Document 6477136

621
Disseminated Infection Due to Actinomyces meyeri: Case Report and Review
C. Apotheloz and C. Regamey
From the Service de Medecine, Hopital Cantonal, Fribourg, Switzerland
Actinomyces meyeri is rarely isolated in cases of actinomycosis. We present a case of disseminated
actinomycosis due to A. meyeri; the patient had an abscess of the lung, osteomyelitis of the tibia,
and multiple skin abscesses. Cure was achieved with surgical debridement and administration of
intravenous penicillin, followed by oral penicillin, for 1 year. A concomitant gram-negative bacillus,
Actinobacillus actinomycetemcomitans, was also isolated. Review of the literature revealed only 26
well-documented cases of infection with A. meyeri. Male adults are mainly affected, and alcoholism
is frequently the underlying condition in these patients. Associated bacteria were isolated in twothirds of these cases. In contrast to other species of Actinomyces, A. meyeri often causes pulmonary
infection and shows a tendency for hematogenous dissemination. Even though multiple organs are
involved, the outcome for these patients is excellent when penicillin is administered for several
months and surgical procedures are performed when necessary.
Case Report
A 47-year-old man presented to our hospital in mid-December 1993 with a painful effusion of the left knee. The collection
was drained for analgesic purposes, and no analysis of the fluid
was done at that time. Three weeks later his symptoms recurred,
and red, firm, painless subcutaneous nodules appeared on the
trunk and the extremities. On 16 January 1994, the patient
became febrile and was admitted. His medical history was
Received20 July 1995;revised 6 November 1995.
Reprintsor correspondence: Dr. C. Regamey, Servicede Medecine, Hopital
Cantonal, CH-1708 Fribourg, Switzerland.
Clinical Infectious Diseases 1996;22:621-5
© 1996 by The University of Chicago. All rights reserved.
1058-4838/96/2204-0003$02.00
significant only for alcohol abuse and smoking (he reported
that he consumed 60 g of ethanol and smoked three packs of
cigarettes daily). He had not brushed his teeth for 15 years.
Physical examination revealed a temperature of38.2°C; there
were no enlarged lymph nodes. Examination of the trunk and
extremities revealed red, firm, painless subcutaneous nodules,
some of which had started to form abscesses with fistulization
to the skin (figure 1). The left knee was swollen and painful
on mobilization but showed no sign of effusion. The patient
had dentogingival disease, with numerous caries and stumps.
Laboratory studies revealed that an inflammatory process
was present. The erythrocyte sedimentation rate was markedly
increased (117 mmlh; normal rate, 3-8 mm/h). The WBC
count was 13.3 X 109/L (normal count, 4-10 X 1091L) with
a left shift (14% band forms; normal, <8%), indicating leukocytosis. The platelet count was also elevated (466 X 109/L;
normal count, 150-300 X 109/L). The RBC count (2.95 X
1012/L; normal count, 4.5-5.5 X 1012/L) indicated severe anemia. The serum y-glutamyltransferase level was markedly increased (159 UIL; normal level, 50 U/L), and the levels of
aspartate aminotransferase and alanine aminotransferase were
slightly increased at 42 U/L (normal level, 40 U/L) and 51
U/L (normal level, <40 UIL), respectively; these abnormalities
in liver function were attributed to his alcoholism.
A chest radiograph revealed an infiltrative mass in the middle
lobe of the right lung, which was confirmed by a chest CT
scan (figure 2). The patient refused to undergo CT-guided needle biopsy. Conventional tomograms of the left knee demonstrated a significant osteomyelitic process in the proximal left
tibia (figure 3). An ultrasonogram of the abdomen showed
slight splenomegaly, and the liver was described as normal in
SIze.
One of the subcutaneous nodules was biopsied, and the histological findings were suggestive of disseminated actinomycosis: macroscopically, no sulfur granules were seen, but microscopically, chronic purulent inflammation in association with
radiating microorganisms was observed. Three stains (gram,
periodic acid-Schiff, and Grocott-Gomori methenamine-silver
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The term actinomycete, which is of Greek origin, means
ray fungus; however, actinomycosis is due to a gram-positive
anaerobic bacterium. Bollinger [1] described the microorganism in 1877 as the causative agent of lumpy jaw in cattle and
named it Actinomyces bovis [1]. In 1891, Wolff and Israel [2]
grew a similar microorganism from a pulmonary abscess and
named it Actinomyces israelii [2]. Although most cases of human actinomycosis are due to A. israelii, less common agents
are Actinomyces naeslundii, Actinomyces viscosus, Actinomyces odontolyticus, Actinomyces meyeri, and Propionibacterium
propionicum.
A. meyeri was first isolated from a lung abscess in 1911 by
Meyer, who described it as a Streptothrix [3]. The microorganism was then reclassified as Actinobacterium meyeri in 1938
[4]; it was finally classified in the order Actinomycetales in
1977 [5]. This rare actinomycete has been recovered from a
minority of patients, and the spectrum of infections it causes
is not well known, although it shares all the microbiological
characteristics of the organisms in this order. We describe a
case of disseminated actinomycosis due to A. meyeri.
622
Apotheloz and Regamey
CID 1996;22 (April)
Figure 1. Skin abscess on the wrist of a patient with disseminated
actinomycosis due to Actinomyces meyeri.
Figure 3. Tomogram of the left knee of the patient in figure 1,
showing osteomyelitis of the tibia.
subcutaneous abscesses disappeared within 2 weeks, and the
pulmonary lesion regressed within 4 months. The parameters
indicative of inflammation normalized within 4 weeks. Multiple
dental extractions were done under general anesthesia. The
patient underwent surgical debridement and synovectomy of
the left knee, and 1 year later he underwent surgical reconstruction of this joint. All tissue biopsy specimens were sterile, and
as the parameters that had indicated inflammation were within
normal limits at that time, the antibiotic therapy was stopped.
The patient continues to do well.
Discussion
Figure 2. Chest CT scan of the patient in figure 1 shows infection
of the right lung, which typically extends to the pleura.
The literature from 1960 to 1995 was searched with use of
MEDLINE and Index Medicus to identify reports of human
cases of actinomycosis due to A. meyeri. Because of the taxonomic changes over the years, we used the terms Actinomyces
meyeri, Actinobacterium meyeri, and Streptothrix meyeri. We
found 26 well-documented cases of human infection with
A. meyeri (table 1). Before 1960 a few cervicofacial and pulmo-
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nitrate) were positive. The organisms were subsequently cultured and identified as Actinomyces on the basis of the results
of gas-liquid chromatography, which demonstrated the presence of large amounts of succinic acid in prereduced anaerobically sterilized broth [5]. The species was identified as A. meyeri with use ofthe Rapid ANA II system (Innovative Diagnostic
Systems, Atlanta). A gram-negative bacillus also was recovered
from the same subcutaneous abscess and was identified as Actinobacillus actinomycetemcomitans by means of biochemical
tests.
Treatment with intravenous penicillin G (20 million units
per day for 8 weeks) was started and was then switched to oral
penicillin V (3 million units per day for 12 months). All the
Table 1.
Reference
Summary of 26 cases of actinomycosis due to Actinomyces meyeri that have been reported between 1960 and 1995.
Case no.
[6]
[7]
623
Disseminated Actinomyces meyeri Infection
CID 1996;22 (April)
2
Pathological findings
Treatment
Duration of
antibiotic therapy
(mo)
M/62
Pneumonia, empyema, skin
abscess
Penicillin, surgical drainage
12
Ml16
Empyema, hematogenous
spread to bone marrow
Pneumonia, osteomyelitis
of tibia, skin abscess
Chronic osteomyelitis of
tibia and fibula, skin
abscesses
Spondylitis, skin abscesses,
suspected pericarditis
Pyomyositis of calf,
pleuropulmonary
actinomycosis
Bilateral pneumonia, liver
abscess, mesenteric
abscesses
Pneumonia, brain abscess,
multiple skin abscesses
Pneumonia, skin abscess
Clindamycin, rib resection,
chest-tube drainage
Penicillin
6
Penicillin
4
None
Penicillin, surgical drainage
4
None
Penicillin, surgical drainage
NA
None
Sex of patientJ
age (y)
Ml40
[8]
4
M/49
[9]
5
M/34
[10]
6
Ml58
[11]
7
M/35
[12]
8
M/44
[13]
9
M/46
[PRJ
10
Ml47
[14]
[6]
11
12
F170
Ml49
[15]
13
MIS5
[16]
14
Mll3
[17]
[18]
15
16
M/45
M/40
Asymptomatic mass in
lung
Pneumonia with cervical
extension
Bilateral pneumonia
Liver abscess
[19]
17
M/30
Liver abscess
[20]
18
F/49
Liver abscess
(21]
19
M/50
[22]
20
MIS0
[23]
[24]
21
22
F/46
F/29
Chronic osteomyelitis of
tibia
Osteomyelitis of first
metatarsus
Spondylodiskitis
Recurrent breast abscess
[24]
23
F/36
Recurrent breast abscess
[25]
24
M/64
Cervical abscess
[26]
25
M/24
[27]
26
F/28
Cervicofacial
actinomycosis
Brain abscess
NOTE. NA = not available; PR == present report.
Pneumonia, skin abscesses,
osteomyelitis of the tibia
Pneumonia
Pneumonia, empyema (6
mo later)
Fusobacterium nucleatum,
Bacteroides ureolyticus,
Streptococcus
constellatus,
Peptococcus species,
Propionibacterium
species
None
None
Penicillin, percutaneous
drainage
12
Actinobacillus
actinomycetemcomitans
Amoxicillin
12
A. actinomycetemcomitans
Penicillin, surgical
debridement
Penicillin, surgical
debridement
Penicillin
Clindamycin, tetracycline,
chest-tube drainage
4
Capnocytophaga species
12
A. actinomycetemcomitans
NA
6
Lobectomy
None
A. actinomycetemcomitans.
B. ureolyticus,
Peptococcus prevotii
Eikenella corrodens
Penicillin
None
Ceftriaxone, penicillin
Penicillin, clindamycin,
percutaneous drainage
Penicillin + clindamycin,
then doxycycline
Penicillin + clindarnycin,
then penicillin;
percutaneous drainage
Penicillin, sequestrectomy
Antibiotics, surgical
debridement
Pefloxacin + rifampin
Ampicillin, doxycycline,
surgical debridement
Tetracycline, doxycycline,
surgical debridement
Antituberculous therapy
(three agents)
Penicillin, surgical
debridement
Penicillin, neurosurgical
drainage
12
1
Actinomyces israelii
Streptococcus milleri
F. nucleatum
NA
Streptococcus anginosus
4
Bacteroides species,
Streptococcus mitis
Propionibacterium acnes
NA
4
None
Diverse anaerobes
4
Diverse anaerobes
NA
A. actinomycetemcomitans
9
None
2
Streptobacillus
moniliformis
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[9]
Coinfecting organism
624
Apotheloz and Regamey
Cervicofacial actinomycosis, which accounts for approximately one-half of cases of actinomycosis, is the commonest
form and results from contiguous extension of disease from
the oral cavity [30]. Such infection due to A. meyeri has been
reported for only two (8%) of 26 patients, but, for two reasons,
we suspect that it occurs more often. In the majority of cases,
the diagnosis of actinomycosis is based on the clinical history,
on typical histological findings, or on culture of Actinomyces
species, and identification of the exact microorganism is rarely
required in clinical practice [25]. In addition, many cases of
lumpy jaw due to A. meyeri will not appear in the literature,
while a case of systemic infection due to the same microorganism will certainly be published.
Pulmonary actinomycosis accounts for 15% of all cases of
actinomycosis [30] and generally is caused by aspiration of
oral secretions when dental disease is present. Infections with
A. meyeri involved the lung in 13 (50%) of 26 cases. It is
unclear why this clinical presentation was so preponderant,
although the relative rarity of reports of the cervicofacial form
of the infection probably causes a statistical bias.
Abdominal actinomycosis is reported in about 20% of cases,
and liver involvement is infrequent [30]. We found four cases
of liver abscess due to A. meyeri. Three were isolated lesions
that were probably the result of the seeding, via the portal vein,
of an occult focus of infection along the gastrointestinal tract
[18-20]. No abdominal site other than the liver was infected
by A. meyeri.
Actinomycosis of the CNS is rare; A. meyeri was recovered
in two cases of brain abscess, which is the most common
clinical presentation of CNS actinomycosis [30].
Like other actinomycetes, A. meyeri is susceptible to most
antibiotics, and penicillin remains the most cost-effective drug
for treating infections due to this organism. Clindamycin, tetracyclin, erythromycin, or ceftriaxone may be alternatives [30].
Because actinomycosis leads to the formation of scarred, amorphous tissue into which the penetration of antibiotics is poor,
a prolonged course of antibiotic therapy (6-12 months) has
been recommended [30], but the duration of therapy remains
controversial. In our series, the mean duration of antibiotic
therapy was 6 months (range, 1-12 months) (table 1). It is
interesting that no recurrence of infection was observed among
10 patients treated for 3 -4 months. The associated bacteria
were generally susceptible to the antibiotics used against
A. meyeri. Five strains of the associated organisms had variable
susceptibility, and one was resistant to the antibiotic regimens
used, but even so, cure was achieved. These data are insufficient
to conclude whether the presence of coinfectants may lead to
treatment failure. The outcome was favorable for all but one
of the 26 patients treated for A. meyeri actinomycosis [6].
Conclusion
Our patient had disseminated actinomycosis due to A. meyeri, which involved the lung, bone, and skin. Cure was achieved
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nary infections due to this actinomycete had been described,
but the reports were not detailed [28, 29].
Infections due to A. meyeri are more prevalent among males
(the male-to-female ratio is 20:6), and the mean age of patients
with this illness is 42.5 years (range, 13-70 years). Seventy
percent of the patients in the present report were between 30
and 60 years old. The male-to-female ratio and mean age are
similar for patients with A. meyeri infection and for those with
other types of actinomyces infections [30].
Actinomycetes are oral saprophytes that particularly affect
periodontal lesions and carious teeth [30]. As with other actinomycoses, dentogingival disease is a major risk factor for the
development of infections due to A. meyeri: 18 (69%) of the
26 patients in this review had poor dentition or a recent history
of tooth extraction. Alcoholics are at risk for pulmonary actinomycosis because they often have poor dentition, and they are
prone to aspiration of oral secretions. In our series, 11 patients
(42%) were alcoholics; 10 of these patients had pulmonary
actinomycosis.
Actinomycosis can develop at the site of surgical procedures
[30]; previous surgery at the site of infection due to A. meyeri
was noted in two cases [21, 23]. However, it remains unclear
whether infection occurred as a result of direct contamination
or as a result of hematogenous spread of the oropharyngeal
flora secondary to intubation maneuvers.
Two-thirds (65%) of the infections with A. meyeri were
polymicrobial, and up to five different bacteria have been cultured from an infected site (table 1). It is of interest that all of
these associated pathogens, except one, also belonged to the
human oropharyngeal flora. Anaerobic bacteria or facultative
anaerobic bacteria predominated in these infections, and
A. actinomycetemcomitans, an aerobic gram-negative bacillus
that classically is isolated along with Actinomyces species, was
recovered in only five cases. Hypothetical mechanisms by
which associated bacteria may enhance the pathogenicity of
Actinomyces species include reduced oxygen tension and anaerobiosis-induced inhibition of phagocytes [30].
Of 26 infections with A. meyeri, we found that 10 (38%)
were disseminated (defined as the involvement of two distant
organs). The lungs, the skin, the long bones, the liver, the brain,
and the muscles were the sites involved, in order of frequency.
Disseminated actinomycosis has been described on rare occasions [30], but according to one study, dissemination may actually occur more frequently [31]. The propensity of A. meyeri
to disseminate is difficult to explain because this organism
does not differ from other actinomycetes in its microbiological
characteristics. It has been postulated that pulmonary infection
is often the source of the hematogenous dissemination [32].
This assumption is supported by the observation that in eight
of 10 cases of disseminated actinomycosis due to A. meyeri,
pulmonary infection was present. Because most of the patients
had dental disease, direct hematogenous spread from the oral
cavity also may have caused disseminated infection as well as
localized actinomycosis [9, 22, 24].
CID 1996;22 (April)
CID 1996;22 (April)
Disseminated Actinomyces meyeri Infection
with a regimen consisting of an 8-week course of intravenous
penicillin followed by a 12-month course of oral penicillin, in
addition to surgical debridement of an osteomyelitic focus.
Infections with A. meyeri are rare; however, they are probably underdiagnosed because identification of the exact Actinomyces species is not necessary for adequate treatment. Dentogingival disease and alcoholism are important risk factors that
permit the development of pulmonary infection via aspiration
of mouth flora, including A. meyeri. Hematogenous seeding to
distant organs occurs particularly frequently with A. meyeri,
compared with other actinomyces, and often originates from a
pulmonary focus of infection that should be carefully sought.
Treatment with penicillin for several months, coupled with
surgical procedures when necessary, results in a good prognosis, even when localizations are multiple.
The authors thank Professor J. Wust, Institut fur medizinische
Mikrobiologie der Universitat Zurich (Zurich), for identifying
A. meyeri and Dr. D. Fracheboud, Institut de Microbiologie et
d'Hygiene, Fribourg (Fribourg, Switzerland), for identifying A.
actinomycetemcomitans.
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Acknowledgments
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