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Localised Cryptococcal Scalp Osteomyelitis in an Immunocompetent Man- A Rare Case |
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Sreenath K, Sanjeeb Sharma, Th Gojendra Singh, Kh Gojen Singh, Khuraijam Ranjana Devi 1. Junior Resident, Department of Surgery, Regional Institute of Medical Sciences, Imphal, India. 2. Junior Resident, Department of Microbiology, Regional Institute of Medical Sciences, Imphal, India. 3. Assistant Professor, Department of Neurosurgery, Regional Institute of Medical Sciences, Imphal, India. 4. Assistant Professor, Department of Surgery, Regional Institute of Medical Sciences, Imphal, India. 5. Associate Professor Department of Microbiology, Regional Institute of Medical Sciences, Imphal, India. |
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Correspondence Address : Dr. Th Gojendra Singh, Assistant Professor Neurosurgery Unit, Department of Surgery, RIMS Imphal, Manipur- 795004, India. Phone : 9615839541, E-mail: gojendraneuro@gmail.com |
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| ABSTRACT |  | ||||||||||||||||||||||
: Cryptococcosis is one of the commonly associated opportunistic fungal infection in patients with AIDS, caused mostly by Cryptococcus neoformans. Lung and the central nervous system are the most common sites affected. Osseous involvement occurs in 5-10% of patients with disseminated Cryptococcus. Although extremely rare, sometimes it can present as isolated cryptococcal osteomyelitis in HIV-negative patients. Here we present a case report of seventy two year old healthy male with isolated swelling of parietal bone on right side of scalp who is HIV negative with prior history of completely cured pulmonary tuberculosis. The patient was treated with fluconazole and repeated aspirations were performed. He was cured completely after three months of treatment. Even though isolated cryptococcal osteomyelitis of the parietal bone of scalp in HIV-negative is extremely rare, it should be considered in the relevant differential diagnosis of any localized swelling of scalp since it can be cured completely. | |||||||||||||||||||||||
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| Keywords : Antifungal, Cryptococcal, Immunocompetent,Osteomyelitis | |||||||||||||||||||||||
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| Case Report |
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A seventy two year old male presented to the surgery outpatient department of Regional institute of medical sciences, Imphal with chief complaint of swelling over the right side of scalp for one months (Table/Fig 1). Initially started as a small swelling, which gradually increased in size and reached the present size. The swelling was occasionally associated with mild pain. No history of trauma, seizure, headache, focal neurological deficits, altered behavior or any other history suggestive of central nervous system involvement. There was no other major complaints suggestive of pulmonary, cardiovascular, gastrointestinal, or genitourinary systems involvement. Patient is neither hypertensive nor diabetic. One year back he was treated for pulmonary tuberculosis. He was declared as cured after six months of antitubercular treatment. There is no other significant past history. On local examination there is a 3*3 cms sized, cystic, transluminent, nontender, non pulsatile swelling over the right side of scalp. Detailed general and systemic examination was performed. Initially, FNAC of swelling and skull X-ray was advised. Skull X-ray showed lytic lesion in the right side of scalp bone. FNAC revealed degenerated neutrophils and numerous capsulated budding yeast cells morphologically resembling cryptococcal species. Cryptococcal abscess of parietal bone was suspected. Antifungal treatment started with Tb. Fluconazole 400mg once daily. Further investigations were planned. Complete haemogram was normal apart from raised ESR levels (100mm/1 hr). Liver function tests, renal function tests chest X-ray were with in normal limits. HIV testing for type 1 and 2 were non reactive as well as negative for hepatitis B and C. CT scan of head showed focal bony lytic lesion involving full thickness of right parietal bone with rim enhancing hypodense collection in the adjacent subcutaneous tissue and epidural space suggestive of osteomyelitis (Table/Fig 2),(Table/Fig 3). Aspiration with wide bore needle was done and specimen (Table/Fig 4) sent for microbiological analysis. Routine bacteriological culture report was negative. India ink preparation, Geimsa staining, Meyer’s mucicarmine staining, PAS staining showed Cryptococcus morphology (capsulated budding yeast cells) (Table/Fig 5),(Table/Fig 6),(Table/Fig 7). The CALAS test (Cryptococcal Antigen latex Agglutination System,Meridian diagnostics,Cincinnati, Ohio) for Cryptococcus was positive with a serum titre of 1:100. Specimen was sent for fungal culture and report came as positive for cryptococcus after 2 wks of incubation. Fungal culture of the aspirate on Sabouraud’s dextrose agar and Bird seed Agar showed growth of brown pigmented colonies of Cryptococcus neoformans due to melanin production (Table/Fig 8),(Table/Fig 9). The fungal isolates obtained were further confirmed by positive urease test , positive inositol assimilation test and the growth on L-Canavanine glycine bromothymol blue (CGB) agar. Sputum, urine and blood were collected and subjected to india ink preparation and fungal culture which was turned out to be negative for cryptococcal speciecs. Antifungal susceptibility testing was done by disc diffusion method for Amphotericin B (20µg), fluconazole (25µg) and voriconazole (1µg) using reference for Cryptococcus species as given in CLSI M44A guideline. It was sensitive to Amphotericin B, fluconazole as well as for voriconazole. Repeated aspirations were performed with proper dressing. Anti fungal therapy with fluconazole continued. Patient responded well with treatment (Table/Fig 10). Size of swelling completely reduced after one month of treatment. Fluconazole was continued for 3 months and stopped after that. | |||||||||||||||||||||||
| Discussion |
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Cryptococcus neoformans is an encapsulated heterobasidiomycetous fungus which crosses the entire spectrum of patient populations, from the apparently immunocompetent host without an underlying disease to those severely immunocompromised from infection with the human immunodeficiency virus (HIV), an organ transplantation, or a malignancy (1). It has a wide range of clinical presentations, which can vary from asymptomatic colonization of the respiratory airways to dissemination of infection into any part of the human body [1,2]. Cryptococcal osteomyelitis usually occurs as part of a disseminated cryptococcal infection (2). Osseous involvement occurs in 5-10% of patients with disseminated Cryptococcus (3). Isolated cryptococcal osteomyelitis is very rare and more so in immunocompetent patients [2-4]. Of all cryptococcal infections, 10-40% of patients have no apparent immune deficiency (5). Disseminated disease may occur in up to 62% of HIV- seronegative patients with cryptococcosis; however, cryptococcal osteomyelitis is rare (5). Seventy-five percent of cases involved only one single site of bone infection, with vertebrae being the most common site. Scalp osteomylitis is extremely rare (6). Sarcoidosis is the most common underlying disease, followed by tuberculosis and previous steroid therapy (6). Till 2005 only 47 cases of isolated cryptococcal osteomyelitis have been reported in HIV-negative patients (7). Cryptococcal osteomyelitis usually present with swelling and pain of the soft tissue surrounding the bone that may or may not be tender which is usually not associated with erythema or sinus formation but with mild elevation of local temperature. Radiologic examination reveals a lytic lesion with mild or absent periosteal reaction (8). As per the Infectious Diseases Society of America (IDSA) 2010 guidelines non-meningeal, non-pulmonary cryptococcosis with infection limited to a single site can be successfully treated with fluconazole (400 mg [6 mg/kg] per day orally) for six to twelve months (9). Previous study have shown that single site cryptococcal osteomyelitis can be completely cured after ten weeks of fluconazole at a dose of 200 mg/day (10). In our case, the patient was completely cured with oral fluconazole 400 mg for a period of three months. | |||||||||||||||||||||||
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