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CASE REPORT
Nine-Month-Old Patient With Bilateral Earlobe Keloids
AUTHORS: Michael H. Tirgan, MD,a Christopher M. Shutty,
DO,b and Tae Hwan Park, MDc
abstract
aKeloid
Keloid disorder (KD) is a fibroproliferative ailment of the cutaneous
connective tissue secondary to dysregulation in various skin repair
and healing processes. This disorder is characterized by excess collagen and/or glycoprotein depositions in the dermis. Age of onset
of KD is not well documented. Based on clinical observations, various
authors have reported the onset of KD to be between the ages of 10 and
30 years. We report on an African American female who developed bilateral auricular keloids at the age of 9 months. To our knowledge, this
is the youngest age at which a patient has been documented to have
developed KD. Pediatrics 2013;131:e313–e317
Research Foundation, New York, New York; bDepartment
of Pathology, Staten Island University Hospital, Staten Island, New
York; and cDepartment of Plastic and Reconstructive Surgery,
Kangbuk Samsung Hospital, Sungkyunkwan University School of
Medicine, Seoul, Korea
KEY WORD
keloid
ABBREVIATION
KD—keloid disorder
www.pediatrics.org/cgi/doi/10.1542/peds.2012-0075
doi:10.1542/peds.2012-0075
Accepted for publication Aug 9, 2012
Address correspondence to Michael H. Tirgan, MD, Keloid
Research Foundation, 23 West 73rd St, Suite GD, New York, NY
10023. E-mail: [email protected]
PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).
Copyright © 2013 by the American Academy of Pediatrics
FINANCIAL DISCLOSURE: The authors have indicated they have
no financial relationships relevant to this article to disclose.
FUNDING: No external funding.
PEDIATRICS Volume 131, Number 1, January 2013
e313
Keloids are benign skin tumors that form
as a result of injury or inflammation of
the dermis, typically in individuals who
are genetically predisposed to this disorder. Rarely, keloids may develop de
novo, without a previous noticeable irritation of the skin.
On June 19, 2009, at the age of 3.5 years,
the patient underwent surgery under
general anesthesia, and her keloids
were excised from both earlobes. The
tissue specimens were examined
pathologically and confirmed to be KD
(Figs 2 and 3).
Age of onset of keloid disorder (KD) is
not well documented. Based on clinical
observations, various authors have
reported the onset of KD to be between
the ages of 10 and 30 years.1–3 Mode of
inheritance of KD is not known. Several
theories have been proposed, including autosomal recessive,4 autosomal
dominant with incomplete penetrance,5
and variable expression.
Postoperatively, bilateral pressure earrings were prescribed, but they were
not well tolerated and not used. Within
2 months of surgery, both keloids relapsed, and despite several intralesional
triamcinolone injections, they continued
to grow, mostly anteriorly. On October 6,
2011, at the age of 5 years, the patient
presented to the corresponding author.
On examination, she was found to have
gross bilateral anterior earlobe keloids
(Figs 4 and 5), with minimal posterior
earlobe keloids at the previous surgical
sites.
Keloids are characterized by formation
of excess scar tissue beyond the
boundaries of the precipitating wound.
Although benign, keloids can cause
aesthetic or functional problems, all
of which negatively affect the individual’s quality of life. Our patient developed bilateral auricular keloids when
she was 9 months old, subsequent to
earlobe piercing that had been performed at the age of 3 months. At 6.5
years of age, she is still challenged by
this disorder.
PATIENT PRESENTATION
The female patient described here was
born to African American parents on
January 31, 2006. At 3 months of age, the
pediatrician pierced her earlobes. No
immediate complications or infections
followed. When the child was 9 months
old, her mother detected hard lumps in
both earlobes. On October 18, 2006, the
child’s pediatrician noticed and documented keloids in the posteriors of
both earlobes. Over the next several
months, the keloids grew gradually. At
3 years of age, the patient was referred for surgical removal of the
keloids. Figure 1, the only extant preoperative photograph, depicts the
appearance of the left earlobe keloid
before surgery.
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TIRGAN et al
The child had normal growth and development. She was breastfed until the
age of 9 months. She had no other
keloids. The only known family history of
KD was found in the child’s mother.
A single, dark spot on her left upper
arm, on examination, had the appearance of a small, flat keloid.
With parental consent, the child underwent cryotherapy with monthly
topical applications of liquid nitrogen to
her keloids under local anesthesia.
Within 5 months, and after receiving 2
treatments to the right anterior earlobe
keloid and 3 treatments to the left anterior earlobe keloid, both keloids
nearly flattened (Figs 6 and 7). Magnetic pressure disks were prescribed
at this juncture; however, they were not
well tolerated and were not used. By
mid–June 2012, both keloids showed
evidence of early relapse. Cryotherapy
was resumed in late June 2012.
DISCUSSION
This case is a true representation of
a disorder that is difficult to treat. Although keloids can occur at any age,
they tend to develop more rapidly
FIGURE 1
Image of left posterior auricular keloid at 3 years
of age before its surgical removal.
FIGURE 2
Histologic specimen of the child’s earlobe keloid
depicting typical collagen bundles.
FIGURE 3
Histologic specimen of the child’s earlobe keloid
depicting typical collagen bundles, higher magnification.
during and after puberty. In recent
studies by Park et al6–8 that reported on
a large cohort of keloid patients from
Asia, the youngest subject described
was a 10-year-old boy with auricular
CASE REPORT
in November 2011 by the corresponding
author to capture detailed information
directly from patients.13 Of the 339
patients with KD who had completed
this survey as of July 11, 2012, only 1
reported development of first keloid at
the age of 1 year; 2 patients reported
developing their first keloids at the age
of 2 years; and 1 patient reported keloid development at the age of 4 years.
FIGURE 4
Right earlobe keloid at presentation to corresponding author when patient was 5 years of age.
FIGURE 7
Left earlobe keloid after treatment.
FIGURE 5
Left earlobe keloid at presentation to corresponding author when patient was 5 years of age.
velopment varied from 5 to 52 years.
Sanders et al10 presented data on 15
patients with keloids in the head and
neck region. Among them, a 10-year-old
boy with preauricular keloids was the
youngest. Orimolade et al11 described
the case of a 6-year-old boy who had
developed keloids in the pretibial area
subsequent to chronic osteomyelitis
with purulent discharge. Bermueller
et al12 reported treatment results for
33 patients with earlobe keloids; the
youngest patient in their series was 10
years old. The aim of their study was to
present long-term results in patients
with auricular keloids after surgical excision and/or medical therapy by corticoid injection. A retrospective study at
an academic tertiary referral center is
presented.
A recent keloid survey (institutional
review board approved) was launched
TABLE 1 Age Distribution of 883 Patients
With Auricular Keloids
FIGURE 6
Right earlobe keloid after treatment.
keloids. The age distribution of patients
with auricular keloids from their
studies is described in Table 1.
Clark et al9 studied 5 families with a
total of 35 affected individuals with various keloid phenotypes and locations.
The ages reported for first keloid dePEDIATRICS Volume 131, Number 1, January 2013
Age
10–19 y
20–29 y
30–39 y
40–49 y
50–59 y
60–69 y
70–79 y
Total
From Park et al.
Distribution
No.
%
79
664
96
29
12
2
1
883
8.95
75.20
10.87
3.28
1.36
0.23
0.11
100
Ear piercing is by far the leading cause
of earlobe keloid formation in genetically predisposed individuals. Ear
piercing is a widespread practice,
commonly performed by nonmedical
personnel in jewelry shops, stores, and
malls. Parents often choose to have
their daughters’ earlobes pierced at
very early ages. The practice of ear
piercing has never been the subject of
scientific research. The results of a recent online consumer survey,14 however, revealed that 38% of participants
claimed to have had their infant
daughters’ ears pierced. The same
survey indicated that 52% of parents
had chosen to have the piercings performed at jewelry stores; 19% had chosen body-piercing parlors; and only 13%
had elected to have the procedure performed at a physician’s office (Table 2).
Lane et al15 reported that keloids are
more likely to develop when ears are
pierced after age 11 years than when
they are pierced before that age, even
in patients with a family history of KD.
He recommended that patients with
a family history of keloids should consider avoiding ear piercing altogether,
and, if this is not an option, piercing
should be considered during early,
rather than late, childhood.
Although the conclusion of Lane et al15
holds true in a great majority of patients,
the case we have presented here defies
this finding and testifies to the continuing gaps in our understanding of the
pathogenesis and genetics of KD.
Some authors have suggested that
the typical latency in earlobe keloid
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TABLE 2 Practice of Ear Piercing Among
Young Females: Results of an
Online Consumer Survey (Data as
of July 11, 2012)
Percentage of parents whose
daughters do or do not have
pierced ears (n = 9836)
Yes
No
Age at ear piercing (n = 4428)
,2 y
2–3 y
4–5 y
6–7 y
$8 y
Location where the ear
piercing was performed (n = 4296)
Jewelry store
Body-piercing parlor
Physician’s office
Other
%
38
62
48
9
12
9
22
52
19
13
16
development may be due to hormonal
stimulation at puberty16 or during
pregnancy.17 Clearly, neither of these
conditions applied to this patient. One
can only hypothesize that the breastfeeding and passage of mother’s hormones to her infant daughter may have
played a role in the young child’s development of keloids.
KD’s true incidence and prevalence in
the United States are unknown.9 To the
best of our knowledge, no population
study has ever been undertaken to assess this disorder’s epidemiology. Based
on clinical observations, we do know
that the disorder is most common
among Africans and African Americans,16,18 Asians, and Indians from India.
The pathogenesis of KD is for the most
part unknown and poorly understood,
and beyond the scope of this article. The
acknowledged culprit in keloid formation, however, is the wounding of the
skin and subsequent dysregulated
wound healing, a complex, dynamic
process that normally results in the
restoration of the skin’s anatomic
continuity and function.
The tendency of earlobe keloids to
worsen after surgical excision (as exemplified in this case) is most probably
secondary to the retriggering of the
same dysregulated wound-healing
mechanisms by the excision itself: a
new dermal injury more extensive than
the original wound from the piercing.
Keloids may develop in an earlobe’s
anterior, its posterior, or in both sides.
They may appear as pedunculated,
sessile mononodular, or sessile multinodular, or they can take a complex
form. Keloid nodules may be superficial
or have a root and be buried deep inside the earlobe tissue. Generally, it is
agreed that with surgery alone, the
recurrence of earlobe keloids is unacceptably high; therefore, multimodality
treatment protocols have been proposed
to reduce the recurrence rate.19–24
Most therapeutic approaches to earlobe keloids incorporate surgical excision with immediate postoperative
steroid injections, followed by some
form of pressure device, such as
a pressure earring or magnetic disk.
Cryotherapy for earlobe keloids as
monotherapy has also been reported as
effective.25 On keloids’ recurrence after
a multimodal approach, treatment
becomes challenging. As shown in this
case, cryotherapy can be used to
debulk such recurrent keloids.
Although some practitioners use adjuvant radiation therapy to reduce
keloids’ recurrence rate after surgery,16 this modality carries an unacceptable risk of inducing secondary
neoplasms26 and should therefore be
avoided in treating pediatric and young
adult patients.
CONCLUSIONS
Thorough and scientific population and
epidemiology studies of KD are needed.
Scant data exist concerning KD’s incidence and prevalence in children
aged ,10 years. The case presented
here is the youngest patient ever
reported to have developed keloids.
Ear piercing is the leading cause of
earlobe keloid formation in the young.
Although most ear piercings take place
outside of physicians’ offices, many
pediatricians offer this service. Given
the difficulty of treating keloids, pediatricians need to be aware of the risk
of keloid formation and, at a minimum,
should obtain a thorough family history
of KD, especially from African American
and Asian children, before performing
ear-piercing procedures.
ACKNOWLEDGMENT
The corresponding author obtained parental consent for publication of this case
report and the accompanying images.
REFERENCES
1. Marneros AG, Norris JE, Olsen BR, Reichenberger E. Clinical genetics of familial keloids.
Arch Dermatol. 2001;137(11):1429–1434
2. Bayat A, Arscott G, Ollier WE, McGrouther
DA, Ferguson MW. Keloid disease: clinical
relevance of single versus multiple site
scars. Br J Plast Surg. 2005;58(1):28–37
3. Seifert O, Mrowietz U. Keloid scarring:
bench and bedside. Arch Dermatol Res.
2009;301(4):259–272
e316
TIRGAN et al
4. Omo-Dare P. Genetic studies on keloid.
J Natl Med Assoc. 1975;67(6):428–432
5. Bloom D. Heredity of keloids; review of the
literature and report of a family with
multiple keloids in five generations. N Y
State J Med. 1956;56(4):511–519
6. Park TH, Seo SW, Kim JK, Chang CH. Outcomes of surgical excision with pressure
therapy using magnets and identification
of risk factors for recurrent keloids.
Plast Reconstr Surg. 2011;128(2):431–
439
7. Park TH, Seo SW, Kim JK, Chang CH. Clinical
characteristics of facial keloids treated with
surgical excision followed by intra- and
postoperative intralesional steroid injections.
Aesthetic Plast Surg. 2012;36(1):169–173
8. Park TH, Seo SW, Kim JK, Chang CH. Management of chest keloids. J Cardiothorac
Surg. 2011;6:49
CASE REPORT
9. Clark JA, Turner ML, Howard L, Stanescu
H, Kleta R, Kopp JB. Description of familial
keloids in five pedigrees: evidence for
autosomal dominant inheritance and
phenotypic heterogeneity. BMC Dermatol.
2009;9:8
10. Sanders KW, Gage-White L, Stucker FJ.
Topical mitomycin C in the prevention of
keloid scar recurrence. Arch Facial Plast
Surg. 2005;7(3):172–175
11. Orimolade EA, Olabanji JK, Oladele AO, Yusuf
MB. Chronic osteomyelitis in the lower extremity predisposing to the unusual formation of keloids. Singapore Med J. 2011;
52(9):e190–e193
12. Bermueller C, Rettinger G, Keck T. Auricular
keloids: treatment and results. Eur Arch
Otorhinolaryngol. 2010;267(4):575–580
13. Tirgan MH. Keloid online survey. Web
based investigation of natural history of
keloid. Available at: www.keloidsurvey.
com
14. BabyCenter. When are girls ready for pierced
ears? Available at: www.babycenter.com/
viewPollResults.htm?pollId=74866
PEDIATRICS Volume 131, Number 1, January 2013
15. Lane JE, Waller JL, Davis LS. Relationship
between age of ear piercing and keloid formation. Pediatrics. 2005;115(5):1312–1314
16. Gauglitz GG, Korting HC, Pavicic T, Ruzicka T,
Jeschke MG. Hypertrophic scarring and
keloids: pathomechanisms and current and
emerging treatment strategies. Mol Med.
2011;17(1-2):113–125
17. Moustafa MF, Abdel-Fattah MA, Abdel-Fattah
DC. Presumptive evidence of the effect of
pregnancy estrogens on keloid growth.
Case report. Plast Reconstr Surg. 1975;56
(4):450–453
18. Froelich K, Staudenmaier R, Kleinsasser N,
Hagen R. Therapy of auricular keloids: review of different treatment modalities and
proposal for a therapeutic algorithm. Eur
Arch Otorhinolaryngol. 2007;264(12):1497–
1508
19. Rosen DJ, Patel MK, Freeman K, Weiss PR. A
primary protocol for the management of
ear keloids: results of excision combined
with intraoperative and postoperative steroid injections. Plast Reconstr Surg. 2007;
120(5):1395–1400
20. Savion Y, Sela M. Prefabricated pressure
earring for earlobe keloids. J Prosthet
Dent. 2008;99(5):406–407
21. Vachiramon A, Bamber MA. A U-loop pressure clip for earlobe keloid. J Prosthet
Dent. 2004;92(4):389–391
22. Yencha MW, Oberman JP. Combined therapy in the treatment of auricular keloids.
Ear Nose Throat J. 2006;85(2):93–94,
96–97
23. Aköz T, Gideroglu K, Akan M. Combination of
different techniques for the treatment of
earlobe keloids. Aesthetic Plast Surg. 2002;
26(3):184–188
24. Chang CH, Song JY, Park JH, Seo SW. The
efficacy of magnetic disks for the treatment of earlobe hypertrophic scar. Ann
Plast Surg. 2005;54(5):566–569
25. Fikrle T, Pizinger K. Cryosurgery in the
treatment of earlobe keloids: report of
seven cases. Dermatol Surg. 2005;31(12):
1728–1731
26. Botwood N, Lewanski C, Lowdell C. The risks
of treating keloids with radiotherapy. Br J
Radiol. 1999;72(864):1222–1224
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