LGCmain
Indian Journal of Pathology and Microbiology
Home About us Instructions Submission Subscribe Advertise Contact e-Alerts Ahead Of Print Login 
Users Online: 8917
Print this page  Email this page Bookmark this page Small font sizeDefault font sizeIncrease font size
IJPM is coming out with a Special issue on "Genitourinary & Gynecological pathology including Breast". Please submit your articles for these issues


 
  Table of Contents    
IMAGE  
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

Click here for correspondence address and email

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 2019 Nov 21];54:216-8. Available from: http://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)

Click here to view


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

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

Click here to view
Figure 4: PAS stain highlighting the fungal element (PAS, ×1000)

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

Click here to view


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

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

Click here to view


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
[PUBMED]  [FULLTEXT]  
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
[PUBMED]  [FULLTEXT]  
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
[PUBMED]  [FULLTEXT]  
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
[PUBMED]  [FULLTEXT]  
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
[PUBMED]  [FULLTEXT]  

Top
Correspondence Address:
Karun Jain
Department of Orthopaedics, JSSMC, Mysore - 570 004
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.77417

Rights and Permissions


    Figures

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

This article has been cited by
1 Disseminated Cryptococcal Osteomyelitis to the Hand in an Immunosuppressed Lymphoma Patient
Wayne A. Chen,Cynthia L. Emory,Benjamin R. Graves
The Journal of Hand Surgery. 2018; 43(3): 291.e1
[Pubmed] | [DOI]
2 Disseminated crytptococcosis presenting as fracture of clavicle and humerus
Kafil Akhtar,Sadaf Haiyat,Saquib Alam,Abdul Qayyum,Rana K Sherwani
Advances in Cytology & Pathology. 2018; 3(5): 115
[Pubmed] | [DOI]
3 Cryptococcal osteomyelitis: a report of 5 cases and a review of the recent literature
Leigh Ann Medaris,Brent Ponce,Zane Hyde,Dennis Delgado,David Ennis,William Lapidus,Matthew Larrison,Peter G. Pappas
Mycoses. 2016; 59(6): 334
[Pubmed] | [DOI]
4 Skeletal cryptococcosis from 1977 to 2013
Heng-Xing Zhou,Lu Lu,Tianci Chu,Tianyi Wang,Daigui Cao,Fuyuan Li,Guangzhi Ning,Shiqing Feng
Frontiers in Microbiology. 2015; 5
[Pubmed] | [DOI]
5 Chronic Hand Infections
Mohammad M. Al-Qattan,Adel A. Helmi
The Journal of Hand Surgery. 2014; 39(8): 1636
[Pubmed] | [DOI]
6 Cryptococcose osseuse chez une patiente porteuse d’une leucémie lymphocytique traitée par fludarabine-cyclophosphamide-rituximab
N. Ettahar,L. Legout,F. Ajana,P. Patoz,M. Massongo,C. Rose,E. Senneville
Journal de Mycologie Médicale / Journal of Medical Mycology. 2013; 23(1): 57
[Pubmed] | [DOI]
7 Utility of fine needle aspiration cytology in the diagnosis of infective lesions
Sridhara Satyanarayana,Abhinandan T. Kalghatgi
Diagnostic Histopathology. 2011; 17(7): 301
[Pubmed] | [DOI]
8 Utility of fine needle aspiration cytology in the diagnosis of infective lesions
Satyanarayana, S., Kalghatgi, A.T.
Diagnostic Histopathology. 2011; 17(7): 301-312
[Pubmed]



 

Top
 
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
    Email Alert *
    Add to My List *
* Registration required (free)  


    Acknowledgment
    References
    Article Figures

 Article Access Statistics
    Viewed5987    
    Printed155    
    Emailed2    
    PDF Downloaded81    
    Comments [Add]    
    Cited by others 8    

Recommend this journal