Download Pediatric cervicofacial actinomycosis – case

52 Jornal de0021-7557/01/77-01/52
Pediatria - Vol. 77, Nº1, 2001
Jornal de Pediatria
Copyright
© 2001 by Sociedade Brasileira de Pediatria
CASE REPORT
Pediatric cervicofacial actinomycosis –
case report and review of the literature
Fabiana Bononi,1 Antônio Vladir Iazzetti,2 Nasjla Saba da Silva3
Abstract
Objective: to emphasize important features in the diagnosis and monitoring of patients with childhood
cervical actinomycosis.
Patients and methods: we report the case of a patient with cervicofacial actinomycosis. We also carried
out a review of the literature from the past few years (Lilacs and Medline).
Results: we followed a male patient admitted to the pediatric infectious disease ward. Diagnosis was
carried out through biopsy of a cervical node and isolation of bacteria. Specific penicillin treatment for
actinomycosis was administered for 14 days. Subsequently, we observed remission of the node. The patient
was discharged from the hospital. At least 6 months of amoxycillin therapy with simultaneous outpatient
follow-up were recommended.
Conclusion: early diagnosis of actinomycosis enables appropriate and prompt treatment, thus
preventing the involvement of other areas such as CNS, face, and neck.
J Pediatr (Rio J) 2001; 77(1): 52-54: actinomycosis, Actinomyces israelli, cervicofacial bacterial
tumor.
Introduction
Actinomycosis is a granulomatous, chronic, and infectious disease with characteristics of suppuration and that is
produced by gram-positive bacilli of the Actinomyces strain.
These bacteria can be divided into six subgroups: israelii,
naeslundii, viscosis, odontolylicius, meyeri, and
pyogenes.1-5 The Actinomyces israelii is the bacillus that
most commonly and significantly affects humans.1,5,6
presented with complaints of neck lumps for the previous
three months at emergency services elsewhere. According
to the mother of the patient, he had first presented a
moderate edema on the anterior cervical region, more
specifically the medial part near the sternal-clavicular joint,
which was associated with mild local pain and loss of
appetite. Patient had presented spontaneous improvement
15 days after the onset of the lesion and had remained
asymptomatic for one month. Subsequently, the moderate
edema reappeared in the medial cervical region and was
associated with intense local pain, moderate hyperemia,
various daily episodes of fever that were not measured, and
mild dysphasia. Patient sought medical assistance and was
indicated to take symptomatic medication. The mother
reported not perceiving any improvement and, consequently,
brought the child to the state of São Paulo for medical
evaluation.
Case report
We report a case involving JRS, a male patient with 9
years and 10 months of age, with light-brown skin color, and
from the city of Jânio Quadros, state of Bahia. Patient
1. Pediatrician and Intern, Pediatric Infectology, UNIFESP-EPM.
2. Associate Professor, Department of Pediatrics, UNIFESP-EPM.
3. Director, Clinic of the Pediatric Oncology Institute, UNIFESP-EPM.
52
Pediatric cervicofacial actinomycosis... - Bononi F et alii
The patient presented at the Institute of Pediatric Oncology with anterior cervical mass. At the infirmary services
for pediatric oncology, a biopsy specimen was collected for
histopathologic examination. Next, patient was indicated
treatment with cephalexin and symptomatic medication. On
the 5th day of hospital stay, antibiotic therapy was replaced
with ceftriaxone. After one week of evolution, the Actinomyces was isolated in biopsy of specimen collected from the
patient. Patient was indicated to the department of Pediatric
Infectious Diseases and submitted to treatment specific for
actinomycosis with administration of crystalline penicillin
for 14 days. Patient did not present intercurrence during this
period and presented general improvement of clinical status. Patient also remained afebrile during the period and
was discharged from the hospital and indicated long-term
antibiotic therapy. The drug of choice was amoxacillin due
to its reduced cost and more suitable dosage. JRS was also
indicated for outpatient follow-up at the clinic for infectious
diseases.
We carried out a review of the literature on cervicofacial
actinomycosis of the last few years.
Discussion
Actinomycosis was first described in the 19th century as
a disease found in bovine animals. In 1876, Bellinger
identified the Actinomyces as a specific parasitic disease
after finding mycelia in purulent specimen taken from the
mandible of cattle. In 1877, the microbiologist Hartz, in
using the material collected by Bellinger, confirmed the
finding of radial microorganisms, which he named Actinomyces bovis (Akino for radial discharge of sulfur granules
and Mycos for mycelia). In 1981, Wolf and Israel isolated
an anaerobic organism in human dental caries presenting
growth of anaerobic oral filamentous organism. Currently,
Actinomyces are formally classified as bacteria.7
The etiologic agent of Actinomyces is a member of the
Actinomycetaceae family, which are colonizers of the oral
cavity and of the mucous membrane of other orifices.7 The
microorganism is classified as an intermediary between
fungus and bacterium.1,2,4,7,8 Actinomyces do not have
nuclear membranes, glycans, or mitochondria; they are
anaerobic or microaerobic, sensitive to antibacterials, and
do not respond to antifungal therapy.4
The bacterium is a colonizer of the oral cavity that rarely
causes infection, which indicates its low potential of virulence or invasion. The reduced frequency of Actinomyces
infection indicates that its inoculation through oral lesion
cannot be the only cause of infection. It has been described
that the Actinomyces requires the presence of other types of
bacteria in order to proliferate. In this sense, the oral flora
determines the potential of oxygen reduction that would
favor the growth of this anaerobic bacterium.1,2,4,7
Fifty-percent of the cases of the disease are of cervicofacial actinomycosis, 20% of pulmonary and thoracic acti-
Jornal de Pediatria - Vol. 77, Nº1 , 2001 53
nomycosis, and 30% of abdominal and pelvic actinomycosis.2,3,7,9-11 The disease affects the cervicofacial area starting at the buccal mucous membrane or pharyngeal membrane. It spreads to other areas in contact and its primary
lesion is usually located at the mandible.1,10 Predisposing
factors for cervicofacial actinomycosis include poor oral
hygiene, break in normal mucosal barriers, and anaerobic
medium.3 In approximately 75% of cases, onset of the
disease occurs on the teeth or tonsils. 2,9,12
Actinomycosis is an entity with universal distribution
and with equal frequency of cases in rural and urban areas.8
It is more common in male patients, for a ratio of 3:1 or
4:1,4,5 this due to more frequent maxillofacial lesions in
men as a result of sports, automobile and motorcycle
accidents, alcohol abuse, and aggressive behavior (does
this read somewhat sexist?). 5 It is reported only approximately once a year in major medical centers.10 Weese and
Smith, in 1975, reported an incidence of 1 case per 17,000
to 53,000 inhabitants.11
The disease disseminates to contiguous areas with subsequent involvement of structures in contact. Hematogenous dissemination, in turn, is described as rare. 3 The
pathology presents characteristics of induration of infected
tissue with a tendency to present abscedation and fistulation.2
The disease displays different clinical courses, ranging
from chronic to acute actinomycosis. The earlier is the most
common form, and it presents slow progression, indurate
infiltrate, and multiple abscesses and fistulas; the latter
presents fast progression with fever (50% of cases),10
ulceration, fluctuating lumps mimicking a typical pyogenic
infection.4
Clinically, the disease presents the 5 symptoms of
Poncet and Bérard: pain, trismus, lumps with fistulation,
discharge of sulfur granules, and moderate general symptoms. Local pain and fever are the most common symptoms.2 Regional adenopathy, in turn, is not common.7
Resulting severe involvement of the cranium and spinal
column can develop to meningitis and death. Invasion of the
orbit and ear, including the middle ear, have been described
as extremely rare.5 The main areas of infection are the face
and neck, including, also, the tongue, larynx, hypopharynx,
lachrymal glands, mandible, malar region, paranasal sinuses, palate, parotid gland, lachrymal canaliculus, and
periodontal abscess of the maxillae.
Early diagnosis of cervicofacial actinomycosis has been
reported in less than 10% of cases. The final diagnosis is
established according to bacterial growth in brain-heart
infusion agar or blood agar with 5% carbon dioxide and at
37º C. Diagnostic procedures should include 14-day anaerobic culture of pus and biopsy specimens with subsequent
antibiogram.4 The use of the culture medium for diagnosis
may prove unsuccessful in 50% of cases due to growth of
secondary, gram-negative bacteria, such as Streptococcus
and Staphylococcus; to a breach in the anaerobic medium;7
and to previous use of antimicrobials.2 Another method for
54 Jornal de Pediatria - Vol. 77, Nº1, 2001
the diagnosis of cervicofacial actinomycosis is the histopathologic examination of biopsy specimens, in which case
diagnosis is established by chronic granulomatous reaction
with fibrotic and avascular periphery and, also, sulfur
granules with central area presenting numerous polymorphonuclear, lymphocyte, and plasmatic cell structures and
measuring 0.1 to 1.0 mm.2,3,13,14
Other additional methods are tomography of the head
for assessment of bone involvement and of development of
the pathology; gallium scintigraphy for delimitation of the
natural inflammatory process (also a good method for easy
and effective follow-up procedures); ultrasonography for
differential diagnosis of natural inflammation and the disease process; and sialography for assessment of salivary
gland involvement.4
The disease should be included in differential diagnosis
of fungus infection, tuberculosis, Yersínea enterocolitica,
pseudo-appendicitis, osteomyelitis, amebiasis, hepatic abscess, chronic and bacterial infection, and nocardiosis. 3
In the history of treatments for actinomycosis, in 1938,
sulfonamides were the first antimicrobials used with successful results. Moreover, in 1948, Nichols and Herrell
were the first to use penicillin in the treatment for actinomycosis.4
The drug of choice is the crystalline penicillin. The
bacterial resistance to this antibiotic, however, is still not
well understood.3,10
Actinomyces produce extensive fibrotic reactions with
central necrotic lesion, resulting in hypovascular tissue with
low potential for oxygen reduction and limited antibiotic
penetration. Consequently, treatment requires high-dose,
long-term antibiotic therapy,4 it depends on individual
clinical and radiological improvement of patients, and it
should be administered intravenously for 3 to 6 weeks and
orally, after hospital discharge, for 6 to 12 months.3,4,7
Actinomyces are also sensitive to semi-synthetic penicillin, tetracycline, streptomycin, erythromycin, chloramphenicol, vancomycin, clindamycin, and lincomycin.3,5,15
In addition to antimicrobial therapy, surgical cleaning with
abscess drainage and excision and curettage of devitalized
tissue should be considered.7
It is estimated that approximately 90% of cases are
cured.11
Comments
The referred clinical case exemplifies the involvement
of the medial-anterior cervical region by Actinomyces with
late diagnosis following abscess drainage and collection of
biopsy specimen.
Pediatric cervicofacial actinomycosis... - Bononi F et alii
The actinomycosis is a rare pathology worldwide. Following the use of antibiotic therapy, especially with penicillin, this entity has become even more rare and less lethal
since it is sensitive to various antimicrobials.4
References
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MTG, Iazzetti AW, et al. Actinomicose cervicotorácica na
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3. Maxson S , Jacobs RF. Actinomicosis. In: Feigin RD , Cherry JD,
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palatinas, valor do exame histopatológico de rotina. Revista
Brasileira de Otorrinolaringologia 1981; 47:122-7.
10. Steward MG, Sulik M. Pediatric actinomycosis of the head and
neck. Ent Journal 1993; 72:614-9.
11. Weese WC, Smith MI. A study of 57 cases of actinomycosis over
a 36-year period. A diagnostic “failure” with good prognostis
after treatment. Arch Intern Med 1975; 135:1562-8.
12. Fernandez JMB, Fernandez MG. Actinomicose cervicofacial.
Presentación de 6 casos. Rev Cubana Estomatol 1994; 31:38-40.
13. Boor A, Jurkovic I, Friedmann I, Benicky M, Dubrikov K.
Pathology in focus actinomycosis of the middle ear. J Laryngol
Otol 1998; 112:800-1.
14. Piens MA, Patricot LM, Berger F, Bejui F. Les actinomycosis.
Étude anatomo-pathologique. Ann Pathol 1985; 5:167-72.
15. Martin MV. Antibiotic treatment of cervicofacial actinomicosis
for patients allergic to penicillin: a clinical and in vitro study. Br
J Oral Maxilof Surg 1985; 23:428-3.
Correspondence:
Dr. Antonio Vladir Iazzetti
Universidade Federal de São Paulo-Escola Paulista de
Medicina
Rua Loefgreen, 1998
CEP 0404-003 – São Paulo, SP - Brazil
Phone: +55 11 5732.0009 / 576.4117