth and jaw, dysphagia and problems of nutrition and breathing. After
surgical treatment, the primary disorders are generally corrected. Secondary disorders such
as complex speech and voice disorders (Neumann, 2000), hearing loss (Schönweiler R.,
1999) and psychological problems are often still present after surgical closure of the cleft.
This study particularly engages in the speech and hearing disabilities of the cleft patients and
therefore these topics will be discussed in more detail below.
2.6
8
Linguistic Aspects of CLP Patients
The area of speech and language has been an integral part of cleft palate care from the very
beginning (Kuehn, 2000). Etiological factors contributing to speech production problems in
CLP patients are numerous, including for example velopharyngeal inadequacy, oronasal
fistulae, impaired lip movement, dental-occlusional status, tongue posture and mobility,
mislearning and perceptual deficits. Therefore, even after adequate surgical and nonsurgical
treatment, the effects of a CLP on speech can be manifold. The most important linguistic
aspects include (Kuehn, 2000):

Resonance: rhinolalia- audible nasal emission and altered vocal timbre.

Articulation disorders: for example shift in localization of articulation and a modified
articulatory tension (such as the weakening of the plosives /t/, /k/, /p/).

Voice and other speech disorders: for example breathy voice quality, hoarseness,
unusual pitch and reduced loudness.
2.6.1 Velopharyngeal Inadequacy
To understand the cause of speech disorders in cleft patients, it is important to be familiar
with the effects of a velopharyngeal inadequacy (VPI) (Schuster M., 2012; Vogt, 2007).
The resulting key problems of a VPI, as stated by Aronson (1990), are
“the isthmus between the oral and nasal cavities must be closed during swallowing
and speech, or else food will be regurgitated out of the nares, vowels will be
excessively resonated in the nasal chambers, pressure consonants will lose their
characteristic explosive and friction noises, and air will be audibly emitted through
the nares.”
VPI is defined as the insufficiency of the velum and the involved muscles to effectively seal
off the pharynx. In the case of a CLP-patient, the child is either born with a velum that is
too short, or after the cleft operation, scars can cause the velum to contract, also resulting in
a shortened velum. Clefting involving the palate can therefore often cause the incomplete
separation of the nasal cavity from the oral cavity. Reports estimate that despite primary
palatal repair, 20% to 43% of these children will still have velopharyngeal insufficiency
(Cable, 2004). Hence, children with a cleft affecting the palate have a high risk of reduced
9
intelligibility. Further difficulties which can be caused by a VPI, concerning breathing,
swallowing, sucking and hearing disorders (Hirschberg, 2006), are also not to be neglected.
The increased air flow resulting from a VPI is the main cause of the primary speech
disorders listed above: rhinolalia, articulation disorders, and voice disorders (Kummer A.
W., 2012). These ultimately lead to speech disorders and hence altered intelligibility. In
case of a severe hypernasality, pharyngeal flap surgery is a possible therapeutic option,
although treatment indications are uncertain and disputed. In a study undertaken by
Kummer et al. (2012) most surgeons (79.2%) reported that the surgical decision is based on
the results of an endoscopy and/or video fluoroscopy.
2.6.2 Rhinolalia
Rhinolalia (nasality) is the speech disorder most commonly associated with cleft patients.
The naso-oral imbalance of resonance is often due to a VPI, caused by a structural
inadequacy or functional incompetence of the velum (Bressmann T. , 2000). The intensity
and quality of the nasality depends primarily on the size of the cleft and the spatial
conditions of the nasal cavity and paranasal sinuses.
Counteracting a nasal speech is problematic and done in several ways (Warren, 1986). In
some cases the child can improve his nasal speech himself, in other cases an operation such
as a velopoharyngoplasty can be undertaken to eliminate nasality through correction of a
VPI. However, the child’s own compensation mechanisms often lead to further problems,
e.g. formation of similar but yet different phones through a shift in location of the
articulation, which can result in a chronic speech disorder and a lower rate of speech.
Occasionally these compensation mechanisms can also cause the face to move in mime-like
expressions that border on grimaces. Mimic muscles around the mouth, nose and even
forehead are engaged in counteracting the pathological airflow. As a result, the disorder
cannot only impede speech and language development but also negatively influence the
self-concept.
2.6.3 Articulation disorders
VPI is not the only cause of articulation disorders in cleft patients. Other etiologies
responsible for the speech deficits in patients with a cleft of the lip/alveolus are for example
impaired movement of the upper lip or dental abnormalities (Wohlleben, 2004). These
10
modifications can lead to a patient’s inadequacy to speak clearly and may ultimately cause
additional difficulties in the communication of the child. Even with adequate operative
results of the cleft, these articulation disorders can develop and are target for further
conservative or surgical treatment.
2.6.4 Speech Therapy
After palate surgery, yearly audiological and speech follow up examinations are carried out
through various specialists, including ENT doctors, pediatricians, speech therapists and
pedaudiologists. Depending on the findings, grommets and/or speech therapy can be
recommended. Based on the records in Freiburg, almost all CLP patients in this study
underwent at least temporary speech therapy. Unfortunately however, the records are not
exact on how many sessions of speech therapy the children received, and how long they
were carried out. The records in the phoniatrics department in Freiburg may be incomplete
due to the different specialties involved that prescribe the speech therapy. Seventeen of the
126 children investigated in this study also visited special schools for children with speech
defects.
2.7
Hearing Impairment of CLP Patients
The association between hearing loss and a cleft palate is well established (Bennett, 1972;
Bluestone C. D., 1971; Paradise, 1975). The cause of hearing problems in cleft patients is
mainly due to the fissure in the muscles of the soft palate and/or their displaced attachment.
The result is an impaired opening of the Eustachian tube which can cause frequent
unfavorable middle ear conditions of the cleft patients (Paradise, 1975). As the anatomical
and physiological integrity of the hearing system, auditory pathway maturity and sound
stimulation are essential for the acquisition and development of verbal language (Amaral,
2010), adequate cleft treatment is especially important.
2.7.1 Middle Ear Disease and the Eustachian Tube
Middle ear disease is very common in all children. The cumulative incidence at the age of
three reaches up to 80% (Teele, 1989). Nevertheless, otitis media with effusion (OME3) and
3
OME: is defined as a middle ear effusion without signs or symptoms of an acute infection.
11
acute otitis media (AOM4) have been proven to be even more common in cleft palate
patients. The incidence of OME has been reported to be greater than 90% (Goudy, 2006;
Grant, 1988; Muntz, 1993; Stool, 1967) prior to palatal closure.
The traditionally accepted explanation for the high prevalence of middle ear disease in cleft
patients is that the cleft interferes with the ability of the Eustachian tube to open (Leuwer,
2003; Peterson-Falzone, 2010; Sehhati-Chafai-Leuwer, 2006). The Eustachian tube, also
known as the auditory tube, connects the tympanic cavity with the nasopharynx. It opens
into the pharynx approximately at the level of the inferior nasal concha. The Eustachian
tube is normally closed, and opens for example through yawning or swallowing. The
musculature of the veolopharyngeal system is intimately tied to that of the Eustachian tube;
musculus tensor veli palatini (see Figure 2-5) is deemed necessary for its opening. Precisely
this muscle is often affected in CLP patients (Peterson-Falzone, 2010). The resulting
functional obstruction of the tube inhibits mucus drainage and the equalizing of pressure
between ear and atmosphere. A negative pressure in the middle ear can develop causing an
accumulation of secretions in the tympanic cavity and drawing bacteria from the
pharyngeal opening of the Eustachian tube, through the tube and into the middle ear cavity.
An OME is the undesirable result. The fluid present in the middle ear creates an optimal
medium for bacteria and an acute bacterial infection (AOM) can therefore also develop.
Occasionally the infection can also spread to the inner ear causing further damage.
4
AOM: an acute inflammation of the middle ear, usually with a rapid onset and short duration. Acute otitis
media is typically associated with fluid accumulation in the middle ear, together with signs or symptoms of
an ear infection.
12
Figure 2-5 Opening of the Eustachian Tube: the tensor veli palatine muscle pulls the lateral
hooked cartilage away from the membranous wall of the tube. (Peterson-Falzone, 2010), p.208
These unfavorable middle ear conditions in CLP patients cause a high probability of
hearing problems, both on a temporary or long-term basis. The presence of fluid in the
middle ear, as well as a change in the compliance of the tympanic membrane, may lead to a
short term conductive hearing loss of 20-50 dB (Schönweiler R, 2006). This is due to
interference in the transmission of sound vibrations to the inner ear. Although the rate of
spontaneous resolution is high in all children, some may go on to develop a chronic otitis
media with various structural changes: tympanosclerosis, segmental atrophy and
retractions, cholasteoma and perforations (Barfoed, 1980). Those few children who suffer
from chronic otitis media and eventually structural changes of the inner ear may have to
endure a prolonged hearing disability. Bennett (1972) states that approximately 50% of
congenital cleft lip and/or palate individuals in his study continued to have ear problems as
adults, and probably for their lifetime.
2.7.2 Hearing as a Prerequisite for Speech Acquisition
Children with cleft palates eventually recover normal Eustachian tube function after
palatoplasty, but for the majority of children this does not occur for many years (Smith,
1994). It is essential to keep in mind that a defective function of the Eustachian tube, at an
early age, can be disadvantageous. The consequences of an early hearing disability may be
as mild as inattentiveness of the CLP child or, if persistent and severe, may involve long
term effects on speech, language, emotional and cognitive development (Paradise, 1975).
13
The normal child acquires language through a gradual coordination of sensory input and
central processing. This is a simplification of a complex process and is far from being well
understood (Bergstrom, 1978). The reduction in the conductive hearing level (sensory
input) of the cleft children results in an insufficient relay of information to the central
auditory system. However, this exchange of information is essential for the auditory system
to develop and mature appropriately over the years and may be absent in CLP patients.
Even if the auditory system does develop correctly, continuous conductive hearing losses
can still have a negative effect on speech acquisition. We must hear each other’s speech in
order to learn a language correctly. Hearing allows the child to monitor and make
improvements in his speech, as hearing provides important feedback to the speaker. After
the age of school enrolment, delayed development of speech in cleft children is difficult to
compensate (for example through speech therapy) and the consequence may be a
permanent speech deficit. Adequate hearing at an early age is therefore an essential
requirement for appropriate speech acquisition.
2.7.3 Treatment of Middle Ear Disease
Countless options for the treatment of middle ear disease are available, the most common
medical options being the use of decongestants, mucolytics, steroids, antihistamines and
antibiotics. Surgical treatment alternatives include myringotomy (puncture of the tympanic
membrane), grommet insertion and adenoidectomy (Lous, 2005).
To avoid hearing impairment in cleft patients, it is often decided on a myringotomy or an
insertion of a grommet at a very early age. However, indication and optimal time for
surgical treatment are yet to be established and are momentarily controversially disputed
(Bluestone C., 1996; Browning, 2010; Maw, 1999; Moore, 1986; Paradise, 1974; Paradise,
2005; Ponduri, 2009; Rach, 1991; Robson, 1992; Shaw R., 2003; Valtonen, 2005). Under
certain circumstances, even multiple myringotomy or grommet insertions may be
considered necessary.
2.8
Interdisciplinary Therapy of CLP Patients
The care of a patient with cleft lip and palate is part art and part science (Peterson-Falzone,
2010). The treatment of a cleft is a complex process with the ultimate goal of a perfect
anatomical, functional and aesthetic reconstruction of the lip, alveolus and palate (Schultes,
14
2000). Optimally, the patients should receive interdisciplinary treatment in specialized
hospitals and a complete follow up from birth until adulthood. Follow up examinations are
required at least once a year until bone growth is completed (usually at 18 years or older). In
the course of this extensive treatment, various medical specialties are involved to ensure a
sufficient medical rehabilitation, most importantly maxillofacial surgery, otolaryngology,
pediatrics, plastic surgery, orthodontics, gynecology, phoniatrics and pedaudiology, speech
therapy, psychology and human genetics (Maier, 2009). To facilitate treatment and ensure
continuity of care, most centers have developed a multidisciplinary craniofacial anomalies
team providing comprehensive health care in a setting that is convenient for both parents and
health care workers.
Surgical closure of the cleft generally shows good results, but the effect on speech outcome
varies. This may possibly depend on the surgical method used (Van Lierde Kristiane M.,
2004). The treatment concepts in Freiburg and in Erlangen will be discussed, as the speech
outcome of the patients are compared in the results.
2.8.1 Outline of the Treatment Concept of CLP patients:
The birth of a child with a CLP requires immediate attention, as the child and parents are in
need of care and guidance. Complete surgical repair of a CLP generally occurs within the
first 18 months and consists of a lip operation, and if necessary, an operation of the palate.
Multiple methods are in use for the lip and the palate operation depending on the cleft type,
but also on the surgeon/hospital. In most European treatment centers the lip operation is
undertaken at the age of four to six months (Hausamen J. E., 2003). At this age the lip is
sufficiently developed and the extensive reconstructive procedure, closure of the nasal floor
and a rhinoplasty can be carried out without difficulty.
Timing and operation method of the cleft palate operation vary significantly in each
treatment center, as diverse concepts with various strategies exist. Most CLP centers in
German speaking countries undertake the palate operation at the age of 12 to 18 months, as
at this age adequate anatomical conditions are necessary for appropriate language
development. However, recent studies indicate a widely accepted tendency towards early
surgical repair of palatal clefts, i.e. between 5 and 12 months of age (Dorf, 1982; Hausamen
J. E., 2003). It is well accepted that such an early palatal closure is a basis for a more
physiological condition in the oral cavity, as it is mandatory for middle ear ventilation and
15
development of phonology (Schönweiler R., 1999). These factors are the foundation for
proper speech and language development. On the other hand, premature operation may
have negative effects on the growth of the maxilla (Ysunza, 2010). The age of the patient at
closure must therefore be considered carefully.
Treatment of a CLP does not only involve numerous operations, but also further medical
action.
Further
treatment
options
include
speech
therapy,
grommet
insertion,
velopharyngoplasty or a secondary osteoplastic. These additional medical treatments are
adjusted to the individual needs and disorders of the child. They ultimately affect the
speech outcome of the patients and therefore should be taken into consideration when
investigating intelligibility, as in the present study.
For over 30 years the interdisciplinary treatment concept in Erlangen has remained almost
unchanged (Wohlleben, 2004). This allows for a relative unproblematic evaluation of the
results when considering the speech and auditory outcome of the patients. In Freiburg, the
operation method was changed in 1979 (Meltzer, 2005). As none of the assessed children in
this study are born before this date, there is a low risk of distortion. The relevant treatment
concepts applied in Erlangen and Freiburg are outlined below.
2.8.2 Treatment Concept in Freiburg:
Treatment Concept of CLP patients in Freiburg
First few days
The parents are counseled and informed.
3rd -6th month
- The cleft of the lip is closed using the principles
of Delaire.
 Unilateral cleft: Millard Procedure
 Bilateral cleft: Veau III Procedure
- Nasal floor is formed and a rhinoplasty
undertaken in the same operation.
7-12th month
- The hard and soft palate is closed using the
Delaire operation method
- The hearing ability of the child is tested, and if
necessary, a tympanostomy tube is inserted.
End of the primary surgical treatment
Follow-up examinations every year regarding speech, hearing and
orthodontics. Depending on the findings grommets or for example speech
therapy can be prescribed.
7th year
Begin of the orthognathic surgery: closure of remaining
clefts between the oral and the nasal cavity
10th year
Correction of the jaw (secondary osteotomy)
th
12 year
Alignment of the upper and lower jaw if necessary
18th year
Further speech improving interventions if necessary
16
Table 2.8.2: Chronological overview of the Treatment Concept of CLP Patients in Freiburg
2.8.2.1 Lip Operation
In Freiburg, the closure of the lip cleft is performed at the age of 3 to 6 months. The mean
age is 5.2 months +/-1.2. In the same operation, the complete formation of the nasal floor
using the Veau-Axhausen method and a primary rhinoplasty is also undertaken. The cleft
alveolus is only closed temporarily during the lip operation with a gingivo-periostoplasty
and, if necessary, is later revised with a secondary osteoplastic during mixed dentition.
The operation technique of the lip/alveolus depends on the extent of the clefting: unilateral
or bilateral. Patients in Freiburg with a unilateral lip cleft are operated using the popular
Millard procedure (see Figure 2-6 and Figure 2-7), which is also known as the rotationadvancement technique. The first and most important step in the Millard repair is
identifying the landmarks and marking the key points used in the repair. The technique
repositions the laterally displaced alar base, restoring symmetry to the nostril and ala and
closing the floor of the nose defect using a columella flap. The incision technique also
allows easy access for primary rhinoplasty. The results are aesthetically appealing, with an
intact cupid’s bow and a scar that is vertically oriented and approximates the normal
philtral ridge. The Millard repair produces consistent results in the unilateral incomplete
cleft lip (Dolan 2004).
Figure 2-6 Millard Procedure, just before the closing procedure (Van de Ven, 2008)
17
Figure 2-7 Chronological Overview: Millard Procedure for Cleft lip Repair (Bitter, 2000)
CLP Patients with bilateral clefts are operated using the Veau III procedure (see Figure
2-8). The Veau III operation is a straight line closure. It is simple, forms a Cupids bow,
and is not difficult to revise. A disadvantage can be that the resulting lip can turn out too
short (Cronin, 1971).
18
Figure 2-8 Veau III Procedure: A- Lines of Incision, B- Sutured Bilateral Cleft (Cronin,
1971)
Further important aspects of the lip operation technique applied in Freiburg, which are
worth elaborating, include the simultaneous nose repair and the use of the functional
surgery principles of Delaire (Delaire, 1978a). There is always some degree of nasal
deformity associated with cleft lip/alveolus. The nasal deformity is produced both by the
embryological error that led to the cleft and by the deforming effects of inappropriately
inserted fibers of the orbicularis oris muscle. By releasing these inappropriately inserted
fibers, the columella and ala can be restored to a more natural position. Also, by
rearranging and reshaping the lower lateral cartilage, the surgeon is able to improve nasal
tip projection and create a more symmetrical nostril and ala.
Delaire postulates the importance of the reattachment of the three facial muscles
(perinasal, perioral, perimental) involved in the cleft according to their initial function (see
Figure 2-9) (Delaire, 1978b; Markus, 1992; Markus, 1993; Precious, 1993; Webb, 2001).
It is important that the perinasal muscles (pars transversa musculi nasalis) and the upper
parts of the musculus orbicularis oris are sutured to the nasal septum or the spina nasalis
anterior (Hemprich, 2011). The functional closure of the lip cleft using the Delaire
technique has the following advantages. It allows the restoration of an accurate muscle
function of the buccal and nasal region with the beneficial outcome of a normal maxillary
growth. The reconstruction of the lip muscle tissue provides a foundation for the adequate
movement of the mouth. The correct attachment of the muscles around the nose is also
significant, as a failure to do so could cause an inadequate growth of the premaxilla and
19
neighboring regions of the upper jaw (due to the lack of stimulus from the muscles and
periost) (Hemprich, 2006). Furthermore, excellent aesthetic results are achieved.
Figure 2-9 Schematic drawing of the three facial muscular rings (perinasal, perioral,
perimental) which are all disturbed by the cleft lip. Note the attachment of the muscles to the
primary growth centre (nasal cartilage) and the secondary growth centre (zygomaticomaxillary
suture). (Hemprich, 2006)
2.8.2.2 Middle Ear Treatment Indication
The need of a grommet (tympanostomy tube) insertion or myringotomy is determined in
Freiburg at the Department of the Phoniatrics and Pedaudiology; a conservative approach
is taken. Standard procedure is to conduct an otoscopic, audiometric and impedance
screening shortly before the palate operation, and also when it is deemed necessary during
the course of treatment. Subsequently, the audiometric data, otoscopic results, language
development of the CLP child and also the parents` impression of the child`s hearing are
all taken into account. A hearing loss of at least 15 dB is deemed as an operation
indication. After discussing the results in detail with the parents, it is decided if a
ventilation tube is necessary. The procedure of a grommet insertion is described briefly in
Chapter 2.8.4.
20
2.8.2.3 Palate Operation
The closure of the palate cleft is carried out at the age of 7 to 12 months. The mean age
during the palate operation is 10.6 months +/- 2.1. The goal of this operation is the closure
of the cleft without a remaining perforation and the reconstruction of a functional velum of
adequate length (Hausamen J., 2012). The Delaire method is applied in Freiburg. This
method is founded on the Langenbeck operation technique, which was developed in 1862
and described below in Chapter 2.8.3.4.
Following the principles of Delaire,
“the most important part of the closure is careful re-establishment of the continuity
and, therefore, the functioning of the soft palatal muscles” (Markus, 1993)
The restoration of the intact “muscular rings” of the soft palate (see Figure 2-10),
involving the tensor and levator palatini and palato-pharyngeus and –glossus, is therefore
obligatory.
a.
b.
c.
d.
e.
Tensor palati
Levator palate
Palatopharyngeus
Palatoglossus
Muscularis
uvulae
Figure 2-10 Muscles of the soft Palate: Left= cleft, Right= non-cleft
(Markus, 1993)
Figure 2-11 illustrates an outline of the surgical steps applied in Freiburg. Two bipedicle
mucoperiosteal flaps (bridge flap technique) are created by incising along the oral side of
the cleft edges and along the posterior alveolar ridge (A). The oral mucosal layer is
developed and separated from the muscles and nasal mucosa (B). The palatal muscles can
then be identified. The fibers of the transverse and levator veli palatine muscles are
oblique to the edges of the cleft and must be appropriately reoriented after dissection (C).
21
The nasal mucosal layer is closed and the muscles are repositioned transversely and
appropriately reattached. The palatoglossus and palatopharyngeus muscles are mobilized
bilaterally and advanced toward the midline, where they are then united (D). Lastly the
oral mucosal layer is also closed at the midline. (Anastassov, 2001)
A. Diagram of markings for
incisions for functional
palatoplasty (dotted line)
B. Diagram of elevation of
the oral mucosal layer
C. Diagram of elevation of
the muscle layer
D. Diagram of closure of the
palatal muscles including
the palatoglossus and
palatopharyngeus on the
midline after horizontal
reorientation
Figure 2-11 Delaire Operation Method. (Anastassov, 2001) p. 106
22
2.8.3 Treatment Concept in Erlangen
The treatment concept in Erlangen is similar to that of Freiburg, although a few
modifications are worth mentioning. The application of a palatal obturator is for example
considered beneficial, which is not the case in Freiburg. The operation techniques also
differ and are explained below.
Concept of Erlangen
First few hours
A palatal obturator is used to occlude the clefting. This helps
the nutrition and breathing of the child.
First few days
The parents are counseled and informed.
3rd- 6th month
- The cleft of the lip is closed.
 Unilateral cleft: Tennison Randall Procedure
 Bilateral cleft: Forked Flap technique
- The mucosa of the hard palate is closed: single layer
vomer-flap closure of the anterior nasal floor during lip
repair (Bilwatsch, 2006)
- A tympanostomy tube is inserted if required.
- The hearing ability of the child is tested.
10-15th month
- The soft and hard palate is closed using the
Langenbeck and Widmaier operation method.
- A tympanostomy tube is inserted if required.
End of the primary surgical treatment
Follow-up examinations every year regarding speech, hearing and orthodontics.
Depending on the findings grommets or for example speech therapy can be
prescribed.
th
7 year
Begin of the orthognathic surgery: closure of remaining clefts
between the oral and the nasal cavity
th
10 year
Correction of the jaw (osteotomy)
12th year
Alignment of the upper and lower jaw if necessary
th
18 year
Secondary osteotomy and further speech improving
interventions if necessary
Table 2-2 Chronological overview of the Concept of Erlangen. Adapted from (Wohlleben,
2004)
2.8.3.1 Palatal Obturator
In the first hours after the child`s birth, a palatal obturator is fitted and inserted to alleviate
the nutrition and breathing of the child (Wohlleben, 2004). In Freiburg the use of a palatal
obturator is regarded as an additional stress factor for the child and is not considered
substantially beneficial to be included in the standard therapeutic scheme.
23
2.8.3.2 Lip Operation
As in Freiburg, the lip of the CLP patient is operated at the age of 3 to 6 months. The
mean age is 4.1 months. There is no primary surgery performed on the nose during lip
repair. The hard palate (anterior nasal floor) is temporarily closed using a single layer
vomer-flap (see Figure 2-13) (Bilwatsch, 2006). The Tennison Randall operation method
is applied on a unilateral cleft (see Figure 2-12). The three layers of the lip tissue (mucosa,
muscles, skin) are, as in Freiburg, meticulously reattached. A symmetrical form of the lip
is achieved, with a harmonious cupid’s bow (Hausamen J. E., 2003).
Figure 2-12 Tennison Randall Procedure (Van de Ven, 2008)
Figure 2-13 Lip closure combined with a vomer flap closure of the hard palate. (Lehner, 2003)
24
Bilateral clefts are operated with a forked flap technique (see Figure 2-14). This approach
not only lengthens the columella, but also allows revision of bilateral lip scars and reduces
the wide prolabium to a more natural philtrum dimension (Strauch, 2009).
Figure 2-14 Forked Flap Procedure (Kastenbauer, 2005)
2.8.3.3 Middle Ear Treatment Indication
The indication for a
myringotomy or grommet insertion is also determined in the
phoniatrics and pedaudiological department of the University Clinic of Erlangen. The
procedure is similar to that in Freiburg.
2.8.3.4 Palate Operation
In Erlangen, the operation on the palate takes place at the age of 10 to 15 months, and
combines the Langenbeck and Widmaier operation methods (Dames, 2009). The mean
age of the CLP patients at the palate operation is 13.2 months (Bilwatsch, 2006). The
Widmaier procedure concentrates on the reconstruction of the soft palate. It can be used
to singularly repair the soft palate, or can be applied after closure of a combined soft and
hard palate cleft. The principle of this method is the mobilization of two extremely short
pedicle flaps. This allows the surgeon to retract and adapt the separated velum muscles
25
exactly, and also to displace the velum posteriorly. An outline of the incision technique is
shown in Figure 2-15: “a” is pushed back to “a`” in VY shape and “b” is pushed back to
“b`”. The cleft of the soft palate is subsequently sutured and the velum can be lengthened
by up to 2 cm using this operation method (Wiedmaier, 1991).
Figure 2-15 Widmaier Operation Method (Wiedmaier, 1991)
The Langenbeck procedure includes the repair of the hard palate and is briefly illustrated
in Figure 2-16. Two bipedicle mucoperiosteal flaps are created by incising along the oral
side of the cleft edges and along the posterior alveolar ridge (B). The flaps are mobilized
medially under preservation of the greater palatine arteries (C and D) (Patel, 2012).
26
Figure 2-16 Langenbeck Operation Method (Patel, 2012)
2.8.4 Myringotomy and Grommet Insertion
A myringotomy is a small incision of the eardrum, its main indication being a purulent
otitis media. This operation is often necessary in CLP patients. The incision is placed in the
lower quadrant (posterior or anterior) of the tympanic membrane and mucus can
subsequently be suctioned out. The defect in the eardrum generally heals quickly,
approximately within two weeks.
Is a permanent drainage of the middle ear necessary, a grommet can be inserted in the same
operation as the myringotomy. A grommet is a small plastic or metal tube constructed as in
Figure 2-17.
27
Figure 2-17 Grommet Insertion. (Donaldson, 1966)
The result is a “stented” tympanic membrane, allowing prolonged middle ear ventilation
and pressure equalization (see Figure 2-18). Removal of the grommet is rarely necessary as
the tubes typically extrude spontaneously.
Figure 2-18 Otitis media with effusion: multiple air/fluid levels. Same after treatment with
grommet. (Shaw R., 2003)
2.9
28
Diagnostic Methods for Pathological Speech
Background information on speech evaluation techniques and especially the PEAKS
program are essential to understanding and appreciating the results of this thesis. A clinical
examination of a speech disorder can answer questions as to type, severity and cause of the
disorder. In this investigation, PEAKS assesses the patients´ speech intelligibility, which
correlates to the severity of the disorder. The type and cause of the speech disorder is not to
be investigated.
Many methods to evaluate speech are found in the literature and can in general be divided
into two groups, perceptive and objective evaluation (Maier, 2009):

Perceptive evaluation is performed by a human subject. For the perceptive evaluation,
many different methods and scales can be applied. For example, the speech
intelligibility determined by the raters in this study uses an interval scale of 1 to 5.

Objective evaluation is obtained by a device or algorithm, which is independent of a
human rater. An example is the use of the PEAKS software as in this thesis.
2.9.1 Perceptive Evaluation
The current state of the art analysis of speech disorders involves the perceptive evaluation
through a single or multiple specialists and is often described as objective in the literature.
However, this method has several disadvantages. These are described in more detail below.
A single rater evaluation, as is common in clinical practice, can lead to the following
problems:

Different experience levels: Previous studies have shown that experience is an
important factor that influences the perceptive estimation of speech disorders. A
single rater evaluation can therefore lead to inaccurate evaluations by persons with
only few years of experience (Paal, 2005).

Inter-individual differences: The ratings have to always be performed by the same
labeler in order to be comparable, as one rater might observe more strictly or is more
lenient towards his patients. This can even be the case if the labelers have the same
amount of experience. The speech therapists assessing and treating the CLP patient
can therefore not easily be substituted. (Maier, 2009)

29
Intra-individual differences: Ratings of a single labeler can show considerable
variance and lead to intra-individual differences. Voice quality, phonetic and
morpho-syntactic structure, background noises, loudness and rate of speech can all
influence perception. Context and prosodic elements in particular contribute to a
better understanding of a human listener. These factors can therefore also limit the
reliability of the perceptual evaluation of speech (Keuning, 1999; Mayr, 2010).
Concluding, the method of using a single rater works quite well as long as the person (i.e.
therapist) sees the patient regularly, is experienced and is not exchanged. However, the
difficulty in comparing the subjective evaluations remains (Maier, 2009).
An evaluation with multiple raters is therefore to be preferred to single rater evaluations, as
calculating the average of the multiple raters can attenuate the high subjectivity (Maier,
2009). In clinical practice however, the multi-rater evaluation is rarely performed, as it is
time and manpower consuming. Such an effort is often only done for scientific purposes.
Thus, if panels of experts are too time-consuming and single experts are not reliable, an
objective evaluation method would undeniably be advantageous.
2.9.2 Objective Evaluation
Only a small variety of objective methods to evaluate speech exist, these often being
expensive, invasive, and/or also time consuming. Therefore these methods are clinically
seldom in use. Objective methods include the following:
1. Detection of articulation disorders: For example the Iowa Pressure Articulation Test:
IPAT (van Demark, 1977)
2. Detection of nasal emissions:
a. Mirror test: placing a mirror under the child’s nares and documenting the
presence or absence of condensation (fogging) through each pressure consonant.
Although clinically useful, a mirror examination is imprecise and provides only
gross information about nasal air escape.
b. Dental reflector: similar to the mirror test with the advantage that dental
reflectors clear rapidly and spontaneously; they do not have to be continually
wiped.
c. Nasometer: quantitive measurement of nasal emissions (Hogen Esch, 2004; Joos,
2006; Kuttner, 2003; Meltzer, 2005)
30
d. Nasopharyngoscopy, videofluoroscopy Cine MR imaging to detect a VPI
(Shprintzen, 1989; Silver, 2011).
3. Detection of secondary voice or speech disorders. (Bressmann T., 1998).
4. Further instrumental equipment: spectography, laryngography, electromyography,
electropalatography (Lohmander, 2004)
5. Intelligibility assessment: PEAKS. Until recently, the assessment of global
intellibility could not be sufficiently objectively quantified. PEAKS is an innovative
program which shows this potential. As yet, only a few studies are found in the
literature for the global assessment of speech intelligibility (Dames, 2009; Haderlein,
2009; Maier, 2009; Maier, 2006; Schuster Maria, Haderlein, 2006; Schuster M.,
2012; Vogt, 2007; Windrich, 2008).
2.9.3 PEAKS
The University of Erlangen developed an automatic speech recognition technique in 2006
as a project of the Deutsche Forschungs Gesellschaft (DFG). This new software enables a
straightforward objective assessment of speech intelligibility. It is a system, which can be
easily applied in the clinical daily routine, as it is accessed via Internet at any computer and
can be used without experts` knowledge. PEAKS does not require either special hardware
or software and therefore works on any standard PC with Internet access, a soundcard and a
microphone. An overview of the PEAKS system architecture is shown in Figure 2-19.
Figure 2-19 The architecture of the PEAKSlocal System. The normal PEAKS client is
connected to a simulated PEAKS server, which has just the ability to store new recordings.
Later on, the recorded data can be committed to the real PEAKS server. (Maier, 2009)
31
The idea of PEAKS is to use a word recognizer instead of a speech therapist as “listener”.
In order to simulate a naïve listener the authors apply a speech recognition system, which
was trained with normal speech of children and adults from the age of 10 to 40 from all
dialect regions of Germany (Maier, 2006). It is therefore independent from listener
perception but depends on automatic speech recognition (ASR). An ASR system is
software that analyzes the acoustic properties of spoken speech. To calculate the word
recognition (WR) value, PEAKS analyses speech by subdividing it into units and
comparing these with its internal “dictionary” (Schuster Maria, Maier, 2006). Words are
then chosen that with a very high probability correspond to the actual words spoken. The
WR is calculated as follows (Maier, 2009):
WR (%) = C/R x 100%
where C is the number of correctly recognized words and R is the number of words of the
reference text.
Hence, the recognition data (percentage of correctly recognized words) should resemble the
intelligibility of the speaker (Vogt, 2007). Intelligibility however also includes a “human
factor” (Vogt, 2007). As the WR value is therefore not exactly similar to intelligibility, the
resulting WR will be called “degree of intelligibility” (Schuster M., 2012) in the remaining
paper. If the recording conditions are kept constant, the speaker should be the only varying
factor, as influencing factors on the listener’s side such as individual expertise or familiarity
to the speech can be minimized.
In this investigation, PEAKS was used in combination with a semi-standardized test
commonly used by German speech therapists for children. The test named PLAKSS (Fox,
2002) is an abbreviation for “Psycho-linguistische Analyse kindlicher Sprechstörungen”
(Psycholinguistic Analysis of Children’s speech disorders). It is designed for the
assessment of speech disorders of children aging between 4 and 18 years old; the pictures
involved correspond with a vocabulary of a 2,5 year old. The test consists of 99
words/pictograms on 33 slides, which are to be named by the participant. Figure 2-20
shows an example of the slides; reference words are also included in the test as subtitles.
The vocabulary of the PLAKSS test can be reviewed in the Appendix (Chapter 9.3).
PEAKS displays the slides on a computer monitor, records simultaneously, and sends the
speech data for central analysis to the server via the Internet.
32
Figure 2-20 Example of a Pictogram Slide used in the PEAKS Program (Maier, 2006)
33
2.10 The Hypotheses of this Study
The following hypotheses were established:

Hypothesis 1: Concerning the speech intelligibility assessments:
a) The mean word recognition (WR) values are expected to be lower in the CLP
patient collective compared to that of the control group consisting of non-cleft
school children.
b) It is to be expected that the most experienced rater (linguist) shows the highest
correlation with the WR values of the PEAKS assessment. The untrained rater
(medical student) shows the lowest correlation with the WR values of PEAKS. The
trained raters (linguist and ENT doctor) show the highest consistency amongst one
another in rating the speech of the CLP patients.

Hypothesis 2: It is to be expected that the Word Recognition (WR) values calculated
by the PEAKS program are higher:
a) in patients without a palate cleft, compared to patients with a palate cleft.
b) in female patients compared to male patients.
c) in older cleft children compared to the younger cleft children.

Hypothesis 3: It is to be expected that the Word Recognition (WR) values calculated
by the PEAKS program are higher:
a) in patients operated in Freiburg, compared with cleft patients operated in
Erlangen.
b) in patients who underwent a conservative grommet treatment regime.
34
2.11 The Objectives of this Study
The present study focused on the following questions:

The ability of the PEAKS program to distinguishing between “good” and “bad”
speakers. Are the WR values of the CLP patients lower than that of the control
group?

The evaluation ability of human raters with different experience levels (trained and
untrained) compared with the PEAKS program and to one another. Does the speech
assessment of the trained rater have a higher correlation to the WR Value calculated
by the PEAKS program than the untrained rater? How do the subjective assessments
of the 3 different raters correlate amongst each other?

The influence of cleft type, gender and age on the degree of intelligibility of the CLP
patients. Can PEAKS generate the adequate speech results, which are expected from
prior research regarding these criteria?

The evaluation of the speech outcome of the CLP patients, regarding the treatment
received. Is there a significant difference in the degree of speech intelligibility of the
patients operated in Erlangen to those operated in Freiburg? Does the number and
timing of grommets inserted have an effect on the degree of speech intelligibility of
the cleft patients?
35
3 Materials and Methods
3.1 Patients
3.1.1 Data Source
After approval through the Ethics Committee, the desired CLP patient documents were
acquired in the archives of the Department of Phoniatrics and Pedaudiology at the
University Clinic of Freiburg. The patients were selected regarding certain criteria; only
CLP patients who were born between 1990 and 2004 were included. The resulting number
of children amounted to 126.
The period of examination for the PEAKS investigation was from March until December
2009. In this time period, 60 of the 126 selected CLP children were examined and recorded.
Three of the patients were excluded due to certain malformations or syndromes, whilst two
children who had undergone a velopharyngoplasty and 1 child with a polish mother tongue
were also excluded.
Further three were excluded due to unsuccessful recordings.
Ultimately 51 patients (mean age: 9.71 +/- 3,4) were assessed in the PEAKS results.
The control group used in this investigation was adopted from a study undertaken in
Erlangen (Dames, 2009). The control group consisted of 159 German speaking, non-cleft
students (mean age: 9.1 +/- 2.9 years) which were recorded at an elementary school and a
kindergarten in Erlangen.
In order to compare the different treatment concepts between Freiburg and Erlangen, the
PEAKS results of the CLP patients in Erlangen (Dames, 2009) were also utilized. The
Erlangen patient collective consisted of 72 children with the mean age of 8.7 +/- 3.0 years,
each suffering from a complete lip, alveolus and palate clefts. Of the PEAKS patients
analyzed in our study, only 25 suffered from a complete lip, alveolus and palate cleft and
could therefore be compared.
3.1.2 Data Processing
The CLP patients were subdivided into two groups to simplify the evaluation. Previous
studies (Schönweiler R., 1999; Schuster M., 2012) showed that patients with clefts of the
soft and hard palate had a significantly worse nasality/intelligibility than the patients only
36
with clefts of the lip and alveolus. Taking this into account, a detailed subdivision of the
patients was not deemed necessary. The patients were divided into: Group 1 consisted of
children without clefting of the palate and Group 2 consisted of the children with clefts of
the soft and/or hard palate.
The exclusion criteria for the patients of this study included children with a mother tongue
other than German, patients who had already undergone a velopharyngoplastic surgery and
also those with certain malformations and syndromes associated with clefting.
3.2
Procedure
3.2.1 Data Collection
Approximately 1500 patients partake in the interdisciplinary consulting hour of the Oral
and Maxillofacial Surgery Department at the University Clinic of Freiburg (Meltzer, 2005).
The first step of this investigation was to select candidates for the study. This was done out
of the archives of the Department of Phoniatrics and Pedaudiology. While examining these
medical documents, certain background information of 126 CLP patients was collected.
This information was recorded using a data entry form (see Appendix 9.1) with Microsoft
Excel 2007. The program allowed the simple evaluation and comparison of all the data
collected.
3.2.2 PEAKS Recordings
Approximately half (60) of these selected CLP patients were subsequently examined and
recorded in follow up examinations every Monday after visiting the interdisciplinary
consulting hour in Freiburg. The parents were informed orally about the study and provided
their written consent. They were also given a questionnaire (see Appendix 9.2) to complete,
making available missing information, which was not to be found in the medical records.
The CLP patients were recorded using a USB microphone headset, which was adjusted to
be in front of the child’s mouth (distance of approximately 20 cm). The procedure of the
recording was explained to the children. Some of the children who were tested were not yet
able to read. The test therefore consisted of pictograms and the corresponding words (see
Chapter 2.9.3). During the test, the speech therapist showed the pictograms of PLAKSS to
37
the child and encouraged it to say the names of the presented items. The recording took in
average approximately 4 minutes per child.
3.2.3 Speech Evaluation of the CLP Patients
Following these recordings and examinations, a word recognition rate (WR) for each CLP
patient was calculated automatically using the PEAKS program. The children’s speech was
also subsequently assessed individually and subjectively by three different raters (a linguist,
ENT doctor and medical student). This was done using the previously obtained and saved
recordings of the PEAKS program. The three words per slide could be played when
prompted. An interval scale from 1 to 5 was applied to rate the intelligibility of the three
words: 1- excellent, 2- good, 3- neutral, 4- poor, 5- unintelligible. The PEAKS program
subsequently would calculate the average of the 33 subjective evaluations for each patient
(as there were 33 slides per patient), and a floating-point value was computed to represent
his or her intelligibility.
3.2.4 Statistical Analysis
The statistical analysis in this study was carried out using SPSS Statistics Student Version
17.0. With help of the SPSS program, the mean and standard deviation of the WR values
were calculated. The correlation coefficients were determined, comparing the WR ratings
of PEAKS to those of the subjective intelligibility ratings, and also the subjective ratings
amongst one another. SPSS also enabled us to calculate whether the influence of the
independent variables (age, gender, group and number of grommets inserted) on the WR
value were statistically significant. This was done using the p values of a linear regression
analysis. The level of significance was set at p ≤0.05.
Whether a statistically significant difference existed between the WR values of the control
group and the patient groups, and also between the two different patient groups (Freiburg
vs. Erlangen), was calculated manually using the Welch’s t-test. The Welch’s t-test was
implemented in this case, because only the mean, standard deviation and number of
participants was known of the control group and patient group of Erlangen (adopted from
the study of Dames, Maier et al. (2009). In this test p values ≤ 0.05 were also recognized as
statistically significant.
3.3
38
Materials and Methods Analysis
3.3.1 Database
The data obtained from the archives were occasionally incomplete.
3.3.2 PEAKS Recording
The Oral and Maxillofacial Clinic of the University of Freiburg generously allocated the
project a quite room for the investigation. Only slight background noises occurred, and
occasional prompting from the examiner and breathing from the patient was noticeable. The
recordings were of various qualities. Difficulties arose, when the children were not able to
identify the pictogram. In addition, several children slowly lost interest and hence
concentration during the recordings. This could cause the PEAKS program to misrepresent
the actual degree of speech intelligibility and hence WR value.
It is also known that other factors such as the speaker and his native tongue, the speaking
style, vocabulary, grammatical complexity and acoustic properties of the recording system
can greatly affect an automatic speech recognition (ASR) system (Mayr, 2010). In the
present study, children with a mother tongue other than German were excluded. Also, a
standardized procedure was used in order to exclude the above-mentioned confounding
factors; by using a standardized test (PLAKSS) and stable recording conditions, the impact
of most of these factors on the ASR was minimized. Thus, the speaker remains the main
factor of influence as required for the evaluation of intelligibility (Kent, 1992).
3.3.3 Subjective Speech Intelligibility Assessment
The use of the PEAKS program enabled a simple subjective evaluation of the children’s
speech, as the recordings were saved and could be replayed when prompted. The
falsification of the results is reduced by the fact that only single words were used, and
contextual aids were not involved. These aiding words can often be used by raters to
understand what the patients are saying, especially when the speech is poorly intelligible
(Wohlleben, 2004). The subjective factor- meaning the interpretation and processing of the
perceived speech- is therefore kept to a minimum. The accuracy of the evaluation is also
enhanced, due to the fact that each slide is rated individually, and not the whole recording
at once.
39
3.3.4 Improving the Procedure
A suggestion for improvement would be to apply the PLAKSsingle Test, which was used
with one patient in this study. The slides in this test consisted of only one pictogram, which
allowed a swifter recognition of the pictures, and therefore did not need as much prompting
and encouraging. It also enables the subjective evaluation of each word separately, and not
the evaluation of 3 words at once (as on each slide in the normal PLAKS test).
A further suggestion would be to change the 1-5 interval scale used for the subjective
evaluation of the CLP children’s speech, to for example a 1-10 point interval scale. This
would minimize the bias of rating the children in a (German) school system fashion and
therefore may make the ratings more reliable.
40
4 Results
4.1 Overview of the Patients
4.1.1 Total Number of Patients (126 Patients)
Group 1
Group 2
Cleft Type
Facial cleft
Isolated cleft lip
Isolated cleft lip, alveolus
Isolated cleft palate
Cleft lip, alveolus, palate
Other
Number of Patients
1
24
9
14
36
102
60
6
Percent
19%
81%
Table 4-1 Number of Patients according to Group and Cleft Typ. (Group 1: Patients without a
palate cleft. Group 2: Patients with a palate cleft.)
4.1.2 Patients with Additional Illnesses
Additional Illness
Holoprosenzephaly
Turner syndrome
18 Q Syndrome
Complex Syndrome
Van der Woude Syndrome
Goldenhar Syndrome
Down Syndrome (1x PEAKS)
Teratoma (1x PEAKS)
Epilepsy (1x PEAKS)
Embryopathy (1x PEAKS)
Heart defects
Additional Illnesses in all
Number of Patients
1x
2x
3x
18 Patients = 14% of all Patients
Table 4-2 Number of Patients with Additional Illnesses
41
4.1.3 Timing of the Cleft Operations in Freiburg
Lip OperaPon Number of PaPents 30
25
20
15
10
5
0
1
2
3
4
5
6
7
8
>12
Age of PaPent at OperaPon (in Months) Figure 4-1 Timing of Lip Operation in Freiburg: 126 patients assessed. (Age of 3 Patients
unknown)

Mean age of the CLP patients at the time of the lip operation: 5.2 months +/- 1.2
months (excluding the exceptions who were operated on at an age of >12 months).
Palate OperaPon 30
Number of PaPents 25
20
15
10
5
0
6
7
8
9
10
11
12
13
14
15
16
17
18
19
>
24
Age of PaPent at OperaPon (in Months) Figure 4-2 Timing of Palate Operation in Freiburg: 126 patients assessed. (Age of 5 Patients
unknown)

Mean age of the CLP patients at the time of the palate operation: 10.6 months +/- 2.1
months (excluding the exceptions who were operated at an age >24 months).
4.2
42
PEAKS Assessment
4.2.1 Patients Included in the PEAKS Assessment
Sixty patients were investigated in the PEAKS assessment. However, the PEAKS
recordings of 3 Patients were unsuccessful and 3 patients were excluded due to additional
illnesses. 1 patient was excluded due to a polish mother tongue and finally 2 more due to a
velopharyngoplasty. As a result only 51 patients were assessed in the results.
Group 1
Group 2
Cleft Type
Isolated cleft lip
Isolated cleft lip, alveolus
Other
Isolated cleft palate
Cleft lip, alveolus, palate
Number of Patients
3
10
7
1
41
14
26
Percent
20%
80 %
Table 4-3 Number of Patients according to Group and Cleft Type
4.2.2 Comparison of the objective and subjective Speech Intelligibility
Assessment
Comparison of WR
with different Raters:
WR (%) with
Medical student
WR with
ENT Doctor
Mean +/- Standard
deviation
Median
51.52 +/- 14.51
1.67 +/-0.66
53.54
1.48
1.56 +/- 0.44
1.46
1.90 +/- 0.72
1.7
r = correlation
coefficient
WR with
Linguist
-0.65
-0.82
-0.82
Table 4-4 Table comparing the Speech Intelligibility Evaluation (of a medical student, an ENT
Doctor and a linguist) with the estimated WR Values of the PEAKS Program

The linguist’s (most experienced rater) intelligibility rating shows the strongest
correlation with the WR value of the PEAKS program (r = -0.82).

The medical student’s (inexperienced rater) intelligibility rating shows the weakest
correlation with the WR value of the PEAKS program (r=-0.65).
43
Comparison of WR Values to Intelligibility RaPng of the Linguist 100
90
WR Value (%) 80
R = ‐0.82 70
60
50
40
30
20
10
0
0
0,5
1
1,5
2
2,5
3
3,5
4
4,5
5
Intelligibility RaPng Linguist Figure 4-3 Regression Analysis comparing the WR Values and the Linguist’s Speech
Intelligibility Rating
Comparison of WR Values to Intellibility RaPng of the Medical Student WR Value (%) 100
90
80
70
60
50
40
30
20
10
0
R = ‐0.65 0
0,5
1
1,5
2
2,5
3
3,5
4
4,5
5
Intelligiblity RaPng Medical Student Figure 4-4 Regression Analysis comparing the WR Values and the Medical Student’s Speech
Intelligibility Rating

44
The negative correlation between the variables (WR Value and Intelligibility rating)
is made apparent in the regression line of the above presented graphs. The
coefficients are negative because a high recognition rate comes from “good” speech
with a low subjective score number (For example: 1) and vice versa.

It is interesting to note that the inexperienced rater (medical student) tended to give
better intelligibility ratings than the experienced (linguist).
4.2.3 Comparison of the subjective Intelligibility Assessments of the
different Raters- Linguist, ENT Doctor and Medical Student
Comparison of
different Raters:
ENT Doctor with
Medical student
Mean +/- Standard
Deviation
1.56 +/- 0.44
1.67 +/- 0.66
Median
1.46
1.48
Linguist with
Medical Student
1.90 +/- 0.72
1.67 +/- 0.66
1.70
1.48
0.83
ENT Doctor with
Linguist
1.56 +/- 0.44
1.90 +/- 0.72
1.46
1.70
0.93
r= correlation coefficient
0.89
Table 4-5 Comparing the Speech Intelligibility Evaluation of different Raters

The ENT doctor and the Linguists evaluation of the speech intelligibility of the cleft
patients showed the strongest correlation to one another (r = 0.93).

The Linguist and the Medical students evaluation of the speech intelligibility of the
cleft patients showed the weakest correlation to one another (r =0.83)
45
Comparison of Speech Intelligibility of Cle_ PaPents using different Raters: ‐ Linguist vs Medical Student Speech Intelligibility Value: Linguist 4
3,5
3
R= 0.83 2,5
2
1,5
1
0,5
0
0
0,5
1
1,5
2
2,5
3
3,5
4
4,5
5
Speech Intelligibility Value: Medical Student Figure 4-5 Regression Analysis comparing the medical Student’s and Linguist’s Speech
Intelligibility Rating.
4.2.4 Word Recognition Rate considering Group, Gender and Age
All Freiburg
CLP Patients
Group 1
Group 2
Control Group
(Dames 09)
Number of
Patients
51
WR Value (in %)
Arithmetic
Lowest
mean +/- SD
Value
13.13
51.51 +/- 14.5
Highest
Value
81.82
Age (Years)
9.7 +/- 3.4
10
41
46.77 +/- 12.4
52.67 +/-14.8
27.27
13.13
68.69
81.82
8.7 +/- 3.0
10.0 +/- 3.4
159
63.5 +/- 12.1
31.3
85.9
9.1 +/- 2.9
Table 4-6 WR Values according to Group

Group 1 achieved an insignificantly lower mean WR value compared to the Group 2
patients (p=0.228).

The mean WR of the Freiburg CLP patient collective is significantly (p <0.001)
lower than that of the mean WR of the Erlangen Control group.

46
The WR results of both Group 1 (p=0.005) and Group 2 (p<0.001) are both
significantly lower than the control group.
Male
Number of
Patients
34
Female
17
55.98 +/- 16.3
Male
84
63.9 +/-11.1
9.1 +/- 3.0
Female
75
63.2 + 13.3
9.1 +/- 2.9
Gender
CLP
PEAKS
Patients
Control
Group
(Dames 09)
WR Value (in %)
Arithmetic
Lowest Highest
mean +/- SD
Value
Value
49.28 +/-13.2
13.13
68.69
26.26
81.82
Age (Years)
9.7 +/-2.9
9.8 +/- 4.3
Table 4-7 WR Value according to Gender

The female patients in Freiburg achieved a slightly higher mean WR value of 55.98%
+/-16.3% compared to the male (49.28% +/-13.2%), although the gender influence is
not significant (p= 0.155).
Age
4
5
6
7
8
9
10
11
12
13
14
15
16
19
Number of
Children
3
4
3
4
4
5
8
6
5
5
4
1
1
1
WR VALUE (in %)
Arithmetic mean +/Median
Lowest
Standard Deviation
Value
41.75 +/- 13.7
43.43
27.27
26.51 +/- 10.1
27.78
13.13
37.00 +/- 8.6
34.34
30
48.23 +/- 10.3
46.97
38.38
51.01 +/- 9.2
51.52
39.39
52.53 +/- 13.4
51.51
39.39
51.64 +/- 9.6
51.01
36.36
59.80 +/- 5.5
57.58
54.55
62.63 +/- 14.5
68.69
37.37
72.73 +/-10.9
75.76
60.61
55.30 +/- 12.3
56.57
39.39
41.41
41.41
54.55
54.55
64.65
64.65
Highest
Value
54.55
37.37
46.67
60.61
61.61
71.72
67.68
65.66
73.74
81.82
68.69
Table 4-8 WR Values according to Age

A general increase in the WR can be distinguished, corresponding to the age of the
patient. The lowest mean value is shown at the age of 5 with 26.51% and the highest
at 13 years old with a median WR of 72.73%.

The mean WR value of children <8.5 years of age (40.40% +/- 13.5%) was
significantly lower (p <0.001) than those older than 8.5 years, who achieving a mean
WR value of 56.60% +/- 12.0%

47
The 4-year-old children did not follow the general trend, achieving a higher mean
WR value than expected of 41.75% +/- 11.2. The patients older than 13 years old also
did not follow the general trend, achieving lower WR values than expected.

The 12-year-old patient with a WR Value of 37.37% (shown as the red circle on the
box plot below) showed a marked nasality during the speech recording.
Box Plot of WR Values according to the Age of the Cle_ PaPent 100
90
80
70
WR Value (%) 60
50
median
40
30
Pakent
showed
a
marked
nasality
during
PEAKS
recording.
20
10
0
4
5
6
7
8
9
10
11
12
13
14
15
16
19
Age of the CLP PaPent (in years) Figure 4-6 WR values according to the Age of the Cleft Patient
4.2.5 Word Recognition Rate considering Operation Method
Erlangen vs. Freiburg (only Patients with complete lip, alveolus and palate clefts)
72
25
Age (in years) +/SD
8.7 +/- 3.0
10.3 +/- 3.3
WR (in %)
Mean +/- SD
48.1 +/- 16.3
51.3 +/- 15.3
159
9.1 +/-2.9
63.5 +/- 12.1
Operating Hospital
Number of Patients
Erlangen (Dames 09)
Freiburg
Control Group Erlangen
(Dames)
Table 4-9 WR values considering Operation Method
48
 The patients operated in Freiburg achieved a slightly better WR result compared with
those operated in Erlangen. This result was statistically insignificant (p = 0.381).
 The WR values of both the Freiburg and Erlangen CLP groups are significantly lower
than the control group (both p values <0.001).
4.3
Evaluation of Grommet Insertion in Freiburg
4.3.1 Overview of the Grommets Inserted in the 126 patients evaluated:
Were they placed during or after the Palate Operation?
Total # of patients=
126
Number of Patients with Grommet Insertion
Date of
During the
After the Palate
Insertion
Palate Operation
Operation
unknown
# of Patients
%
26
21%
32
25%
7
6%
Number
of
Patients
without
grommets
61
48%
Table 4-10 Grommet insertions in Freiburg: A conservative approach

26 out of 126 patients (21%) underwent a grommet insertion during the palate
operation. These children had substantial hearing deficits of at least 15 dB over more
than one frequency.

32 out of 126 patients (25%) required grommet insertion after the palate operation.

Almost half (48%) of the patients did not receive any grommets.

The date of grommet insertion was not specified in the medical records of 7 patients.
Number of Grommets Inserted
Group 1
# of Patients
% of Patients
18
75%
4
17%
2
8%
0
0%
Number of Patients
with Grommet
Insertion in all
6 of 24 Patients
25%
Group 2
# of Patients
% of Patients
43
42%
42
41%
14
14%
3
3%
59 of 102 Patients
58%
In All
# of Patients
% of Patients
61
49%
46
36%
16
13%
3
2%
65 of 126 Patients
51%
Group
0
1-2
3-5
6+
Table 4-11 Number of Grommets Inserted according to the Group of the Cleft Patient
49

In Group 1: only 6 of 24 patients (25%) underwent at least one grommet insertion.

In Group 2: 59 out of 102 patients (58%) underwent at least one grommet insertion.

In All: 65 out of 128 patients (51%) underwent at least one grommet insertion.

In Freiburg, the mean grommet insertion in all was 1.11 grommets (+/- 1.39) per
patient.
Group 1: Number of VenPlaPon Tubes inserted Group 2: Number of VenPlaPon Tubes inserted 6+
VT
3%
3‐5
VT
8%
3‐5
VT
14%
1‐2
VT
17%
0
VT
42%
0
VT
75%
1‐2
VT
41%
Figure 4-7
left: Pie Graph showing the Number of Grommets Inserted in the Cleft Patients of Group 1
right: Pie Graph showing the Number of Grommets Inserted in the Cleft patients of Group 2
4.3.2 Word Recognition Rate considering the number of Grommets
Inserted
Number of
Grommets
0
1-2
3-5
6+
Number of
PEAKS Patients
23
12
13
3
Mean Age
(Years)
10.1 +/- 4.0
9.4 +/- 2.8
WR Value (in %)
Arithmetic mean +/- Standard
Deviation
54.1 +/- 16.0
54.1 +/- 16.0
50.2 +/- 13.1
49.4 +/- 13.1
52.0 +/- 13.0
35.25 +/- 4.8
Table 4-12 WR Values according to the Number of Grommets inserted

50
The mean WR values of the children without grommet insertion is slightly higher
(54.1% +/-16.0%) to those who have had at least one grommet inserted (49.4% +/13.1%), although the difference is statistically insignificant (p= 0.650)

The patients with at least 6 grommets inserted achieved a considerably lower mean
WR value of 35.25% +/- 4.8% (mean age: 10.0 +/- 4.0) to the other patients. This
result is statistically significant (p=0.023).

In the box plot below, it is interesting to note that the patients who did not undergo a
grommet insertion, achieved a wide range of WR values. (min: 13.13% max:
81.82%)
Box Plot: The Influence of the Number of Grommets Inserted on the WR Value 100
90
80
70
WR Value (%) 60
50
median
40
30
20
10
0
0
1
to
2
3
to
5
6+
Number of Grommets Inserted Figure 4-8: Box Plot showing the Influence of the Number of Grommet Insertions on the WR
Value
51
The influence of the number of Grommets Inserted on the mean WR Value 60
WR Value (%) 55
50
45
40
mean
WR
35
30
0
1
to
2
3
to
5
6+
Number of Grommets Inserted Figure 4-9: Bar Chart showing the Influence of the number of Grommets inserted on the mean
WR value
4.3.3 Word Recognition Rate considering the Timing of the Grommet
Insertion
Patients with Grommet Insertion
Total # of patients= 51
During the Palate
Operation
After the Palate
Operation
# of Patients
%
Age
Mean WR Value (in %)
11
22%
8.9 +/-2.9
46.9 +/- 17.5
17
33%
9.6 +/- 2.7
51.0 +/- 9.6
Patients
without
grommets
inserted
23
45%
10.1 +/-4.0
54.1 +/-16.0
Table 4-13 The effect of the early or late grommet insertion on the CLP patients’ degree of
intelligibility: according to the achieved WR Value.

The mean WR of the children with early grommet insertion during the palate
operation is slightly lower (46.9% +/-17.5%) to those who have undergone a more
conservative operative treatment regime (51.0% +/- 9.6). This result is however
statistically insignificant (p =0.483).

The best results were achieved by the patients who did not have any grommets
inserted at all, with a mean WR value of 54.1% +/-16.0.
52
4.4 Summary: PEAKS Results considering the Objectives of the Study

The ability of the PEAKS program to distinguishing between “good” and “bad”
speakers. Are the WR values of the CLP patients lower than that of the control
group?
In the entire Freiburg patient collective of 51 patients, the word recognition rate (WR)
varies between 13,13% and 81,82% (arithmetic mean: 51.51% +/- 14.5%). The
control group (Dames, 2009) of 159 patients, achieved WR values between 31,3%
and 85,9% (arithmetic mean: 63.5% +/- 12.1%) The mean WR value of the control
group is significantly higher to that of the Freiburg patient collective (p <001).

The evaluation ability of human raters with different experience levels (trained and
untrained) compared with the PEAKS program and to one another. Does the speech
assessment of the trained rater have a higher correlation to the WR Value calculated
by the PEAKS program than the untrained rater? How do the subjective assessments
of the 3 different raters correlate amongst each other?
The WR correlated strongly with the subjective speech intelligibility evaluations,
with correlation coefficient (r) values reaching up to -0.82 (assessment of the linguist
and ENT doctor compared with the WR value). The medical student with a
correlation coefficient of -0.65 achieved the weakest correlation to the PEAKS` WR
values. The speech intelligibility assessments of the 3 different raters were also
compared among each other. The strongest correlation is shown between the
linguist’s and the ENT doctor’s evaluation of the cleft children’s speech (r= 0.93),
whilst the weakest was between the linguist and the medical student (r= 0.83)

The influence of cleft type, gender and age on the degree of intelligibility of the CLP
patients. Can PEAKS generate the adequate speech results, which are expected from
prior research regarding these criteria?
When comparing the WR values considering certain variables, results are obtained
which only in part correspond to previous studies. The Group 1 Patient collective had
a lower WR value (46.77% +/- 12.4%) than that of the Group 2 patients (52.67% +/14.8%), a result which conflicts with previous studies. Female patients had, as
expected, slightly higher WR values (55.98% +/- 16.3%) than the male patients
(49.28% +/- 13.2%). The difference in WR values regarding gender and the type of
53
cleft were not significant. Age, however showed the predicted influence: the WR
value generally improved with age.

The evaluation of the speech outcome of the CLP patients, regarding the treatment
received. Is there a significant difference in the degree of speech intelligibility of the
patients operated in Erlangen to those operated in Freiburg? Does the number and
timing of grommets inserted have an effect on the degree of speech intelligibility of
the cleft patients?
Lastly the WR values considering the treatment methods applied were compared. The
patients operated in Freiburg showed a slightly better overall result (50.7% +/14.6%) compared with those in Erlangen (48.1% +/- 16.3%), although statistically
insignificant (p= 0.381). The influence of a conservative approach to grommet
insertion on the degree of speech intelligibility of the CLP patients was also
investigated in this study. The WR value considering the number of grommets
inserted was evaluated. The mean WR value stayed relatively constant until the 6th
grommet insertion. The mean WR value of patients with 0-5 grommets insertions was
52.5% +/- 14.3%, whilst the patients who had undergone at least 6 grommet
insertions, achieved a significantly worse (p = 0.023) mean WR value of 35.25% +/4.8%. In addition, this study also evaluated the timing of grommet insertion on the
WR value. The mean WR of the children with early grommet insertion during the
palate operation was slightly lower (46.9% +/-17.5%) to those who had their first
grommets inserted after the palate operation (51.0% +/- 9.6%). The best results were
achieved by the patients who did not have any grommets inserted, with a mean WR
value of 54.1% +/- 16.0%.
54
5 Discussion
5.1 Speech Analysis and the PEAKS Program
“Speech is an important, although controversial, aspect of the management of cleft lip and
palate around the world.” (Lohmander, 2004)
Assessment of speech-language problems in individuals with a CLP begins in infancy and,
for some, will extend through adolescence and beyond. Stengelhofen (1989) estimates that
40% of all CLP patients continue to have speech deficits throughout adulthood. The
continual evaluation of the speech of CLP patients can therefore be deemed as an essential
part of their management, allowing for a timely diagnosis of speech deficits and confirming
indications for further treatment. Moreover, research on speech intelligibility of CLP patients
could lead to further improvements of the various treatment strategies involved.
Currently however, there are numerous problems involved in the clinical research
methodologies used for the evaluation of cleft lip and palate treatment. Researching speech
of CLP patients is a complex process and often lacks in uniformity. Multidimensionality of
outcome, length of follow up, diversity of management and small sample sizes (Roberts,
1991) are for example factors, which make evaluation of the results extremely difficult.
Furthermore, large age ranges, different cleft types, the use of highly variable speech
samples and the lack of information about listeners and on reliability of the procedures
further complicate cross-sectional studies (Lohmander, 2004).
Perhaps the most significant difficulty in researching speech pathologies of CLP patients
however, pertains to the fact that the diagnostic procedures are numerous (Chapter 2.9) and
continue to vary considerably. Lohmander and Olsson (2004) wrote an informative review of
88 articles published between 1980 and 2000, with the purpose of investigating the
methodology for perceptual speech assessment in patients with CLP. The review states that
the interpretation and comparison of speech research often is complicated by differing or
flawed methodologies, regarding for example the collection and documentation of data and
methods for measurement. Roberts et al. (1991) also evaluated the clinical research
methodologies used for the evaluation of cleft lip and palate therapies in reports between
1964 and 1988. Roberts et al. conclude that
55
“the reproducibility of outcome measures in CLP patients (the degree to which repeated
examinations would reach intra and interexaminer agreement) and their validity (that the
assessment actually measures what it proposes to) frequently diminishes the credibility of
research findings and the extent to which they can be generalized.”
Hence it is apparent, methods for speech research in the past have been far from ideal.
So which method should be used to rate speech pathologies of the CLP children? There is
little agreement on the answer to this question (Kummer A. W., 2012), and no single
universally recognized method has to date been established. Several formalized rating
systems and protocols for evaluating CLP speech have been proposed in the literature. For
example the Eurocleft Boimed Project developed a cross linguistic method of assessment of
CLP speakers when used by trained listeners (Shaw W. C., 2001), which included not only
speech variables to be assessed but also recommendations for speech material and ages for
assessment. Dalston et al. (1988) identified minimal standards for reporting the results of
cleft palate surgery and recommended that speech results should include measures of hyperand hypo-nasality, intelligibility, nasal emission and articulation. Another example is
reported by Henningsson et al. (2008), who lists a set of five universal reporting parameters
(hyper- and hyponasality, audible nasal air emission and/or nasal turbulence, consonant
production errors and voice disorders) and two global speech parameters (speech
understandability and acceptability), which are meant to provide a framework within which
speech results can be reported in a consistent and uniform manner. The detail and
complexity of some of these systems however, make them very hard to follow in a clinical
situation, especially if there is no perceived value added by such an in depth analyses
(Kummer A. W., 2012).
Even when these minimal standards and recommendations for reporting results are adhered
to, comparing the studies is difficult, as the parameters are often reported by different raters
and also rated on different scales (for example a 3 point or a four point Likert scale).
Comparing speech outcome results among studies where speech is judged by such variable
rating systems is essentially impossible. Therefore there cannot be much progress in
improving speech outcomes until there is a reliable way to actually compare outcome results.
So who should in the end evaluate the speech data? At the moment, the “objective” gold
standard for research purposes (Sell, 2005) is the blind independent auditory evaluation of
speech data through several speech language pathologists (SLPs) (Kuehn, 2000), where the
56
mean or median is subsequently calculated. This method is still the most widely used
technique for assessing speech intelligibility. Nonetheless, professionals other than SLPs
routinely reported speech outcomes in previous studies. Surgeons, SLPs, parents of children
with or without cleft plate and also cleft palate children themselves have all been used to
judge overall intelligibility (Whitehill, 2002).
Currently, SLPs are not only responsible for the evaluation of speech for research purposes,
but also for clinically evaluating speech of CLP patients throughout their lengthy treatment.
Speech language pathologists are now valued members of the interdisciplinary team
(Watson, 2001). Moll (1964) made a strong case for the desirability and necessity of making
these listener judgments the basis of all assessments of speech. He suggested that listener
judgments provide the only measures that are direct and logically valid, given the basic
perceptual nature of speech.
“The single most important assessment tool that the speech-language pathologist will use
in clinical practice is his or her ears. ” (Peterson-Falzone, 2010)
Although listener judgments intuitively appear to be the most valid indicator of speech
deficits, the reliability and accuracy leaves much to be desired. The listener variables listed
in Chapter 2.9.1 (different experience levels, inter- and intra-individual differences) are
confounding factors, which have the potential to influence speech ratings and hence cause
biased results. Even when the average of multiple raters is taken into account, the measure is
still subjective and rather not reproducible (Maier, 2009). The auditory assessment also
requires much time and effort and the applied methods for perceptual speech analysis often
vary (Lohmander, 2004).
The shortcomings of the perceptual analysis and the need for a more consistent and
comparable method or speech findings are stressed in several studies: (Dalston, 1988;
Lohmander, 2004; Morris, 1973; Watson, 2001).
“It is hoped that ongoing national and international collaborative efforts to standardize
procedures for collection and analysis of perceptual data will help to eliminate such
concerns and thus make comparison of published results possible in the future.”
(Lohmander, 2004)
An instrumental assessment of speech language problems could, in our opinion, overcome
many of the shortcomings of a perceptual analysis. Moll (1964) emphasizes nonetheless that
57
any instrumental approach, if it is to be useful, must provide reliable data that are closely
related to listener judgments.
In this study the PEAKS program is implemented as an instrumental, objective assessment of
the speech of CLP patients. Objective evaluations have the advantage that they are
reproducible, reliable and fast in most cases (Maier, 2009). The results are also easily
comparable in inter-center studies. However, several disadvantages of instrumental
techniques are also worth mentioning. They are often expensive or invasive, which might be
uncomfortable for the patient. The PEAKS program is however neither expensive as it is
freely accessible over the internet, nor is it invasive as recordings are done with a
microphone headset (Maier, 2009). One has to also keep in mind that some instrumental tests
are rather unspecific, merely evaluating aspects of speech such as articulation disorders,
voice quality and resonance (Chapter 2.9.2). PEAKS however evaluates speech
intelligibility; intelligibility is a global parameter for speech outcome in children and adults
with CLP (Schuster M., 2012). Intelligibility refers to the ‘understandability’ of speech, and
is the most immediate criterion by which a child’s communicative attempts are judged.
A myriad of factors can contribute to the speech errors typical for CLP patients. It is often
assumed that the primary goal in the assessment of children with clefts is identifying and
treating the speech production problems associated with velopharyngeal inadequacy (VPI),
and as a result, resonance is the most often recorded speech parameter (Lohmander, 2004).
Although this is true for many children, factors such as dental occlusion, palatal fistulae, and
nasal obstruction also play a role in the assessment (Wyatt, 1996). PEAKS calculates the
word recognition, which corresponds to the overall intelligibility of the child (Mayr, 2010).
Therefore by calculating degree of intelligibility, PEAKS can take into account many of the
etiological factors which could contribute to the typical pathological speech of CLP patients,
such as for example nasality and articulation errors (McWilliams, 1954) but also phonation
and prosody (De Bodt, 2002). Therefore we believe intelligibility is a creditable parameter
for reporting overall speech outcome in cleft patients.
In the review of Lohmander and Olsson (2004), the type of speech variables assessed in 88
previous studies on the speech of CLP patients have been listed (see Figure 5-1). It is
interesting to note that only 11 of these studies used intelligibility as an evaluation
parameter. There has however recently been an increase in the number of studies using
intelligibility as an outcome measure in investigations of speakers with cleft palate
58
(Whitehill, 2002). In 50% of the studies reviewed by Lohmander and Olsson (2004), only
one speech variable was investigated, while in 13%, 4-5 were investigated. Most commonly
an interval rating scale was used, with 3 to 10 points. Additionally, in 59% of the articles,
instrumental analysis had been used as a complement to perceptual evaluation. It is evident
from this review that the analysis of the speech of CLP patients is far from standardized, and
therefore causes extremely difficult conditions for inter-center comparisons of speech data.
Variables
Resonance
Articulation
Perceived airflow
Intelligibility
Voice quality
Velopharyngeal function
Grimace
Included in No. of Studies
62
56
29
11
11
9
2
Figure 5-1: Type of Assessed Speech Variables in Reviewed Articles (n=88) (Lohmander,
2004)
The PEAKS program shows potential for such a standardized and objective method to assess
speech data. As yet, automatic speech recognition (ASR) has been regularly used for
automatic communication systems in order to recognize speech, e.g. for telephone or
dictating systems. ASR is now increasingly being used to quantify speech alterations for
medical purposes (Gales, 1996). Not only can modern ASR systems produce text from
speech, but they are also able to supply a scoring for their performance (Kitzing, 2009), an
example being the PEAKS program.
Speech quality assessments are expected to fulfill certain requirements; they must make
reliable and meaningful data available as quickly and cheaply as possible. PEAKS fulfills
these criteria. As mentioned above, PEAKS is freely accessible over the Internet and only a
microphone and soundcard would cause extra expenses. The recordings only take in average
around 4 minutes and the actual processing and evaluation of the speech is done
automatically by the program in about 1 to 2 minutes. PEAKS also has additional
advantageous from a clinical aspect: it runs in any web browser and has the necessary
security features (Maier, 2009). Clinical application is therefore uncomplicated and
enforceable.
59
Various recent clinical studies (Dames, 2009; Haderlein, 2009; Maier, 2009; Maier, 2006;
Schuster Maria, Haderlein, 2006; Schuster M., 2012; Vogt, 2007; Windrich, 2008) have
shown promising results using the PEAKS program, and include a wide range of patients
with varying articulation or voice disorders. The studies of Haderlein et al. (2009) and
Windrich et al. (2008) have for example demonstrated that the PEAKS program has a high
consistency with experts` estimations of the degree of intelligibility of adult patients.
Haderlein et al. (2009) assessed the speech of 41 postlaryngectomy patients and came to the
conclusion that,
“PEAKS can be used for objective intelligibility ratings with results comparable to those
of human experts.”
Windrich et al. (2008) studying the post operative speech intelligibility of forty-six patients
with oral carcinoma, came to the conclusion that automatic speech recognition may serve as
a valuable tool to assess global speech outcome objectively and quantitatively for clinical
and research purposes.
Children with CLP have also already been evaluated with PEAKS program in several studies
(Maier, 2009; Maier, 2006; Schuster Maria, Maier, 2006; Vogt, 2007). In these studies, the
CLP patients achieved significantly worse WR results to those of the control group, as was
to be expected. This shows that PEAKS was able to differentiate normal children’s speech
from pathological speech. In our study, the control group adopted from a study from Dames,
Maier et al. (2009) achieved a significantly (p<0.001) higher mean WR value (WR: 63.5%
+/- 12.1%) compared to our 51 CLP patients (WR:51.5% +/- 14.5%). The validity of the
PEAKS program is therefore again underlined.
As to further proof of the validity of the program, in previous studies (Maier, 2006;
Windrich, 2008), the PEAKS program reached 0.90 and 0.93 correlation respectively with
the expert raters; these included voice professionals, experienced clinicians and a speech
recognition expert. The inter-rater correlations varied between 0.81- 0.95 in these studies. In
our study, not only were expert ratings compared to that of the PEAKS program, but also the
ratings of an amateur. This method of comparing the results of experienced and completely
inexperienced raters to the PEAKS program was to our knowledge not yet carried out in
previous studies. The most experienced raters in our study (the linguist and ENT doctor)
both achieved a high correlation of 0.82 with the WR of the PEAKS program. The
inexperienced rater (medical student) achieved the lowest overall correlation of 0.65.
60
So why have the experts in our study achieved a lower correlation to that of the experts of
the previously mentioned studies? Unfortunately, it is not apparent exactly how much
experience the raters have in these mentioned studies (Maier, 2006; Windrich, 2008) in
rating speech of CLP patients. This is also something, which is difficult to quantify. One can
assume that the experience level of our linguist and ENT doctor do not compare to that of
the experts used in the previously mentioned studies; the linguist having more of a scientific
approach to speech and the ENT doctor in this study having only limited experience in
judging speech of CLP children. This could be the reason why they achieved a lower
correlation to the PEAKS program compared to the expert raters in the studies of Maier et al.
(2006) and Windrich et al. (2008). As experience is an important factor in judging speech of
CLP patients (Keuning et al. 1999; Paal et al. 2005), the results would hence be logical: the
more experienced the rater, the better the correlation to the calculated WR value. This would
imply that the PEAKS program correlates far better with the results of an experienced to that
of an inexperienced rater.
On the other hand, if we are only to look at the results of our study, and are to assume that
the linguist is our gold standard (as she is the rater with the most experience), we may come
to a slightly different conclusion. We have calculated that the PEAKS program has an 82%
correlation with the linguist’s ratings. When evaluating the 3 different raters amongst each
other, it is interesting to note that the medical student also has around the same correlation
with the linguist’s ratings, reaching a consistency of 83%. From these results we could
deduce that the PEAKS program is approximately “as good as” a medical student in rating
the degree of speech intelligibility of CLP patients. One could therefore conclude that the
PEAKS program is not yet on level with that of an experienced rater, but on that of an
inexperienced rater. However, it is important to stress the fact that the linguist is not the ideal
gold standard, but rather a SLP. SLPs are professionals that deal on a day-to-day basis with
communication disorders. A future study comparing the subjective ratings of several
experienced SLPs and those of amateurs, to the objective results of the PEAKS program,
would allow for a more conclusive result on how “good” the PEAKS program actually is at
rating speech intelligibility.
All things considered, the correlation between the subjective ratings of trained raters and the
PEAKS program has already been proven to be very high, from 0.83 in our study, to 0,93 in
the study of Windrich et al. (2008). We believe that the PEAKS program is an excellent
option in objectively quantifying the degree of intelligibility. As various listener variables
61
can distort results of SLPs, we believe PEAKS can in comparison, produce results with a
higher scientific credibility and impact. PEAKS has the advantage that its results are
reproducible: the program always produces the same result when evaluating the same speech
data. Therefore PEAKS has a very high “intra-judge” reliability. This is not the case in
subjective ratings of speech intelligibility, as even expert speech language pathologist can
show a strong intra-rater variance and also low correlations. Keuning et al. (1999) wrote for
example a paper, which investigates the intelligibility of 15 CLP patients with 5 speech
language pathologists and one oral and maxillofacial surgeon. In this study, the mean overall
intra-judge reliability for rating intelligibility was calculated at 0.61 (p <0.01), which can be
seen as a very low correlation for experts. In a comprehensive review of 57 papers written by
Kreiman et al. (1993), no clear relationship was found between intrarater reliability and a
rater’s experience. Correlations between the first and second ratings ranged from 0.66 to
0.98 for expert listeners (10 papers), and from 0.59 to 0.95 for graduate students (5 papers).
The inter-judge reliability also varies considerably in various studies using perceptual
analysis (Kreiman, 1993), and Kreiman et al. concludes that from the studies reviewed, no
consistent relationship between listeners` experience background and the levels of inter-rater
reliability is apparent. The correlation between raters ranged between 0.57 and 0.79.
Research could not be found to the “inter-rater” reliability of the PEAKS program. It would
be interesting to test the PEAKS program in a future study on its “inter-rater” reliability,
recording a person speaking the same speech sample more than once, and calculating the
consistency in which the PEAKS program is able to rate this persons speech samples.
Listener variables are one of the main reasons why “evidence based medicine” in the field of
analyzing pathological speech has not been possible in the past. However, not only listener
variables, but also the speaker variables must be considered when evaluating speech.
Numerous factors, known and unknown, can affect children’s speech performance and
render evidence regarding speech outcome inconclusive. The following need to be
considered when evaluating the speech of CLP patients; the age of the child, the presence of
an isolated cleft palate, the type and timing of surgery and the presence of normal hearing
are known variables (Van Lierde K. M., 2010). Other factors which should be taken into
account are gender, the presence of certain malformations or syndromes and his/her
language background; the effects of many variables are however still unknown. In previous
studies, the information regarding variables is often lacking and/or the inclusion criteria
differ (Lohmander, 2004). In this study, patients with additional malformations/syndromes,
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those who had undergone a velopharyngoplasty and those with a mother tongue other than
German were excluded. The variables age, gender and cleft type were investigated and the
effects of different treatment concepts on the degree of speech intelligibility were
additionally evaluated.
5.2 Effect of Cleft Type on the Degree of Speech Intelligibility
In previous studies, the effect of the different cleft types on intelligibility has shown
inconclusive results (Dames, 2009; Van Lierde K. M., 2002). However, severely increased
nasal resonance and air emission is a frequent finding in subjects with clefts involving the
hard or soft palate, but is seldom observed in subjects with cleft lip and alveolus
(Schönweiler R., 1999). This lead us to the hypothesis that CLP patients with a cleft palate
would have a reduced intelligibility compared with cleft patients without palate involvement,
as nasality is correlated with intelligibility (McWilliams, 1954; Van Lierde K. M., 2002).
Hence, we divided the patients into Group 1 (without cleft palate) and Group 2 (with cleft
palate).
On the other hand, abnormalities of the anterior articulation zone can also lead to a decreased
intelligibility. Van Lieshout et al. (2002) however suggests that there seems to be a relatively
high tolerance for the effects of for example the upper lip movement variability on speech
intelligibility. Schuster et al. (2012) also came to the conclusion that, in average, we would
expect children with cleft including the palate to have a diminished intelligibility whereas
isolated clefts of the lip should not have a lack of intelligibility in relation to children at the
same age without clefts.
This hypothesis was confirmed in the PEAKS study undertaken by Schuster et al. (2012).
Using PEAKS, they observed that the WR results were significantly lower in the cleft lip,
palate and the cleft palate group (in our case: Group 2) compared to that of the control
group; whereas no significant difference between the comparison group and children with a
cleft lip or cleft lip and alveolus (in our case: Group 1) was observed. Schuster et al. (2012)
suggest that from their result,
“an alteration of the anterior articulation zone by a cleft in CLP children has a lower
impact on speech intelligibility compared to the influence of a cleft palate.”
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These results are however not in accordance with our study. Both Group 1 and Group 2
achieved statistically significant lower mean WR values compared to the control group.
Surprisingly Group 1 achieved even a slightly lower (WR: 46.77% +/- 12.4%), however
statistically insignificant (p= 0.228), mean WR value compared to Group 2 (WR: 52.67% +/14.8%). This could be due to the small sample of Group 1 patients (10 patients) compared to
Group 2 (41 patients). On the other hand it is known that clefts of the lip and alveolus can
lead to restricted lip movement and misaligned teeth, which ultimately can affect speech
intelligibility (Wohlleben, 2004). The connection between intelligibility and the anterior
articulation zone is inconsistent and not enough research has been done to date to come to a
reliable conclusion. A larger sample should be investigated in the future using the PEAKS
software, with a more detailed cleft subdivision considering their involvement with the
palate and the anterior articulation zone.
5.3 Effect of Gender on the Degree of Speech Intelligibility
Gender has also shown varying influence on speech intelligibility in previous studies,
although not much research was to be found. Hardin et al. (1986), assessing 50 adolescent
CLP patients through speech pathologists, concluded that females achieve better speech
proficiency than males, regardless of the cleft typ. Therefore in the present study, the
hypothesis was established that the female patients would achieve higher mean WR values
than the males. On the other hand, more recent studies (Strauss, 1988; Van Lierde K. M.,
2010) have shown no significant difference between male and female speech results of CLP
children. These ratings were however from the patients` themselves or their parents.
Interestingly, Dames, Maier et al. (2009) also observed no gender influence on the WR value
in children with a one-sided complete cleft lip palate, whereas the male children with
bilateral cleft lip palate achieved significantly better results than the females. In this PEAKS
investigation, the female patients achieved a slightly higher (WR: 55.98% +/- 16.3, Mean
Age: 9.8 +/- 4.3), however statistically insignificant (p=0.155) mean WR value compared to
the male (WR: 49.28% +/-13.2, mean age: 9.7 +/-2.9). Further PEAKS studies with larger
patient collectives could resolve the question as to whether gender influences the degree of
speech intelligibility.
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5.4 Effect of Age on the Degree of Speech Intelligibility
Learning how to speak takes time to develop. Every speech-language pathologist knows that
there is a huge variation in the time it takes for children to learn to talk. The improvement of
intelligibility with age of cleft and non-cleft children has been proven in several studies
(Schuster M., 2012; Strauss, 1988; Van Lierde K. M., 2010). In the study of Schuster et al.
(2012), where the PEAKS program was also implemented, WR significantly increased with
age. Our study also shows a general increase in the mean WR values the older the patient,
the lowest mean WR value being at the age of 5 (WR: 26.51% +/- 10.1%) and peaking at the
age of 13 (WR: 72.73% +/-2.9%). The 14-19 year old patients however achieved lower mean
WR values than the 13 year olds. This is possibly due to the small sample sizes in these age
groups: the age groups 15,16 and 19 having only one patient. One has to also keep in mind
that the results of the older children could be distorted due to the fact that these children are
eventually difficult cases. As clefts are only treated in specialized cleft centers, some
children have very long commutes. Thus, as the children get older, they are inclined to attend
their yearly follow up examinations less regularly, as most (if not all) of their treatment is
already concluded. The CLP patients however who still are having difficulties due to their
cleft, would naturally attend more often. Surprisingly, the 4-year-old children also did not
follow the general trend, achieving a higher mean WR value than expected (41.75% +/11.2%).
It is important to keep in mind, that the recognition system of PEAKS was trained with
“normal” speakers from 10 to 40 years old. In comparison, children deviate from the
standard speaker and within the speaker group (Wilpon, 1996). Therefore not only
pathological speech, but also the speech of younger children should show much lower WR
values than 100% (Schuster M., 2012). In the study of Shriberg et al. (1994), researchers
found that the final age of normal speech acquisition, called the speech normalization
boundary, is 8.5 years. This means, at this age, children should be able to say all sounds
correctly and that sound differences persisting past 8.5 years are not likely to spontaneously
correct. Therefore, one can assume that the PEAKS program was trained with participants
who should have a normal degree of intelligibility. Consequently, the younger children
(under 8.5 years) in this study should have worse WR values to the older children (over 8.5
years old). This is in fact the case with the mean WR value of children <8.5 years of age
reaching 40.40% +/- 13.5% and those older than 8.5 years achieving a significantly (p <
0.001) higher mean WR value of 56.60% +/- 12.0%. These low recognition rates in young
65
children can therefore not only be due to speech deficits related to the clefting, but also
partly due to phonetic and voice development (Schuster M., 2012). This development should
be completed at the age of 8.5. It is however interesting to note, that the WR rates do not
peak at 8.5 years in this study, as one would suspect in “normal” children. The continual rise
in WR value until the age of 13 could imply that CLP patients may have a slower speech
development to that of healthy children. It has already been shown in literature that cleft
palate children were found to be retarded in both language comprehension and language
usage (Scherer, 1999). Bzoch (1956) found for example that 50% of the mothers of 60 cleft
patients reported delays in babbling, jargon, use of the first word, and use of the first twoword sentences.
Wohlleben (2004) states that accurate and detailed evaluations of the intelligibility of cleft
patients are not possible until exact comparative studies can be carried out. Research
concerning speech intelligibility of healthy children (Gordon-Brannan, 1994; Shriberg, 1994;
Van Lierde K. M., 2010) has been carried out since the 1930s, however limited normative
data are available for speech intelligibility in young children (Gordon-Brannan, 1994). A
scale showing their intelligibility development does not exist to our knowledge (Wohlleben,
2004). The PEAKS program could possibly be the solution, as by implementing this ASR
system to a large amount of healthy children, one could obtain a significant amount of
speech data on a linear scale and could hence graph the results. These results could then be
applied by comparing the normative speech data of the healthy children’s speech to the
speech of for example CLP patients; this would allow for more accurate and objective
results.
5.5 Inter-Center Study: Effect of different Treatment Strategies on the
Degree of Speech Intelligibility
CLP children present a complex diagnostic puzzle that requires interdisciplinary teamwork
to solve. The treatment of CLP patients has spurred much interest in the literature, as many
different treatment regiments are in use. A recent survey revealed that 201 different
European teams used 194 protocols for one cleft subtype, making comparison of outcomes
impossible (Shaw W. C., 2001). The treatment methods regarding the lip and palate repair
vary according to the clinic/surgeon. In our study, the degree of speech intelligibility results
of CLP patients treated in Erlangen are compared to those treated in Freiburg. Details of the
66
different treatment regimes used in Freiburg and Erlangen are given in Chapters 2.8.2 and
2.8.3.
Intelligibility has been described as the most important measure of speech disorder and
increasing intelligibility as the primary goal of surgical and therapeutic intervention
(Yorkston, 1978). Van Lierde et al. (2010) also suggests that the success of cleft palate
surgery is determined by the subsequent speech intelligibility. Evaluation of the efficacy of
palatal surgery in CLP patients and a determination of the long-term speech outcome is
therefore of interest to surgeons who treat cleft palates. The importance of assessing the
interactive components of speech intelligibility is well recognized in the cleft palate
population; however, it is interesting to note that only a few authors (Dalston, 1988;
Hirschberg, 1997; Whitehill, 2002) recommended the inclusion of intelligibility in a standard
speech assessment. Also Lohmander and Olsson (2004) show that out of 88 studies
regarding speech of CLP patients, only 11 evaluate speech intelligibility.
The relationship between speech intelligibility and the effects of the different treatment
methods has not yet adequately been explored. Intelligibility levels are however frequently
used in making clinical decisions, and for this reason measurements need to be accurate and
reliable. Clearly it is a very important concept, but one which is often difficult to measure
and address in intervention for children with speech difficulties (Pascoe).
In our study, the effect of different treatment regimes on the degree of speech intelligibility
of the cleft patients was investigated using the objective PEAKS program. Fifty-one patients
in Freiburg were assessed and the resulting WR values were calculated. Of these fifty-one
patients, only 25 children suffered under a complete lip, alveolus and palate cleft, and could
therefore be compared with the WR results of the 72 patients assessed in Erlangen (in the
study of Dames and Maier 2009), as these suffered under complete lip, palate clefts. The
mean WR values indicated that the regime applied in Freiburg shows slightly better degree
of intelligibility results (mean WR =51.3 +/- 15.3) compared to the CLP patients in Erlangen
(mean WR= 48.1 +/- 16.3). The difference is however statistically insignificant (p= 0.381).
Freiburg patients are also 1.6 years older than the Erlangen patients, which could have
caused the superior result. Also the sample sizes being compared differ, 25 patients in the
Freiburg group being too small to be able to make a well-founded assertion.
As mentioned, treatment of CLP patients is a long and complex process, involving numerous
experts and countless opinions on its optimal therapy.
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“It is difficult, if not impossible to establish the key beneficial or harmful feature of a
specific treatment as a general scientific conclusion, due to the invariably complex and
arbitrary mix of surgical technique, timing and sequence, ancillary procedures and
surgical personnel.” (Roberts, 1991)
Additionally, many confounding variables are involved, such as for example age or gender.
It is therefore very complicated distinguishing which factors contributed to the superior
speech of for example the CLP patients in Freiburg.
Further inter-center studies are
necessary with far larger patient groups. To begin with, an inter-center study comparing the
overall treatment regiments used in the different clinics could be carried out, based on the
mean WR values achieved. Thereafter, if significant disparities in the speech results are
established, specific hypotheses to the cause can be generated and later investigated in more
detailed prospective trials. This could eventually lead to an improvement of long term results
(Roberts, 1991).
PEAKS would, in our opinion offer an excellent method to adequately compare the degree
of speech intelligibility results of various treatment regimes, on an inter-center and even
international scale. The method is simple and quick, the average recording time of the
patients in this investigation being approximately 4 minutes. This allows for the
investigation of large numbers of patients, the patients even able to carry out the test at
home. The WR value is also a parameter that is independent of the language used, as long as
the program is trained with the appropriate speakers of the respective language. In the past,
comparing speech outcomes in different countries has often been difficult to accomplish, as
the speech parameters used at different centers are not always compatible. Henningsson et al.
(2008) have recently reported such a universal system for reporting speech outcome
measures
internationally,
including
five
universal
characteristics:
hypernasality,
hyponasality, nasal air emission and/or turbulence, consonant production errors and voice
disorders. However, due to the perceptual nature of the evaluation and the hereby-caused
drawbacks, we believe that the PEAKS program is a superior method. As apposed to the
method proposed by Henningson et al (2008), it is quick, reliable, objective and basically
needs no training to be utilized.
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5.6 Effect of Grommet Insertion on the Degree of Speech Intelligibility
Not only different treatment regimes can be compared using PEAKS, but also the influence
of specific therapies. In the present study for example, the effect of grommet insertion on the
degree of speech intelligibility of the CLP children was investigated. The application of
grommets, especially its timing and long term benefits, is the controversial topic of many
studies (Bluestone C., 1996; Browning, 2010; Maw, 1999; Moore, 1986; Paradise, 1974;
Paradise, 2005; Ponduri, 2009; Rach, 1991; Robson, 1992; Shaw R., 2003; Valtonen, 2005).
In a study of Schönweiler et al. (1989), investigating 417 cleft children, it was suggested that
language skills did not depend on the type of cleft palate present, but on the frequency and
amount of hearing loss found. The early placement of ventilation tubes (for example
concurrent with the palatoplasty), followed by replacement as/when necessary in children
with CLP, has been advocated by a number of authors as a means of preventing the adverse
effects of hearing loss on speech and language development (Bluestone C., 1996; Hubbard,
1985; Moore, 1986; Paradise, 1974; Valtonen, 2005). It is for this reason that many
interdisciplinary treatment centers routinely place grommets during the first year of life. The
routine grommet insertion involves the placement of ventilation tubes during the lip or palate
operation. This is done so that the children do not have to undergo another general anesthesia
for the placement of the grommet, as it is combined with an already planned and essential
procedure.
Current studies however, postulate that in non-cleft patients, early grommet insertion is not
generally recommended (Browning, 2010; Caye-Thomasen, 2008; Paradise, 2003), as there
is an increase in abnormal otological findings, surgery requirements and beneficial long term
hearing effects have also been elusive (Goudy, 2006; Robson, 1992; Sheahan, 2003).
Browning et al. (2010) wrote an informative review, which concludes that the benefits of
using grommets in non-cleft children were small and that the effect of grommets on hearing
diminished over the first year. It was suggested that there should be an initial period of
observation before considering interventions with grommet insertion. Randomized trials that
have also included language skills, have demonstrated little or no effect of grommet insertion
compared with watchful waiting (Maw, 1999; Rach, 1991). Prospective trials conducted by
Paradise et al. (2005, 2007) confirmed that there were no differences in developmental
outcome to early or late tympanostomy treatment. The study of Paradise et al. (2007) failed
to identify an association between early otitis media and later impairments (such as language
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deficits), even after 9-11 years. These studies therefore imply that a conservative approach to
grommet insertion will not negatively influence a healthy child’s speech development and
language skills.
The evidence for early grommet insertion in children with clefts is however unsatisfactory
and contradicting (Maw, 1999; Merrick, 2007; Phua, 2009; Ponduri, 2009; Szabo, 2010).
Ponduri et al. (2009) wrote an informative review, with the objective of determining whether
early routine grommet insertion in children with cleft palate has a beneficial effect on
hearing and speech and language development compared with conservative management.
Out of 364 citations, only 18 studies met his inclusion criteria, and most were either of poor
quality, small, or both. Ponduri et al. (2009) concludes
“that there is insufficient evidence to determine whether early routine grommet insertion
in patients with CP has long term benefits for hearing, speech and language, and
psycholosocial development.”
Maw et al. (1999) undertook the first randomized prospective study examining cleft palate
patients, dividing 182 children to either immediate surgery or a watchful waiting group. At
18 months there was no significant difference in expressive and receptive language between
the two groups. Unfortunately, no further follow up examinations were carried out. To our
knowledge, no long term randomized prospective studies exist to date comparing the effect
on speech and language development of early grommet insertion versus conservative
management. In a comprehensive retrospective study of 234 cleft palate patients (Phua,
2009) it was concluded that there was no difference in the incidence of persistent conductive
hearing loss between the patients who received and did not receive early grommet insertion.
Recurrent middle ear disease, tympanic membrane abnormalities and the total number of
grommet insertions were on the other hand, significantly higher in the routine grommet
group. In addition, poorer outcomes were noted in patients who had undergone a greater
number of grommet insertions. Patients who underwent routine grommet insertion had a
greater number of grommet procedures (mean: 1.8 grommets) compared to those who had
grommets inserted selectively (mean: 0.55 grommets). In summary, Phua et al noted better
otologic outcomes and fewer grommet insertions in patients treated using a selective
grommet protocol compared to a routine protocol.
Regarding the development of speech and language in children with CLP, it is to date
difficult to come to any firm conclusion about the best management of otitis media with
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effusion. In view of the many variables involved in the investigation of the speech of CLP
patients, the different diagnostic procedures implemented and last but not least the
contradicting results of various studies, it is no wonder that research in this area has lead to
questionable results. Additionally, many of the studies, as with this one, have small sample
sizes, involve children of varying ages, are retrospective and not randomized.
A conservative approach to grommet insertion is taken in Freiburg. The children first
undergo an otoscopic, audiometric and impedance screening shortly before the palatoplasty
(see Chapter 2.8.2.2). Significant hearing loss or a pathological otoscopic result at the age of
the palate operation, and also throughout their lengthy treatment process, is seen as a
possible indication for inserting a ventilation tube. However, the results of the screening, and
also the advantages/disadvantages of grommet insertion are discussed at length with the
parents, before a decision is made. The policy of watchful waiting is preferred.
In the present study, 65 (52%) of the 126 CLP children documented, underwent ventilation
tube insertion in Freiburg. This is not in keeping with other series reporting on conservative
management of OME, a lower insertion rate being recorded by Robson et al. (1992) with
26% and 29% in the study of Shaw, Richardson et al. (2003). 33 (26%) of the Freiburg
patients had their first ventilation tubes inserted during the palate operation. 19 (15%)
underwent grommet insertion on three or more occasions. It is interesting to note, that
considerably less of the Group 1 patients (25%) received ventilation tubes compared with the
Group 2 patients (58%). This could be due to the fact that with cleft involvement of the
palate, the musculus tensor palatae is also more likely involved, which has been proven to
predispose to an OME (Sehhati-Chafai-Leuwer, 2006).
Not only were the number of grommet insertions in CLP patients recorded in this study, but
also their effect on the degree of speech intelligibility; the word recognition rate was
considered according to the number of grommets inserted and also to the point in time they
were inserted (before or after the palate operation). 23 (45%) of the 51 PEAKS assessed CLP
patients did not have any grommets inserted at all and achieved a mean word recognition
rate of 54.1% +/-16.0%. It was interesting to note that these patients showed a wide range of
intelligibility, with WR values ranging from 13.13% to 82.82%. The remaining 28 patients
had at least one ventilation tube inserted, and achieved a mean word recognition rate of
49.4% +/- 13.1%. This shows a slightly but insignificantly (p= 0.650) higher mean word
recognition value for the CLP patients who were not treated with ventilation tubes. The 3
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patients who received at least 6 grommets however, showed a significantly (p= 0.023)
reduced word recognition rate with a mean WR of 35.25% +/-4.8%.
The results of the present study raise the question: are numerous grommet insertions actually
the cause of conductive hearing loss and thus reduced speech intelligibility? Or do these
patients already have a more severe underlying middle ear condition, which leads to hearing
disabilities and thus reduced intelligibility (Goudy, 2006). Unfortunately, this question
cannot be answered in this study. Nevertheless the present study does underline the lack of
long term benefits of ventilation tube insertion on the speech intelligibility of the CLP
patients, as the degree of speech intelligibility of the patients who received grommets did not
improve, even worsening at 6 grommet insertions.
A comparison of the WR values between those who underwent grommet insertion during the
palate operation, and those who had their first grommet inserted after the palate operation,
was also undertaken. The results demonstrate a lower degree of intelligibility in the patients
with grommet insertion during the palatoplasty (WR: 46.9% +/-17.5) compared with those
who had the insertion after the palate operation (WR: 51.0% +/-9-6) and also to those with
no grommet insertion at all (54.1% +/-16.0). This suggests that aggressive management of
OME does not improve the degree of intelligibility results of the CLP patients. This
difference in mean WR values is however insignificant, and may be due to an increase in the
mean age of the respective groups.
Further randomized prospective studies would be needed to confirm the above made
assumptions that: regarding the degree of speech intelligibility, ventilation tube insertion
lacks in long term benefits and early tube insertion is not beneficial. The sample size of this
study is not sufficient to make well-founded conclusions to this topic. Also our study is not
prospective, nor randomized. Although this study did not produce significant results
concerning the indication for grommet insertion, it can be seen as a pilot study for future
research. There is no doubt that studies on grommet insertions in patients with CLP will be
complicated, requiring careful planning, multidisciplinary teamwork, and also a long term
follow-ups. PEAKS could in our opinion simplify this process. The PEAKS program could
be implemented to follow the speech outcomes of patients with hearing difficulties
continually from an early age. Shaw, Richardson et al. (2003) states
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“The use of speech outcome, as a surrogate for hearing, whilst not ideal, has some
validity, as speech and language development is known to be strongly correlated to
hearing.”
And as normal speech acquisition and development is a main argument for early and
aggressive grommet treatment (Paradise, 1974), speech intelligibility can be seen as a valid
parameter to be tested. The benefits or disadvantages of conservative versus routine
grommet insertion on the speech outcome could hence be evaluated with an objective and
simple method, and could even be done on an international scale.
Phua et al. (2009) recommended that ventilation tube placement occur in patients selected on
the basis of symptomatic infection or significant hearing loss; this is in agreement with our
recommendation. From our results, we believe that a wait-and-see strategy should be
employed in the indication for grommet insertion in CLP children, just as it is done in the
normal population (controlled intervention). Whatever the approach taken, it can be
recommended to closely monitor the hearing and language of the CLP children with
aggressive hearing tests and also for example an annual PEAKS assessment. As a result,
hearing loss and speech deficits could be detected at the earliest possible opportunity, and if
necessary, an intervention could be initiated.
5.7 Implications
The PEAKS program is a diagnostic tool that shows great potential in the field of speech
analysis. It may be applied on a day-to-day basis for clinical purposes and also might be a
long sought-after research tool, especially in the field of CLP treatment outcomes. Roberts et
al. (1991) states
“In recent decades the management of CLP patients has made considerable progress, but
a uniform regime has not yet been established. The presence of underlying variation and
confounding variables will demand increasingly sophisticated research methodologies to
observe small improvements.”
In our opinion, this sophisticated research tool could be the PEAKS program. It objectifies
and quantifies speech intelligibility, thus making it a novel tool in speech analysis. Prior
studies have shown absolutely no consistency in the methodologies used to compare speech
outcome of CLP patients, making it virtually impossible to compare research data (Kummer
73
A. W., 2012). Success rates of surgery and also those of specific therapies such as grommet
insertion, speech therapy or even a velopharyngoplasty may be compared objectively with
the PEAKS program in the future. As the comparison of objective intelligibility results was
till currently not possible, PEAKS now provides us with a potential method to improve longterm results of CLP patients. It might eventually lead to a more uniform treatment regime on
a national, or even international scale. Additionally, the variations and confounding variables
mentioned above can also be investigated on a large scale, regarding their influence on CLP
children’s intelligibility, thus enabling future studies to be able to handle the
variations/variables accordingly.
Additionally, PEAKS has the potential to be a useful screening tool, which can be regularly
and easily applied to diagnose speech intelligibility deficits in (CLP) patients. However, it is
important to realize, that PEAKS cannot replace the detailed speech analysis carried out by
the SLPs. Nevertheless, it can supply clinicians with a simple screening method, which saves
time and manpower, and moreover, produces objective results that are easily comparable.
Peterson-Falzone et al. (2010) state that
“Because treatment decisions will be influenced by the judgment of speech intelligibility,
any rating scale used by speech language pathologists should provide a useful description
of the child’s speech that is easily interpreted by other professionals.”
This is the case with the WR value calculated by the PEAKS program, as it is a percentage
that demonstrates the patients` degree of intelligibility, and can therefore be easily
interpreted. Once a problem is identified with the PEAKS program, an in depth diagnostic
evaluation may be initiated to discover the origin of the speech errors. The challenge of
sorting out the relative contribution of each etiological factor to the errors produced by the
child, using both perceptual speech evaluation and further instrumental assessments, can
then be tackled afterwards by the clinicians. Only after considering the results of a thorough
investigation can a suitable treatment be recommended. After treatment the PEAKS program
is useful as a quality check to see if the correct course has been taken. PEAKS could
therefore also offer the physicians and speech therapists, as well as the patient, a useful
feedback regarding the progress made throughout the extensive and lengthy therapy.
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5.8 Conclusion
In the present study, the PEAKS program has proven to be a valid method of evaluating
speech intelligibility, reaching a high correlation with our most experienced raters. This
study also investigated the influence of certain variables and different treatment
strategies/techniques on the speech intelligibility of CLP patients. We demonstrated that age
and the number of grommets inserted have a significant influence on the intelligibility of
CLP patients. As the sample size is relatively small, our study can be seen as a pilot for
further PEAKS investigations of CLP patients in the future. Moreover, and most
importantly, it demonstrates the great potential of the PEAKS program.
PEAKS has a high clinical value, as it can become a useful screening tool and feedback
mechanism during the extensive therapy of CLP patients. Of course not only CLP children,
but also other patients could be evaluated with PEAKS. It is a program that can be applied
on an every day basis either in a clinic or at schools, or even at home on the patient’s own
computer. This form of continual evaluation would allow for a timely diagnosis of speech
deficits and therefore also enable an appropriate intervention. The success of the intervention
could then be objectively evaluated afterwards.
PEAKS also shows potential in being an excellent research tool, especially in the field of
CLP speech diagnostics. PEAKS could provide the much-needed objective speech results
regarding the success of the varying treatment methods. Disparities in outcome between
centers may in the long-term lead to improvements of the various treatment strategies
involved. Furthermore, PEAKS could also evaluate the influence of confounding factors
involved in speech research of CLP patients, making useful data available for future studies.
The possible applications of the PEAKS program for research are abundant.
PEAKS has continuously demonstrated its ability to accurately assess speech intelligibility,
and hence proven itself as a valid speech evaluation method. Our conviction is that the
PEAKS program is a very promising tool, as it can generate objective results without the
need of professionals, and these calculated WR values can be compared even on an
international level. In this paper, we hope to have provided compelling arguments for the use
of the PEAKS program in the future of CLP speech diagnostics, if only as a complement to
perceptual evaluation.
75
6 Summary
Analyzing speech deficits of children with clefts of the lip alveolus and palate (CLP) has been
far from ideal in the past (Lohmander, 2004; Roberts, 1991). In this study, an objective
method named PEAKS was used to evaluate the intelligibility of CLP patients in a pilot study
involving 51 CLP patients treated in Freiburg. PEAKS is an automatic speech recognition
program, developed in Erlangen in 2006.
Craniofacial clefts are among the most frequent innate malformation (11-15%) (Hemprich,
2011). Consequences of a cleft are manifold, including speech (Kuehn, 2000) and hearing
(Bennett, 1972; Bluestone C. D., 1971; Paradise, 1975) disorders. Examining speech disorders
of CLP patients is deemed important (Lohmander, 2004) and the current state of the art
analysis involves the perceptive evaluation through multiple speech language pathologists
(Sell, 2005). However, this analysis has many disadvantages (Keuning, 1999; Maier, 2009;
Paal, 2005) and striving for a refinement of methodology for comparative studies has often
been recommended (Lohmander, 2004; Shaw W. C., 1992).
The validity and clinical value of the PEAKS program in investigating speech deficits of cleft
patients was examined in this study. The word recognition rate (WR) calculated by PEAKS
of the 51 Freiburg patients varied between 13.13% and 81.82% (arithmetic mean: 51.51% +/14.5%), whilst the control group adopted from a study of Dames and Maier (Dames 09)
achieved WR values between 31.3% and 85.9% (159 healthy children, arithmetic mean:
63.5% +/- 12.1%). The mean WR value of the control group was significantly higher
compared to that of the Freiburg patient collective (p <0.001), confirming the ability of the
PEAKS program to differentiate between “good” and “reduced” intelligibility. Three raters
with varying levels of experience also subjectively assessed the Freiburg patient collective.
The WR of the PEAKS program correlated strongly (r= -0.82) with the subjective evaluations
of the two most experienced raters. Palate cleft involvement, gender, timing of grommet
insertion and the treatment method (Freiburg vs. Erlangen) did not show a significant
influence on the WR values. WR values generally increased significantly with age (p <0.001)
and decreased after six grommet insertions (p= 0.023). The results of this study indicate that
PEAKS may be an important step forward in improving the speech evaluation of CLP
patients.
76
7 Zusammenfassung
Die Analyse von Sprachstörungen bei Kindern mit Lippen-Kiefer-Gaumen-Spalten (LKG)
war in der Vergangenheit bei weitem nicht optimal (Lohmander, 2004; Roberts, 1991). In
dieser Studie wurde eine objektive Methode, das sogenannte PEAKS Programm, eingesetzt,
um die Sprachverständlichkeit von 51 in Freiburg behandelten LKG Patienten in einem
Pilotprojekt zu evaluieren. PEAKS ist ein im Jahr 2006 in Erlangen entwickeltes
automatisches Spracherkennungsprogramm.
Kraniofaziale Gesichtsspalten zählen zu den häufigsten angeborenen Fehlbildungen (11-15%)
(Hemprich, 2011). Die Konsequenzen einer Spalte sind vielseitig, einschließlich Sprach(Kuehn, 2000) und Hörstörungen (Bennett, 1972; Bluestone C. D., 1971; Paradise, 1975). Die
Untersuchung der Sprachstörungen von LKG Patienten wird als wichtig erachtet (Lohmander,
2004) und bezieht nach heutigen Standards die perzeptive und somit subjektive Evaluation
mehrerer Sprachtherapeuten mit ein (Sell, 2005). Jedoch weist diese Analyse viele Schwächen
auf (Keuning, 1999; Maier, 2009; Paal, 2005), weshalb häufig empfohlen wurde, die
Methodik in Bezug auf die Vergleichbarkeit von Studien zu verfeinern (Lohmander, 2004;
Shaw W. C., 1992).
In dieser Studie wurde die Validität und die klinische Wertigkeit des PEAKS Programms bei
der Analyse von Sprachstörungen von LKG Patienten untersucht. Die durch das PEAKS
Programm ermittelte Worterkennungsrate (WR) variierte für die 51 Patienten aus Freiburg
zwischen 13,13% und 81,82% (arithmetisches Mittel 51,51% +/- 14,5%), während die aus
einer Studie von Dames und Maier (Dames 09) übernommene Kontrollgruppe WR Werte
zwischen 31,3% und 85,9% erreichte (159 gesunde Kinder, arithmetisches Mittel: 63,5% +/12,1%). Der durchschnittliche WR Wert war für die Kontrollgruppe signifikant höher als für
die Freiburger Patientengruppe (p <0,001), was die Fähigkeit des PEAKS Programms
zwischen “guter” und “verminderter” Sprachverständlichkeit zu unterscheiden bestätigt.
Zusätzlich wurde die Freiburger Patientengruppe subjektiv durch drei Gutachter mit
unterschiedlichem Erfahrungshintergrund bewertet. Die WR Werte des PEAKS Programms
zeigten eine gute Korrelation (r=-0,82) mit den subjektiven Bewertungen der beiden
erfahrendsten Gutachter. Weder Vorhandensein einer Gaumenspalte, Geschlecht, Zeitpunkt
der Paukenröhrcheneinlage noch die Behandlungsmethode (Freiburg im Vergleich zu
Erlangen) hatten einen signifikanten Einfluss auf die WR Werte. Die WR Werte nahmen mit
dem Alter signifikant zu (p<0,001) und bei sechs oder mehr Paukenröhrcheneinlagen
signifikant ab (p=0,023). Die Ergebnisse dieser Studie deuten darauf hin, dass PEAKS einen
wesentlichen Fortschritt in der Sprachanalyse von LKG Patienten darstellt.
77
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9 Appendix
9.1 Data Entry Form
The data entry form was subdivided into four different sections:

Basic Data: Name, date of birth, patient ID, type of cleft, additional illnesses, school
visited, ENT Doctor.

Speech data: Language of parents, dialect, logopedic treatment, language
development, articulation, vocabulary, syntax

Hearing data: Audiometer hearing test, acoustic impedance measurement.

Therapy: Cleft operation, ear operation, osteoplastic surgery, orthodontic treatment,
adenotomy, velopharyngoplastic
9.2
85
PEAKS Questionnaire
PEAKS
Dissertation:
Program
to
Evaluate
and
Analyze
all
Kinds
of
Speech
disorders
Name:
_________________
Datum:
____________
Vorname:
_________________
Fragebogen 1. Spaltart:__________________
OP:
Lippe________
Gaumen________
2. Sprache:
‐
Sprache
der
Eltern:
________________
Dialekt:
________________
‐
Sprachentwicklung:
Fällt
Ihnen
etwas
Spezielles
bei
der
Sprache
Ihres
Kindes
auf?
_________________________________________________________________
3. HNO
Arzt:
_____________________________
4. Logopädie:
ja nein ‐
Alter
bei
Beginn:
_____
Jahre
Alter
bei
Ende:
_____
Jahre
‐
Dauer:
_____Jahre
5. Ohr:
‐
Anzahl
der
Paukenröhrchen:
0 1 2 3 4 5+
‐
Aktuell
Paukenröhrchen:
ja nein
‐
Paukenerguss:
ja nein rezidivierend
‐
Letzter
Hörtest:
______________
6. Zahnspangen
Behandlung:
aktuell geplant abgeschlossen
Fest Lose
7. Soziales:
‐
KIGA/Schule
_________________________________
‐
Geschwister
_________________________________
Klinische Untersuchung ‐
Nase:
_______________________________________________________________
‐
Enoral:
______________________________________________________________
‐
Ohr:
Re_______________
Li_______________
Beurteilung ‐
Rhinophonie:
aperta clausa mixta normal ‐
‐
Note
der
Verständlichkeit:
1 2 3 4 5 ‐
Aussprache:
______________________________________________________________ ‐
WR:
___________
WA:___________
9.3
86
Vocabulary of the PLAKSS Test
Table 9-1 Original Target words of the PLAKSS test (Fox, 2002). For the extended vocabulary
see (Maier, 2009)