Indian Journal of Dermatology
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CASE REPORTS
Year : 2005  |  Volume : 50  |  Issue : 3  |  Page : 152-154
Gluteal primary cutaneus actinomycosis


Department of Dermatology and STD, Military Hospital, Jabalpur, Madhya Pradesh - 482 001, India

Correspondence Address:
Manas Chatterjee
Department of Dermatology and STD, Military Hospital, Jabalpur, Madhya Pradesh - 482 001
India
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   Abstract 

A 58 year old male presented with multiple areas of painless purulent discharge from the buttocks. He had pallor and dermatological examination revealed multiple subcutaneus nodules and discharging sinuses over the buttocks with hyperpigmentation and indurated puckered scarring. Examination of the pus revealed 'sulphur granules' and Gram stain showed narrow bacillary forms and elongated hyphae with occasional branching. Skin biopsy revealed granular colonies in which 'ray fungus' were seen. In brain-heart glucose agar in anerobic environment, white spidery colonies of Actinomyces israelii were grown at 37o C in 4 days, confirming primary cutaneus actinomycosis. He was managed with capsule doxycycline with regression of pus discharge in 6 weeks and therapy is planned for 18 months.


Keywords: Buttocks, Actinomyces israelii, Culture


How to cite this article:
Chatterjee M. Gluteal primary cutaneus actinomycosis. Indian J Dermatol 2005;50:152-4

How to cite this URL:
Chatterjee M. Gluteal primary cutaneus actinomycosis. Indian J Dermatol [serial online] 2005 [cited 2020 Apr 10];50:152-4. Available from: http://www.e-ijd.org/text.asp?2005/50/3/152/18930



   Introduction Top


Actinomycosis is a chronic spreading suppurative granulomatous bacterial infection caused by anaerobic actinomyces, primarily Actinomyces israelii , with sinuses draining and discharging 'sulphur' granules. Access to antibiotics is an important determining factor in the occurrence of this infection and trauma predisposes to its development. It occurs worldwide and males are more predisposed to it than females. It can affect all organs and tissues and five main clinical types are recognized, namely, cervicofacial (60%), thoracic (20%), abdominal (15%), pelvic and primary cutaneus.[1]

Actinomycosis of the skin usually occurs by extension from underlying primary foci in the neck, face, abdomen or lung. When such sources cannot be found, the skin is presumed to be the primary site.[2]-[4] Primary cutaneus actinomycosis is a rarity and an uncommon disease. It may be diagnosed only when the presence of a deeper form of the infection has been excluded. Few such cases have been reported recently in the

International[5],[6] as well as Indian literature including one over the scalp [7] and another similar to ours over the gluteal region.[8] However, culture confirmation of the diagnosis is a rarity and not achieved in most cases.[9]

A case of this rare type of actinomycosis, confirmed on culture, is reported.


   Case report Top


A 58 year old male presented with insidious onset of gradually progressive multiple areas of painless pus discharge from both buttocks, left more than right, of one month duration. This was associated with thickening and darkening of the skin around these areas. There was no history of trauma, surgery, fever, oro-dental illness, pain abdomen/bowel complaints, blood mixed stools, urinary complaints, discharge of colored granules, sexual promiscuity, weight loss, cough, pain chest, jaundice, swollen glands or bone pains. There was no history of diabetes or tuberculosis. He gave past history of similar illness two years back, treated with antitubercular therapy for a period of two months with partial relief, after which he stopped medication on his own, with recurrence. He was resident of an urban slum area and gave history of defecation in the fields.

General examination revealed pallor and dermatological examination showed multiple subcutaneus nodules and discharging sinuses showing pouting unhealthy granulation tissue with profuse thin purulent discharge over the buttocks with hyperpigmentation and indurated puckered scarring [Figure - 1]. Perianal area was free and there was no inguinal lymphadenopathy. Per rectal examination and proctoscopy were normal.

Investigations revealed anemia and raised ESR. All other relevant investigations including routine hemogram, urinalysis, blood sugar, renal and liver function tests, blood VDRL, HIV test, Mantoux test, radiological evaluation of the chest, lumbosacral spine and hip and ultrasound examination of the abdomen and pelvis were within normal limits. Examination of the pus after diluting in normal saline revealed small yellow, 'sulphur granules' and gram stain of crushed granules showed narrow gram positive non acid-fast bacillary forms with elongated hyphae and occasional branching. Skin biopsy revealed basophilic granular colonies from which radiate delicate mycelial filaments. Some with club shaped PAS positive processes ('ray fungus') were also seen. Anerobic culture of the granules in brain-heart infusion revealed frothing with growth of 'fluffy ball' colonies in 48 hours. In solid medium, white spidery colonies of microaerophilic non-sporing, slender bacilli of Actinomyces israelii , with irregular margins, were grown at 37o centigrade in 4 days. Proteus was also grown as co-pathogens. In the absence of any other focus, primary cutaneus actinomycsis was thereby confirmed.

He was managed with capsule doxycycline 100 mg twice a day along with local toilet and supportive hematinic therapy and injection gentamicin 60 mg 8 hourly, the latter for 10 days. There was cessation of pus discharge and reduction of induration of lesions after 6 weeks of oral doxycycline. Doxycycline therapy is planned for 18 months and the patient is on medication on OPD basis.


   Discussion Top


Important conditions which may lead to multiple discharging sinuses in the Indian scenario include actinomycotic mycetoma, hidradenitis suppurativa, tuberculosis, botryomycosis, actinomycosis, syphilitic gummata, osteomyelitis, deep fungal infection such as cryptococcosis, in addition to surgical causes such as inflammatory bowel disease, fistulae in ano, etc.

Approach to a case of multiple discharging sinuses without any evidence of systemic or gastrointestinal disease should consist of naked eye examination for the presence and type of granules including examination after dilution, in addition to routine history taking and clinical examination. This should be followed by microscopy and culture characterisation of pus/granules for bacteria, mycobacteria and fungi in an aerobic and anerobic environment along with histology. Newer diagnostic modalities such as fluorescent antibody technique, gel diffusion and immunofluorescence may be helpful if available.

The group of conditions loosely clubbed under subcutaneus mycosis include conditions caused by true fungi (eumycetoma), actinomycetes (actinomycotic mycetoma and actinomycosis) and bacteria (botryomycosis). Differentiation is made on the basis of epidemiology, in addition to granule morphology, microbiological and histopathological characteristics. Actinomycosis is notoriously misdiagnosed, even by the best of clinicians.[9]

The origin of primary cutaneus actinomycosis has usually been traced to farm work[10], human bites/punches [4] in which the infection occurs over the hand of the assailant; or oral contact with the breast,[11], [12] as the organism is a normal inhabitant of human mouth. A few cases have been reported, in which other injuries have been incriminated, such as a horse rider with involvement of the subgluteal bursae.[13] Very rare cases show no discernible history of injury, occupational contact or exogenous foci and in these cases, minor trauma such as brushing of human feces laden grass or thorn may be assumed to cause implantation of the organism as may have occurred in our case.

A synergism between actinomyces and other organisms may exist and is said to be partially responsible for its pathogenesis.[1] Though the organism is anerobic, sometimes, a period of aerobic growth after initial anerobic culture has been found to be helpful in the growth of the organism.

Quick response is not expected due to fibrosis. Prior to the advent of penicillin, potassium iodide and vaccines were used for therapy.[2] At present, crystalline penicillin is the drug of choice given 10-12 megaunits IV 12 hourly daily for 30-45 days followed by 2-5 megaunits IM/5 megaunits orally for 12-18 months. Due to problems of long term parenteral therapy as also erratic oral bioavailability of penicillin V, doxycycline was preferred in our case. Elsewhere, minocycline has been successfully used in immunocompromised patients with this condition.[14], [15] Other drugs such as ampicillin/amoxicillin, erythromycin, sulphonamides, streptomycin, chloramphenicol, other tetracyclines and rifampicin have been used.[13] Surgery may be useful for single small or treated lesions and is planned in our case after lesions become quiescent.

The case is interesting as this is an extremely rare case, the organism was successfully demonstrated and cultured which permitted a confirmation of the diagnosis, which is often not possible in the best of centres.[15]



 
   References Top

1.Hay RJ, Adriaans BM. Bacterial Infections. In: Champion RH, Burton JL, Breathnach SM, eds. Textbook of Dermatology. 6th ed. London: Blackwell Science, 1998; 1171-3.  Back to cited text no. 1    
2.Cope VZ. Actinomycosis. London: Oxford University Press, 1938.  Back to cited text no. 2    
3.Montgomery RM, Welton WA. Primary actinomycosis of the upper extremity. Arch Dermatol 1959; 79: 578-9.   Back to cited text no. 3    
4.Burrows HJ. Actinomycosis from punch injuries with a report of a case affecting a metacarpal bone. Br J Surg 1944-45; 32: 506.   Back to cited text no. 4    
5.Gayraud A, Grosieux-Dauger C, Durlach A, Salmon-Her V, Elia A, Grosshans E, et al . Cutaneous actinomycosis in the perianal area and buttocks. Ann Dermatol Venereol 2000; 127: 393-6.   Back to cited text no. 5    
6.Sardana K, Mendiratta V, Sharma RC. A suspected case of primary cutaneous actinomycosis on buttock. J Dermatol 2001; 28: 276-8.  Back to cited text no. 6    
7.Karthikeyan K, Thappa DM. Primary Cutaneous Actinomycosis of the scalp. Indian J Dermatol 2002; 47: 63-4.  Back to cited text no. 7    
8.Das D, Majumdar S, Mandal SK, Ghoshal AG. Primary actinomycosis of the gluteal region. Indian J Dermatol 2003; 48: 49-51.  Back to cited text no. 8    
9.Russo TA. Agents of Actinomycosis. In: Mandell GL, Bennett JE, Dolin R, eds. Principles and practice of infectious diseases. 5th ed. Philadelphia: Churchill Livingstone, 2000: 2645.   Back to cited text no. 9    
10.Klaber R. Primary cutaneous actinomycosis. Br J Dermatol 1934; 46: 12.   Back to cited text no. 10    
11.Davies JA. Primary actinomycosis of the breast. Br J Surg 1961; 38: 378.  Back to cited text no. 11    
12.Robinson RA. Actinomycosis of the subcutaneous tissue of the forearm secondary to a human bite. JAMA 1944; 125: 1049.   Back to cited text no. 12    
13.Hingley GB. Actinomycosis, subgluteal bursae. Memphis Med J 1950;25: 112.   Back to cited text no. 13    
14.Takeda H, Mitsuhashi Y, Kondo S. Cutaneous disseminated actinomycosis in a patient with acute lymphocytic leukaemia. J Dermatol 1998; 25(1): 37-40.  Back to cited text no. 14    
15.Okano M. Primary cutaneous actinomycosis of the extremities: a report from Japan. Cutis 1989; 44(3): 231-3.  Back to cited text no. 15    


Figures

[Figure - 1]



 

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