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Year : 2011  |  Volume : 54  |  Issue : 1  |  Page : 216-218
Cryptococcal abscess and osteomyelitis of the proximal phalanx of the hand

Department of Orthopaedics, JSSMC, Mysore - 570 004, India

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Date of Web Publication7-Mar-2011

How to cite this article:
Jain K, Mruthyunjaya, Ravishankar R. Cryptococcal abscess and osteomyelitis of the proximal phalanx of the hand. Indian J Pathol Microbiol 2011;54:216-8

How to cite this URL:
Jain K, Mruthyunjaya, Ravishankar R. Cryptococcal abscess and osteomyelitis of the proximal phalanx of the hand. Indian J Pathol Microbiol [serial online] 2011 [cited 2021 Oct 25];54:216-8. Available from: https://www.ijpmonline.org/text.asp?2011/54/1/216/77417

Cryptococcus neoformans is a ubiquitous fungus and usually causes pulmonary infection and meningitis in immuno-compromised hosts, particularly in patients with acquired immunodeficiency syndrome (AIDS). [1] It is an uncommon cause of osteomyelitis; with less then 50 cases reported in the literature since 1956, the year that amphotericin B became available. [2]

A 43-year-old woman was seen in the out-patient department with 2-month-long history of swollen, painful and erythematous right middle finger. She denied a history of fever, chills, sweats or weight loss. There was no history of trauma or septic focus elsewhere in her body. General physical and systemic examination was unremarkable with regard to any major illness. She was not taking any medications at the time of presentation. There was diffuse swelling of the proximal phalanx with purulent discharge at its radial aspect, along with restricted and painful movements of the finger. Osteolytic lesion was seen at base of the proximal phalanx of the finger on radiographs [Figure 1].
Figure 1: Radiographs showing osteolytic lesion at the base of proximal phalanx of middle finger, right hand (AP and oblique view)

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Clinically, bacterial infection including tuberculosis and neoplasms such as enchondroma and giant cell tumor were considered as differential diagnoses and the patient was started on intravenous injection of Cefazolin, 1 g twice daily.

Blood picture showed hemoglobin of 12.5 g/dl, total leukocyte count of 9000 cells/μ l with 69% neutrophils. Erythrocyte sedimentation rate was 35 mm at the end of the first hour. Random blood sugar was 139 mg/dl. Renal and liver function tests were normal. Tests for human immunodeficiency virus, hepatitis B virus and tuberculosis were negative. Culture and sensitivity report of discharge did not show any bacterial growth. The patient was taken up for surgery. Following the drainage of the abscess, the bony lesion was biopsied and curetted. As a preventive measure against the development of a pathological fracture after curettage, and to give additional stability to the finger, Joshis external stabilization system (JESS) fixator was applied spanning over the metacarpo-phalangeal joint of the finger [Figure 2]. Examination of the biopsied sample revealed granulation tissue with necrosis, multinucleate giant cells and foci of acute and chronic inflammation, in which many yeast forms of fungi were seen both extracellularly and in the multinucleated cells. Special fungal stains (periodic acid Schiff (PAS), Gomori's Methenamine silver (GMS), mucicarmine) revealed capsulated budding yeast forms. Masson-Fontana stains showed black to brown colored forms which were consistent with cryptococci [Figure 3], [Figure 4], [Figure 5].
Figure 2: Postoperative clinical and radiological photographs of right hand with JESS fixator in situ

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Figure 3: Small discrete retractile bodies of C. neoformans (H and E, ×400)

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Figure 4: PAS stain highlighting the fungal element (PAS, ×1000)

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Figure 5: Masson-Fontana stain showing brown to black yeast forms of C. neoformans (Masson-Fontana, ×1000)

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The patient was treated with intravenous infusion of amphotericin B (1 mg/kg per day) and oral Fluconazole 200 mg/day. Amphotericin B was discontinued after 10 days. Fluconazole (200 mg/day) was continued for 10 weeks. Patient was reviewed regularly. At the sixth month of follow-up, imaging studies showed a well-healing bony lesion and the proximal phalanx remained stable with satisfactory metacarpo-phalangeal joint movements [Figure 6] and [Figure 7].
Figure 6: Patient achieved full range of movements of hand at 6 months of follow-up

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Figure 7: Radiograph showing a well healing bony lesion at 6 months of follow-up

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C. neoformans occurs widely in nature with a common reservoir in soil and pigeon droppings. The portal of entry of Cryptococcus is the lung from where it disseminates systemically most commonly to the central nervous system. It usually occurs in immunocompromised patients, particularly those with defective cellular immunity. Healthy individuals are rarely affected. Very few cases of isolated cryptococcal osteomyelitis without any other system involvement have been documented in the literature. [3] In a 17-year review by Kiertiburanakul et al., [4] only 40 HIV-negative patients with cryptococcal osteomyelitis were reported. The present case appears to be the first case reported to have cryptococcal osteomyelitis of the proximal phalanx of hand in a HIV-negative patient. [1]

Involvement of bone has been reported in 5% of cases as part of a systemic infection and usually presents with lytic bone lesions with vertebrae being the most common site. [1],[5] The most common radiological presentation of cryptococcal osteomyelitis is a lytic lesion with mild or absent periosteal reaction. [4] The differential diagnoses include neoplasms, certain forms of tuberculosis and infections caused by other agents such as other fungi, Actinomyces and  Brucella More Details. Reliance on the degree of periosteal inflammation may be misleading. [4],[5] The most effective way to establish the diagnosis is by identification of the organism in the surgical biopsy material. Histologic findings typically include fibro-histiocytic and granulomatous inflammation with necrosis, multinucleate giant cells, and acute and chronic inflammation. The staining of the organism capsule with mucicarmine and Masson-Fontana allows differentiation from other fungi morphologically. [5]

Osseous lesions respond variably to treatment and even heal spontaneously. In the published reports, patients were treated successfully with medical treatment alone or with a combination of medical treatment and surgical curettage. According to the infectious disease society of America (IDSA), surgery should be performed for patients with persistent or refractory bone disease. [1] Medically, the patients are usually treated with amphotericin B, 5-flucytosine and long-term maintenance therapy with Fluconazole. IDSA indicates that the treatment of choice in immunocompetent patients with non-CNS disease is Fluconazole. [1] The outcome of HIV-negative patients with cryptococcal osteomyelitis is usually favorable. Single agent therapy is generally avoided, since secondary drug resistance has been documented. The duration of therapy is not well determined and should be based on clinical and radiological improvement.

The possibility of cryptococcal infection should not be forgotten when a patient presents with persistent bone pain and lytic lesions, even though other diagnoses may seem obvious. The key to the successful management lies in a high degree of clinical suspicion, appropriate investigations including bone sampling, and mapping an individualized medical and surgical therapeutic strategy.

   Acknowledgment Top

We are thankful to Dr. Sunila, Professor, Department of Pathology and Dr. Manjunath G.V., Professor & Head, Department of Pathology for Histo-pathological analysis, Special staining and Microscopic photography of Biopsied sample.

   References Top

1.Al-Tawfiq JA, Ghandour J. Cryptococcus neoformans abscess and osteomyelitis in an immunocompetent patient with tuberculous lymphadenitis. Infection 2007;35:377-82.   Back to cited text no. 1
2.Cook PP. Successful treatment of cryptococcal osteomyelitis and paraspinous abscess with fluconazole and flucytosine. South Med J 2001;94:936-8.   Back to cited text no. 2
3.Amit A, Sudish K, Pople IK. Primary calvarial cryptococcal osteomyelitis in a patient with idiopathic lymphopenia. Acta Neurochir (Wien) 2008;150:713-4.  Back to cited text no. 3
4.Kiertiburanakul S, Wirojtananugoon S, Pracharktam R, Sungkanuparph S. Cryptococcosis in human immunodeficiency virus-negative patients. Int J Infect Dis 2006;10:72-8.  Back to cited text no. 4
5.Goldshteyn N, Zanchi A, Cooke K, Agha R. Cryptococcal osteomyelitis of the humeral head initially diagnosed as avascular necrosis. South Med J 2006;99:1140-1.  Back to cited text no. 5

Correspondence Address:
Karun Jain
Department of Orthopaedics, JSSMC, Mysore - 570 004
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0377-4929.77417

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  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]

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