Scandinavian Journal of Infectious Diseases
ISSN: 0036-5548 (Print) 1651-1980 (Online) Journal homepage: http://www.tandfonline.com/loi/infd19
Disseminated Actinomycosis due to Actinomyces meyeri and Actinobacillus actinomycetemcomitans Ed J. Kuijper, Henri O. Wiggerts, Gwan J. Jonker, Klaus P. Schaal & Jan De Gans To cite this article: Ed J. Kuijper, Henri O. Wiggerts, Gwan J. Jonker, Klaus P. Schaal & Jan De Gans (1992) Disseminated Actinomycosis due to Actinomyces meyeri and Actinobacillus actinomycetemcomitans, Scandinavian Journal of Infectious Diseases, 24:5, 667-672, DOI: 10.3109/00365549209054655 To link to this article: http://dx.doi.org/10.3109/00365549209054655
Published online: 08 Jul 2009.
Submit your article to this journal
Article views: 14
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Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=infd19 Download by: [La Trobe University]
Date: 23 May 2016, At: 19:39
Scand J Infect Dis 24: 667-672. 1992
CASE REPORT
Disseminated Actinomycosis due t o Actinomyces meyeri and Acti nobacillus acti nomycetemcom itans ED J . KUIJPER’, HENRI 0. WIGGERTS2, GWAN J. JONKER3, KLAUS P. SCHAAL‘ and JAN DE GANS’
Downloaded by [La Trobe University] at 19:39 23 May 2016
From the Departments of ‘Bacteriology. ’Neurology, and ’Pulmonology, Academic Medrt ill Centre, Amsterdam, The Netherlands, and ‘The Institute for Medical Microbiology and Immunology, University of Bonn, Bonn, Germany
A 44-year-old man presented with pulmonary lesions and neurological symptoms suggestive of lung carcinoma with cerebral metastases. He had non-specific chest X-ray findings since 6 years and he also suffered from relapsing purulent skin lesions which resolved spontaneously or by short courses of antibiotic treatment. When corticosteroids were given, multiple subcutaneous swellings developed that spontaneously ruptured. The pus contained Actinomyces meyeri and Actinobacillus actinomycetemcomitans. On operation, the intracerehral lesions appeared to be abscesses and the same bacteria were cultured as from the skin lesions. Bronchoscopical examination did not reveal a diagnosis. Amoxicillin was given for 12 months and the patient recovered.
E . J . Kuijper, MD, Department of Bacteriology, L1,Academic Medical Centre, Meibergdreef 9 , 1105 AZ, Amsterdam, The Netherlands
INTRODUCTION Actinomycosis is characterized as an opportunistic, chronic, suppurative, and granulomatous infection with abscess formation and draining sinus tracts (1-3). The primary sites of infection are the cervicofacial region, the thorax, and the abdomen and pelvic region. Human actinomycosis is caused mainly by Actinomyces israelii and A. gerencseriae followed in order of importance by Propionibacterium propionica, A. naeslundii, A. viscosus, A. odontolyticus and A. meyeri (1-3). Actinomycosis spreads as a malign tumor to adjacent tissues without regard to the anatomic barriers. Blood-borne spread of actinomycosis is rare, but has been reported to occur more often from thoracic actinomycosis than from other forms. A. israelii is the most frequent cause of disseminated actinomycosis. A. meyeri and “secondary invaders” such as Actinobacillus actinomycetemcomitans are very rare. We present a case history of pulmonary actinomycosis who presented with cutaneous lesions and brain abscesses due to A. meyeri and A. actinomycetemcomitans. CASE REPORT A +-year-old single African man was admitted to the Department of Neurology with weakness of the right arm and leg, and a speech disorder. H e smoked daily a pack of cigarettes for 25 years and consumed daily approximately 10 bottles of beer. The patient was well until 6 years before admission when he was seen at the Department of Pulmonology for symptoms of fever, hemoptysis, loss of weight, and malaise. X-ray films of the chest showed peripheral air space infiltrates and pleural fluid in the right lower lobe. Sputum cultures remained negative for pathogenic bacteria, fungi or mycobacteria. Erythrocyte sedimentation rate (ESR) was 85 mm/h and the white blood cell count (WBC) was 23 X 10y/l.A pleural biopsy was taken during thordcoscopy and chronic inflammatory reaction with suppuration and
Downloaded by [La Trobe University] at 19:39 23 May 2016
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Scand J Infect Dis 24
Fig. 1. Chest X-ray at the second admission showing infiltrates in the upper and lower fields of the right lung with a closed pleural sinus.
fibrosis was diagnosed. Cultures of pleural fluid and pleural biopsy were negative. There was no evidence of an autoimmune disease. The patient refused to undergo bronchoscopy and did not present himself for follow-up. Since then, the patient experienced relapsing purulent skin lesions on arms and legs. which resolved spontaneously or by short courses of antibiotic treatment. The skin lesions healed with scars. One month before admission, the patient awoke with weakness of the right thumb and forefinger. On the following day. he noted weakness of his right arm. During the next days, he complained of a disturbance of gait due to a scuffing right leg. He also experienced speech difficulties in finding words and in articulation. He consulted a neurologist elsewhere who performed an electroencephalographic examination and concluded that there were probably brain metastases of an unidentified tumor. The patient was treated with dexamethasone 1.5 mg q 6 h for 3 weeks. The symptoms subsequently decreased and he was referred to our hospital. On admission, the patient appeared not severely ill with normal pulse, blood pressure and temperature. His speech was slightly dysarthric. Examination of his mouth showed a poor dentition with numerous carious teeth. Multiple scars were present on the skin, especially on the left arm and on his
Fig. 2. High resolution CTsection of the thorax just above the canna, showing a dense infiltrate in the right upper lobe (1). The anterior segment of the left upper lobe shows scar-like strands (2) with peripheral emphysema (3).
Downloaded by [La Trobe University] at 19:39 23 May 2016
S c m d .I lnkct Ilis 24
Dissemitznted nctinonzycosis 669
.?. CT-scan of the brain wing 2 contrast-enhancing .)ns i n the left fronto-parietal
on.
scalp. He had subcutaneous localized swellings on his right forearm (3 cm), his right shoulder (4cm). right pretihial ( 3 cm), and buttock ( 5 cm). These lesions were fluctuant, but neither hot nor tender. On neurological examination there was n o nuchal rigidity and the optic fundi were normal. He had a hemiparesis on the right with hyperactive tendon reflexes. Sensation was normal. Laboratory examination revealed a n elevated ESR of 77 mm/h and an increase of the WBC to 27.5 X lO"/I with a shift to the left. Other blood chemical evaluations were normal. Serology for HIV infection was negative. A specimen o f artcrial hlood showed that the PaO, was 62 mmHg (8.26 Kpa). the PaCO, 43 mmHg (5.73 K p a ) . and the p H 7.46. An X-ray film and a CT-scan of the chest revealed infiltrates in the upper and lower fields of the right lung and in the left upper lobe with pleural fluid and pleural thickening (Figs. 1 and 1 ) . A CT-scan of the brain showed 2 ringlike contrast-enhancing lesions in the left fronto-parietal region and one contrast-enhancing lesion right occipital (Fig. 3). Shortly after admission, the subcutaneous lesions on his right forearm ruptured spontaneously. The Gram stain of the purulent non-smelling fluid from the skin lesions revealed Gram-positive branching filaments and many, small coccobacillary rods that were mainly within the leucocytes. Granules consisting o f Gram-positive filaments were seen. Bronchoscopy was performed with multiple transbronchial biopsies. Stainings and microbiological cultures of the biopsies for fungi, pathogenic bacteria (including actinomyces). and mycobacteria remained negative. Histological examination showed alveolar septa1 fibrosis but no evidence for malignancy. A diagnosis of brain abscesses was considered and a stcrcotactic biopsy was performed. The brain lesions appeared to be abscesses and approximately 5 ml pus from each was obtained. The occipital lesion was not drained. The pus contained Gram-positivc branching rods and fine Gram-negative rods. After the neurosurgical procedure, hemiparesis of the right arm improved immediately. The suhcutaneous abscesses were also drained and the patient was treated with amoxicillin 2 g q 4 h. Two weeks after starting treatment. a CT-scan of the brain showed an increase in size of the occipital lesion. Neurosurgical aspiration was performed, but in the Gram stain of the pus no bacteria were seen. The patient received a 6 week course of amoxicillin intravenously. followed by a 12-month course of
670 E. J . Kuijper et al.
Scand J Infect Dis 24
arnoxicillin by mouth. The neurological symptoms and the skin lesions resolved completely, and his chest X-ray film had cleared. The lesion on the right tibia, however, became superinfected with Pseudomonas aeruginosa. Surgical drainage and antibiotics had a temporal effect only. When the patient developed a septic shock, an amputation below the knee was performed. Thereafter, the
recovery was successful.
Downloaded by [La Trobe University] at 19:39 23 May 2016
BACTERIOLOGICAL FINDINGS Aerobic cultures of the pus from the subcutaneous lesions and the cerebral abscesses revealed, after incubation at 37°C for 48 h, growth of small, coccobacillary Gram-negative rods that were oxidase-negative, catalase-positive, and identified as Actinobacillus actinomycetemcomitans (4). Using the disk diffusion test on Columbia agar medium with 10% sheep blood, the strain was resistant to benzylpenicillin but susceptible to amoxicillin, erythromycin, tetracycline, lincomycin, and ciprofloxacin. After 48 h of anaerobic incubation at 37"C, smooth, white, alfa-hemolytic colonies consisting of catalase-negative branching Gram-positive rods were seen. Biochemical reactions, colonial morphology, and gas-liquid chromatography were consistent with the characteristics of Actinomyces meyeri (4,5). The strain was sensitive to benzylpenicillin, erythromycin, tetracycline, chloramphenicol, and vancomycin. The strain was resistant to metronidazole and ciprofloxacin. Aerobic and anaerobic cultures of the pus from the cerebral abscess obtained 2 weeks after starting antibiotics, remained negative. When the pus was assayed by disk diffusion on a plate streaked with arnoxicillin sensitive streptococci, a clear zone of inhibition was seen representing approximately 1 p.g/ml. Three blood cultures taken before antibiotics were given remained negative after prolonged incubation for 4 weeks. DISCUSSION The patient described here, presented with skin lesions and cerebral abscesses due to Actinomyces meyeri and Actinobacillus actinomycetemcomitans. He had slowly progressive pulmonary lesions which were present already 6 years before admission. Thoracoscopical examination at that time and bronchoscopy during the present admission did not reveal a definite diagnosis of the pulmonary abnormalities. However, it is very likely that the patient had pulmonary actinomycosis as a complication of his pre-existing emphysema, neglected dental care, and alcohol abuse. Hematogenous spread from the pulmonary lesions lead to disseminated actinomycosis with brain abscesses which were initially misdiagnosed as metastasizing tumor. This long and unusual presentation of actinomycosis was probably also influenced by several short courses of incomplete antibiotic treatment. Actinomycosis of the central nervous system (CNS) may develop via hematogenous spread from pulmonary lesions (6,7). Several types of CNS lesions have been reported, such as brain abscesses, subdural empyema, actinomycoma, spinal and epidural abscesses. Brain abscesses account for approximately 67% of all CNS lesions and they most frequently develop in the frontal and temporal lobes (6). Abscesses may be multiloculated or uniloculated, encapsulated or without a capsule. In the culture-proven cases, 34% of 50 CNS actinomycotic abscesses contained a mixed microbial flora (6). Generally, bacteria such as streptococci, anaerobic Gram-negative rods and Streptobacillus moniliformis have been found, whereas A. actinomyceterncomitans has been isolated only once (1, 6-8),A. actinomycetemcomitans belongs to the normal oral flora, but is particularly involved in the development of juvenile periodontitis (9). It is possible that A. actinomyceterncomitans acts synergistically with A. meyeri in the same way as known for other Actinomyces species. The natural habitat of A. meyeri is probably the periodontal sulcus. A. meyeri has been shown to cause infections of the bones, the neck, as well as abscesses of the brain, the
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Scant1 J Infect Dis 24
Disseminated actinomycosis
dentoalveolar region, and the breast (8, 10-14). Pulmonary actinomycosis caused by A . meyeri is considered to be rare. This is probably due to the fact that A. meyeri is difficult to recognize. A . meyeri, named after Kurt Meyer, who described this bacterium in 1911, can be differentiated from other Actinomyces species by cellular and colonial morphology. and by biochemical reactions (4. 5, 15). Microcolonies are smooth and grow fast when compared to A . israelii which is the most common cause of human actinomycosis. We found 6 published reports of 7 cases with pulmonary infection ( 1 6 2 1 ) . Clinical and radiographic findings were similar to those caused by A . israelii. Two of the 7 cases with pulmonary A . meyeri infections were incidental findings on the X-ray of the chest. Of the 7 infections. 4 showed hematogenous dissemination (17, 19-21). This is in contrast to actinomycosis due to other Actinomyces species, where dissemination is not very common and where secondary involvement of the skin has been reported only occasionally (3, 22-25). Interestingly, 3/8 patients (including our patient) with an infection due to A . meyeri had subcutaneous lesions. Thc skin lesions in our patient were rapidly progressive and new lesions developed during treatment with corticosteroids. The blood cultures, however, remained negative. The optimal treatment of actinomycotic brain abscesses consists of surgical drainage combined with antibiotic therapy. This management was successful in 72% of all patients reported in a review article (6). However, it is possible that patients with small cerebral abscesses respond to antibiotics alone (26). Surprisingly, the large encapsulated occipital abscess in our patient was sterile after 14 days of antibiotic treatment. This observation suggests that even large abscesses due to A . rneyeri and A. actinomycetemcomitans respond to antibiotics alone. We choose amoxicillin because of the presence of A . actinomycetemcomitans. which was found to be benzylpenicillin resistant as usual. The patient was treated with intravenous amoxicillin for 6 weeks, followed by oral amoxicillin over a period of 12 months, resulting in clinical cure. REFERENCES I . Lerner PI. Actinomyces and Arachnia species. In: Mandell GL, Douglas RG, Bcnnet JE. eds. Principles and practice of infectious diseases. New York: John Wiley and Sons. 1932-1942, 1990. 2. Weese WC, Smith 1M. A study of 57 cases of actinomycosis over a 36-year period. A diagnostic failure with good prognosis after treatment. Arch Intern Med 135: 1562-1568, 1975. 3. Brown JR. Human actinomycosis. A study of 181 subjects. Hum Pathol 4: 31S330, 1973. 4. Pickett M J , Hollis GH. Bottone EJ. Miscellaneous Gram-negative rods. In: Balows A, Hausler WJ Jr. Herrmann KL, lsenberg HD. Shadomy HJ, eds. Manual of clinical microbiology. Washington. DC: American Society for Microbiology, 410-429, 1991. 5, Schaal KP. Genus Actinomyces. In: Sneath PHA, Mair NS, Sharpe ME, Holt JG. Bergey’s manual of systematic bacteriology, vol. 2. Williams and Wilkens. 1383-1418. 1986. 6. Srnego RA. Actinomycosis of the central nervous system. Rev Infect Dis 9: 855-865, 1987. 7. Bolton CF. Ashenhurst EM. Actinomycosis of the brain: Case report and review of the literature Can Med J 90: 922-928, 1964. 8. Dijkmans BAC, Thomeer RTWM, Vielvoye GJ, Lampe AS, Mattie H. Brain abscesses due t o Streptobacillus moniliformis and Actinomyces meyeri. Infection 12: 262-264, 1984. 9. Slots JL. Subgingival microflora and periodontal disease. J Clin Periodontol 6: 351-382, 1979. 10. Bennhof DF. Actinomycosis: diagnostic and therapeutic considerations and a review of 32 cases. Laryngoscope 94: 1198-1217, 1984. 1 1, Lipton M. Sonnenfeld G. Actinomyces meyeri osteomyelitis: an unusual case of chronic infection of the tibia. Clin Orthop 148: 169-170, 1980. 12. Pang DK. Abdalla M. Osteomyelitis of the foot due to Actinomyces meyeri: a case report. Foot Ankle 8: 169-171. 1987. 13. Lewis MAO. MacFarlane TW, McGowan DA. Quantitative bacteriology of acute dentoalveolar abscesses. J Med Microbiol 21: 101-104, 1986. 14. Allen JN. Actinomyces meyeri breast abscess. Am J Med 83: 186-187, 1987.
hI I
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15. Cat0 EP, Moore WEC, Nygaard G, Holdeman L. Actinomyces meyeri sp. nov. Specific epithet rev. Int J Syst Bacteriol 34: 487-489, 1984. 16. Rippon JW, Kathuria SK. Actinomycosis meyeri presenting as an asymptomatic lung mass. Mycopathologia 84: 187-192, 1984. 17. Rose HD, Vaekey B, Kutty CP. Thoracic actinomycosis caused by Actinomyces meyeri. Am Rev Respir Dis 125: 251-254, 1982. 18. Allworth AM, Ghosh HK, Saltos N. A case of Actinomyces meyeri pneumonia in a child. Med J Aust 145: 33, 1986. 19. Marty HU, Wust J. Disseminated actinomycosis caused by Actinomyces meyeri. Infection 17: 154-155, 1989. 20. Ferrier MC, Janin-Mercier A, Meyer A, Beytout J, Cambon M, Sirot J, Souteyrand P. Actinomycose a Actinomyces meyeri: un cas avec localisation thoracique et tibiale. Ann Med Interne (Paris) 137: 649-651, 1986. 21. Lentino JR, Allen JE, Stachowski M. Hematogenous dissemination of thoracic actinomycosis due to Actinomyces meyeri. Pediatr Infect Dis 4: 698-689, 1985. 22. Webb AK, Howell R, Hickman JA. Thoracic actinomycosis presenting with peripheral skin lesions. Thorax 33: 818-819, 1978. 23. Varkey B, Landis FB, Tang T T , Rose HD. Dissemination to the skin, subcutaneous tissue and muscle. Arch Intern Med 134: 689-693, 1974. 24. Smith DL, Lockwood WR. Disseminated actinomycosis. Chest 67: 242-244, 1975. 25. Butas CA, Read SE, Coleman RE, Abramovitch H. Disseminated actinomycosis. Can Med Assoc J 103: 1069-1071, 1970. 26. Rosenblum ML, Hoff JT, Norman D, Edwards MS, Berg BO. Nonoperative treatment of brain abscesses in selected high-risk patients. J Neurosurg 52: 217-225, 1981.
ISSN: 0036-5548 (Print) 1651-1980 (Online) Journal homepage: http://www.tandfonline.com/loi/infd19
Disseminated Actinomycosis due to Actinomyces meyeri and Actinobacillus actinomycetemcomitans Ed J. Kuijper, Henri O. Wiggerts, Gwan J. Jonker, Klaus P. Schaal & Jan De Gans To cite this article: Ed J. Kuijper, Henri O. Wiggerts, Gwan J. Jonker, Klaus P. Schaal & Jan De Gans (1992) Disseminated Actinomycosis due to Actinomyces meyeri and Actinobacillus actinomycetemcomitans, Scandinavian Journal of Infectious Diseases, 24:5, 667-672, DOI: 10.3109/00365549209054655 To link to this article: http://dx.doi.org/10.3109/00365549209054655
Published online: 08 Jul 2009.
Submit your article to this journal
Article views: 14
View related articles
Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=infd19 Download by: [La Trobe University]
Date: 23 May 2016, At: 19:39
Scand J Infect Dis 24: 667-672. 1992
CASE REPORT
Disseminated Actinomycosis due t o Actinomyces meyeri and Acti nobacillus acti nomycetemcom itans ED J . KUIJPER’, HENRI 0. WIGGERTS2, GWAN J. JONKER3, KLAUS P. SCHAAL‘ and JAN DE GANS’
Downloaded by [La Trobe University] at 19:39 23 May 2016
From the Departments of ‘Bacteriology. ’Neurology, and ’Pulmonology, Academic Medrt ill Centre, Amsterdam, The Netherlands, and ‘The Institute for Medical Microbiology and Immunology, University of Bonn, Bonn, Germany
A 44-year-old man presented with pulmonary lesions and neurological symptoms suggestive of lung carcinoma with cerebral metastases. He had non-specific chest X-ray findings since 6 years and he also suffered from relapsing purulent skin lesions which resolved spontaneously or by short courses of antibiotic treatment. When corticosteroids were given, multiple subcutaneous swellings developed that spontaneously ruptured. The pus contained Actinomyces meyeri and Actinobacillus actinomycetemcomitans. On operation, the intracerehral lesions appeared to be abscesses and the same bacteria were cultured as from the skin lesions. Bronchoscopical examination did not reveal a diagnosis. Amoxicillin was given for 12 months and the patient recovered.
E . J . Kuijper, MD, Department of Bacteriology, L1,Academic Medical Centre, Meibergdreef 9 , 1105 AZ, Amsterdam, The Netherlands
INTRODUCTION Actinomycosis is characterized as an opportunistic, chronic, suppurative, and granulomatous infection with abscess formation and draining sinus tracts (1-3). The primary sites of infection are the cervicofacial region, the thorax, and the abdomen and pelvic region. Human actinomycosis is caused mainly by Actinomyces israelii and A. gerencseriae followed in order of importance by Propionibacterium propionica, A. naeslundii, A. viscosus, A. odontolyticus and A. meyeri (1-3). Actinomycosis spreads as a malign tumor to adjacent tissues without regard to the anatomic barriers. Blood-borne spread of actinomycosis is rare, but has been reported to occur more often from thoracic actinomycosis than from other forms. A. israelii is the most frequent cause of disseminated actinomycosis. A. meyeri and “secondary invaders” such as Actinobacillus actinomycetemcomitans are very rare. We present a case history of pulmonary actinomycosis who presented with cutaneous lesions and brain abscesses due to A. meyeri and A. actinomycetemcomitans. CASE REPORT A +-year-old single African man was admitted to the Department of Neurology with weakness of the right arm and leg, and a speech disorder. H e smoked daily a pack of cigarettes for 25 years and consumed daily approximately 10 bottles of beer. The patient was well until 6 years before admission when he was seen at the Department of Pulmonology for symptoms of fever, hemoptysis, loss of weight, and malaise. X-ray films of the chest showed peripheral air space infiltrates and pleural fluid in the right lower lobe. Sputum cultures remained negative for pathogenic bacteria, fungi or mycobacteria. Erythrocyte sedimentation rate (ESR) was 85 mm/h and the white blood cell count (WBC) was 23 X 10y/l.A pleural biopsy was taken during thordcoscopy and chronic inflammatory reaction with suppuration and
Downloaded by [La Trobe University] at 19:39 23 May 2016
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Scand J Infect Dis 24
Fig. 1. Chest X-ray at the second admission showing infiltrates in the upper and lower fields of the right lung with a closed pleural sinus.
fibrosis was diagnosed. Cultures of pleural fluid and pleural biopsy were negative. There was no evidence of an autoimmune disease. The patient refused to undergo bronchoscopy and did not present himself for follow-up. Since then, the patient experienced relapsing purulent skin lesions on arms and legs. which resolved spontaneously or by short courses of antibiotic treatment. The skin lesions healed with scars. One month before admission, the patient awoke with weakness of the right thumb and forefinger. On the following day. he noted weakness of his right arm. During the next days, he complained of a disturbance of gait due to a scuffing right leg. He also experienced speech difficulties in finding words and in articulation. He consulted a neurologist elsewhere who performed an electroencephalographic examination and concluded that there were probably brain metastases of an unidentified tumor. The patient was treated with dexamethasone 1.5 mg q 6 h for 3 weeks. The symptoms subsequently decreased and he was referred to our hospital. On admission, the patient appeared not severely ill with normal pulse, blood pressure and temperature. His speech was slightly dysarthric. Examination of his mouth showed a poor dentition with numerous carious teeth. Multiple scars were present on the skin, especially on the left arm and on his
Fig. 2. High resolution CTsection of the thorax just above the canna, showing a dense infiltrate in the right upper lobe (1). The anterior segment of the left upper lobe shows scar-like strands (2) with peripheral emphysema (3).
Downloaded by [La Trobe University] at 19:39 23 May 2016
S c m d .I lnkct Ilis 24
Dissemitznted nctinonzycosis 669
.?. CT-scan of the brain wing 2 contrast-enhancing .)ns i n the left fronto-parietal
on.
scalp. He had subcutaneous localized swellings on his right forearm (3 cm), his right shoulder (4cm). right pretihial ( 3 cm), and buttock ( 5 cm). These lesions were fluctuant, but neither hot nor tender. On neurological examination there was n o nuchal rigidity and the optic fundi were normal. He had a hemiparesis on the right with hyperactive tendon reflexes. Sensation was normal. Laboratory examination revealed a n elevated ESR of 77 mm/h and an increase of the WBC to 27.5 X lO"/I with a shift to the left. Other blood chemical evaluations were normal. Serology for HIV infection was negative. A specimen o f artcrial hlood showed that the PaO, was 62 mmHg (8.26 Kpa). the PaCO, 43 mmHg (5.73 K p a ) . and the p H 7.46. An X-ray film and a CT-scan of the chest revealed infiltrates in the upper and lower fields of the right lung and in the left upper lobe with pleural fluid and pleural thickening (Figs. 1 and 1 ) . A CT-scan of the brain showed 2 ringlike contrast-enhancing lesions in the left fronto-parietal region and one contrast-enhancing lesion right occipital (Fig. 3). Shortly after admission, the subcutaneous lesions on his right forearm ruptured spontaneously. The Gram stain of the purulent non-smelling fluid from the skin lesions revealed Gram-positive branching filaments and many, small coccobacillary rods that were mainly within the leucocytes. Granules consisting o f Gram-positive filaments were seen. Bronchoscopy was performed with multiple transbronchial biopsies. Stainings and microbiological cultures of the biopsies for fungi, pathogenic bacteria (including actinomyces). and mycobacteria remained negative. Histological examination showed alveolar septa1 fibrosis but no evidence for malignancy. A diagnosis of brain abscesses was considered and a stcrcotactic biopsy was performed. The brain lesions appeared to be abscesses and approximately 5 ml pus from each was obtained. The occipital lesion was not drained. The pus contained Gram-positivc branching rods and fine Gram-negative rods. After the neurosurgical procedure, hemiparesis of the right arm improved immediately. The suhcutaneous abscesses were also drained and the patient was treated with amoxicillin 2 g q 4 h. Two weeks after starting treatment. a CT-scan of the brain showed an increase in size of the occipital lesion. Neurosurgical aspiration was performed, but in the Gram stain of the pus no bacteria were seen. The patient received a 6 week course of amoxicillin intravenously. followed by a 12-month course of
670 E. J . Kuijper et al.
Scand J Infect Dis 24
arnoxicillin by mouth. The neurological symptoms and the skin lesions resolved completely, and his chest X-ray film had cleared. The lesion on the right tibia, however, became superinfected with Pseudomonas aeruginosa. Surgical drainage and antibiotics had a temporal effect only. When the patient developed a septic shock, an amputation below the knee was performed. Thereafter, the
recovery was successful.
Downloaded by [La Trobe University] at 19:39 23 May 2016
BACTERIOLOGICAL FINDINGS Aerobic cultures of the pus from the subcutaneous lesions and the cerebral abscesses revealed, after incubation at 37°C for 48 h, growth of small, coccobacillary Gram-negative rods that were oxidase-negative, catalase-positive, and identified as Actinobacillus actinomycetemcomitans (4). Using the disk diffusion test on Columbia agar medium with 10% sheep blood, the strain was resistant to benzylpenicillin but susceptible to amoxicillin, erythromycin, tetracycline, lincomycin, and ciprofloxacin. After 48 h of anaerobic incubation at 37"C, smooth, white, alfa-hemolytic colonies consisting of catalase-negative branching Gram-positive rods were seen. Biochemical reactions, colonial morphology, and gas-liquid chromatography were consistent with the characteristics of Actinomyces meyeri (4,5). The strain was sensitive to benzylpenicillin, erythromycin, tetracycline, chloramphenicol, and vancomycin. The strain was resistant to metronidazole and ciprofloxacin. Aerobic and anaerobic cultures of the pus from the cerebral abscess obtained 2 weeks after starting antibiotics, remained negative. When the pus was assayed by disk diffusion on a plate streaked with arnoxicillin sensitive streptococci, a clear zone of inhibition was seen representing approximately 1 p.g/ml. Three blood cultures taken before antibiotics were given remained negative after prolonged incubation for 4 weeks. DISCUSSION The patient described here, presented with skin lesions and cerebral abscesses due to Actinomyces meyeri and Actinobacillus actinomycetemcomitans. He had slowly progressive pulmonary lesions which were present already 6 years before admission. Thoracoscopical examination at that time and bronchoscopy during the present admission did not reveal a definite diagnosis of the pulmonary abnormalities. However, it is very likely that the patient had pulmonary actinomycosis as a complication of his pre-existing emphysema, neglected dental care, and alcohol abuse. Hematogenous spread from the pulmonary lesions lead to disseminated actinomycosis with brain abscesses which were initially misdiagnosed as metastasizing tumor. This long and unusual presentation of actinomycosis was probably also influenced by several short courses of incomplete antibiotic treatment. Actinomycosis of the central nervous system (CNS) may develop via hematogenous spread from pulmonary lesions (6,7). Several types of CNS lesions have been reported, such as brain abscesses, subdural empyema, actinomycoma, spinal and epidural abscesses. Brain abscesses account for approximately 67% of all CNS lesions and they most frequently develop in the frontal and temporal lobes (6). Abscesses may be multiloculated or uniloculated, encapsulated or without a capsule. In the culture-proven cases, 34% of 50 CNS actinomycotic abscesses contained a mixed microbial flora (6). Generally, bacteria such as streptococci, anaerobic Gram-negative rods and Streptobacillus moniliformis have been found, whereas A. actinomyceterncomitans has been isolated only once (1, 6-8),A. actinomycetemcomitans belongs to the normal oral flora, but is particularly involved in the development of juvenile periodontitis (9). It is possible that A. actinomyceterncomitans acts synergistically with A. meyeri in the same way as known for other Actinomyces species. The natural habitat of A. meyeri is probably the periodontal sulcus. A. meyeri has been shown to cause infections of the bones, the neck, as well as abscesses of the brain, the
Downloaded by [La Trobe University] at 19:39 23 May 2016
Scant1 J Infect Dis 24
Disseminated actinomycosis
dentoalveolar region, and the breast (8, 10-14). Pulmonary actinomycosis caused by A . meyeri is considered to be rare. This is probably due to the fact that A. meyeri is difficult to recognize. A . meyeri, named after Kurt Meyer, who described this bacterium in 1911, can be differentiated from other Actinomyces species by cellular and colonial morphology. and by biochemical reactions (4. 5, 15). Microcolonies are smooth and grow fast when compared to A . israelii which is the most common cause of human actinomycosis. We found 6 published reports of 7 cases with pulmonary infection ( 1 6 2 1 ) . Clinical and radiographic findings were similar to those caused by A . israelii. Two of the 7 cases with pulmonary A . meyeri infections were incidental findings on the X-ray of the chest. Of the 7 infections. 4 showed hematogenous dissemination (17, 19-21). This is in contrast to actinomycosis due to other Actinomyces species, where dissemination is not very common and where secondary involvement of the skin has been reported only occasionally (3, 22-25). Interestingly, 3/8 patients (including our patient) with an infection due to A . meyeri had subcutaneous lesions. Thc skin lesions in our patient were rapidly progressive and new lesions developed during treatment with corticosteroids. The blood cultures, however, remained negative. The optimal treatment of actinomycotic brain abscesses consists of surgical drainage combined with antibiotic therapy. This management was successful in 72% of all patients reported in a review article (6). However, it is possible that patients with small cerebral abscesses respond to antibiotics alone (26). Surprisingly, the large encapsulated occipital abscess in our patient was sterile after 14 days of antibiotic treatment. This observation suggests that even large abscesses due to A . rneyeri and A. actinomycetemcomitans respond to antibiotics alone. We choose amoxicillin because of the presence of A . actinomycetemcomitans. which was found to be benzylpenicillin resistant as usual. The patient was treated with intravenous amoxicillin for 6 weeks, followed by oral amoxicillin over a period of 12 months, resulting in clinical cure. REFERENCES I . Lerner PI. Actinomyces and Arachnia species. In: Mandell GL, Douglas RG, Bcnnet JE. eds. Principles and practice of infectious diseases. New York: John Wiley and Sons. 1932-1942, 1990. 2. Weese WC, Smith 1M. A study of 57 cases of actinomycosis over a 36-year period. A diagnostic failure with good prognosis after treatment. Arch Intern Med 135: 1562-1568, 1975. 3. Brown JR. Human actinomycosis. A study of 181 subjects. Hum Pathol 4: 31S330, 1973. 4. Pickett M J , Hollis GH. Bottone EJ. Miscellaneous Gram-negative rods. In: Balows A, Hausler WJ Jr. Herrmann KL, lsenberg HD. Shadomy HJ, eds. Manual of clinical microbiology. Washington. DC: American Society for Microbiology, 410-429, 1991. 5, Schaal KP. Genus Actinomyces. In: Sneath PHA, Mair NS, Sharpe ME, Holt JG. Bergey’s manual of systematic bacteriology, vol. 2. Williams and Wilkens. 1383-1418. 1986. 6. Srnego RA. Actinomycosis of the central nervous system. Rev Infect Dis 9: 855-865, 1987. 7. Bolton CF. Ashenhurst EM. Actinomycosis of the brain: Case report and review of the literature Can Med J 90: 922-928, 1964. 8. Dijkmans BAC, Thomeer RTWM, Vielvoye GJ, Lampe AS, Mattie H. Brain abscesses due t o Streptobacillus moniliformis and Actinomyces meyeri. Infection 12: 262-264, 1984. 9. Slots JL. Subgingival microflora and periodontal disease. J Clin Periodontol 6: 351-382, 1979. 10. Bennhof DF. Actinomycosis: diagnostic and therapeutic considerations and a review of 32 cases. Laryngoscope 94: 1198-1217, 1984. 1 1, Lipton M. Sonnenfeld G. Actinomyces meyeri osteomyelitis: an unusual case of chronic infection of the tibia. Clin Orthop 148: 169-170, 1980. 12. Pang DK. Abdalla M. Osteomyelitis of the foot due to Actinomyces meyeri: a case report. Foot Ankle 8: 169-171. 1987. 13. Lewis MAO. MacFarlane TW, McGowan DA. Quantitative bacteriology of acute dentoalveolar abscesses. J Med Microbiol 21: 101-104, 1986. 14. Allen JN. Actinomyces meyeri breast abscess. Am J Med 83: 186-187, 1987.
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15. Cat0 EP, Moore WEC, Nygaard G, Holdeman L. Actinomyces meyeri sp. nov. Specific epithet rev. Int J Syst Bacteriol 34: 487-489, 1984. 16. Rippon JW, Kathuria SK. Actinomycosis meyeri presenting as an asymptomatic lung mass. Mycopathologia 84: 187-192, 1984. 17. Rose HD, Vaekey B, Kutty CP. Thoracic actinomycosis caused by Actinomyces meyeri. Am Rev Respir Dis 125: 251-254, 1982. 18. Allworth AM, Ghosh HK, Saltos N. A case of Actinomyces meyeri pneumonia in a child. Med J Aust 145: 33, 1986. 19. Marty HU, Wust J. Disseminated actinomycosis caused by Actinomyces meyeri. Infection 17: 154-155, 1989. 20. Ferrier MC, Janin-Mercier A, Meyer A, Beytout J, Cambon M, Sirot J, Souteyrand P. Actinomycose a Actinomyces meyeri: un cas avec localisation thoracique et tibiale. Ann Med Interne (Paris) 137: 649-651, 1986. 21. Lentino JR, Allen JE, Stachowski M. Hematogenous dissemination of thoracic actinomycosis due to Actinomyces meyeri. Pediatr Infect Dis 4: 698-689, 1985. 22. Webb AK, Howell R, Hickman JA. Thoracic actinomycosis presenting with peripheral skin lesions. Thorax 33: 818-819, 1978. 23. Varkey B, Landis FB, Tang T T , Rose HD. Dissemination to the skin, subcutaneous tissue and muscle. Arch Intern Med 134: 689-693, 1974. 24. Smith DL, Lockwood WR. Disseminated actinomycosis. Chest 67: 242-244, 1975. 25. Butas CA, Read SE, Coleman RE, Abramovitch H. Disseminated actinomycosis. Can Med Assoc J 103: 1069-1071, 1970. 26. Rosenblum ML, Hoff JT, Norman D, Edwards MS, Berg BO. Nonoperative treatment of brain abscesses in selected high-risk patients. J Neurosurg 52: 217-225, 1981.
