Cryptococcosis of lumbar vertebra in a patient with rheumatoid arthritis and scleroderma: case report and literature review

BMC Infectious Diseases

Wen-Dong Ruan¹  Hong-Yong Feng¹  Yang Liu¹  Hong-Wei Jia¹  Shi-Qing Feng¹  Guang-Zhi Ning¹  Heng-Xing Zhou¹ 

[1] Department of Orthopaedics, Tianjin Medical University General Hospital, Anshan Road 154, Heping District, Tianjin, 300052, PR China

关键词: Fluconazole;    Scleroderma;    Rheumatoid arthritis;    Lumbar vertebra;    Cryptococcosis;   

Others  :  1170995
DOI  :  10.1186/1471-2334-13-128

 received in 2012-01-26, accepted in 2013-02-20,  发布年份 2013

【 摘 要 】

Background

Although cryptococcosis mainly occurs in the central nervous system and lungs in immunocompromised hosts, it can involve any body site or structure. Here we report the first case of primary cryptococcosis of a lumbar vertebra without involvement of the central nervous system or lungs in a relatively immunocompromised individual with rheumatoid arthritis and scleroderma.

Case presentation

A 40-year-old Chinese woman with rheumatoid arthritis diagnosed 1 year beforehand and with a subsequent diagnosis of scleroderma was found to have an isolated cryptococcal infection of the fourth lumbar vertebra. Her main complaints were severe low back and left leg pain. Cryptococcosis was diagnosed by CT-guided needle biopsy and microbiological confirmation; however, serum cryptococcal antigen titer was negative. After 3 months of antifungal therapy with fluconazole the patient developed symptoms and signs of scleroderma, which was confirmed on laboratory tests. After taking fluconazole for 6 months, the progressive destruction of the lumbar vertebral body had halted and the size of an adjacent paravertebral mass had decreased substantially. On discharge symptoms had resolved and at an annual follow-up there was no evidence of recurrence on the basis of symptoms, signs or imaging investigations.

Conclusion

Although cryptococcosis of the lumbar vertebra is extremely rare, it should be considered in the differential diagnosis for patients with lumbar vertebral masses to avoid missed diagnosis, misdiagnosis and diagnostic delay. Early treatment with antifungals proved to be a satisfactory alternative to surgery in this relatively immunocompromised patient. Any residual spinal instability can be treated later, once the infection has resolved.

【 授权许可】

   
2013 Zhou et al.; licensee BioMed Central Ltd.

【 图 表 】

Figure 1.

Figure 2.

Figure 3.

Figure 4.

Figure 5.

【 参考文献 】

• [1]Henderson HM, Chapman SW: Infections due to fungi, actinomyces, and nocardia. In A practical approach to infectious disease. 4th edition. Edited by Reese RE, Betts RF. Boston: Little, Brown & Company; 1996:672-675.
• [2]Rozenbaum R, Gongalves AJR: Clinical epidemiological study of 171 cases of cryptococcosis. CIin Inf Dis 1994, 18:369-80.
• [3]Hajjeh RA, Conn LA, Stephens DS, Baughman W, Hamill R, Graviss E, Pappas PG, Thomas C, Reingold A, Rothrock G, Hutwagner LC, Schuchat A, Brandt ME: Pinner RW; Cryptococcal Active Surveillance Group: Cryptococcosis: population-based multistate active surveillance and risk factors in human immunodeficiency virusinfected persons. J Infect Dis 1999, 179:449-54.
• [4]Idnurm A, Bahn YS, Nielsen K, Lin X, Fraser JA, Heitman J: Deciphering the model pathogenic fungus Cryptococcus neoformans. Nat Rev Microbiol 2005, 3:753-64.
• [5]Gupta SK, Chhabra R, Sharma BS, Das A, Khosla VK: Vertebral cryptococcosis simulating tuberculosis. Br J Neurosurg 2003, 17(6):556-571.
• [6]Jarvis JN, Harrison TS: Pulmonary cryptococcosis. Semin Respir Crit Care Med 2008, 29(2):141-50.
• [7]Kronstad JW, Attarian R, Cadieux B, Choi J, D’Souza CA, Griffiths EJ, Geddes JM, Hu G, Jung WH, Kretschmer M, Saikia S, Wang J: Expanding fungal pathogenesis: Cryptococcus breaks out of the opportunistic box. Nat Rev Microbiol 2011, 9(3):193-203.
• [8]Bovers M, Hagen F, Boekhout T: Diversity of the Cryptococcus neoformans-Cryptococcus gattii species complex. Rev Iberoam Micol 2008, 25:S4-S12.
• [9]Amit A, Sudish K, Pople IK: Primary calvarial cryptococcal osteomyelitis in a patient with idiopathic lymphopenia. Acta Neurochir (Wien) 2008, 150:713-714.
• [10]Nottebart HC, McGehee RF, Utz JP: Cryptococcus neoformans osteomyelitis: case report of two patients. Sabouraudia 1974, 12(2):127-32.
• [11]Poliner JR, Wilkins EB, Fernald GW: Localized osseous cryptococcosis. J Pediatr 1979, 94(4):597-9.
• [12]Chleboun J, Nade S: Skeletal cryptococcosis. J Bone Joint Surg Am 1977, 59:509-514.
• [13]Freidman GD: The rarity of cryptococcosis in Northern California: the 10 year experience of a large defined population. Am J Epidemiol 1983, 117:230-234.
• [14]Bruno KM, Farhoomand L, Libman BS, Pappas CN, Landry FJ: Cryptococcal arthritis, tendinitis, tenosynovitis, and carpal tunnel syndrome: report of a case and review of the literature. Arthritis Rheum 2002, 47:104-108.
• [15]Zanelli G, Sansoni A, Ricciardi B, Ciacci C, Cellesi C: Muscular-skeletal cryptococcosis in a patient with idiopathic CD4+ lymphopenia. Mycopathologia 2001, 149:137-139.
• [16]Al-Tawfiq JA, Ghandour J: Cryptococcus neoformans abscess and osteomyelitis in an immunocompetent patient with tuberculous lymphadenitis. Infection 2007, 35:377-382.
• [17]Chang WC, Tzao C, Hsu HH, Chang H, Lo CP, Chen CY: Isolated cryptococcal thoracic empyema with osteomyelitis of the rib in an immunocompetent host. J Infect 2005, 51:e117-119.
• [18]Cook PP: Successful treatment of cryptococcal osteomyelitis and paraspinous abscess with fluconazole and flucytosine. South Med J 2001, 94:936-8.
• [19]Wildstein MS, Martin SM Jr, Glaser JA: Cryptococcal osteomyelitis in a 20-year-old male with sarcoidosis. Spine J 2005, 5:467-70.
• [20]Goldshteyn N, Zanchi A, Cooke K, Agha R: Cryptococcal osteomyelitis of the humeral head initially diagnosed as avascular necrosis. South Med J 2006, 99:1140-1141.
• [21]Hawkins C, Flaherty JP: Cryptococcal Osteomyelitis of the Finger Complicated by Meningitis. Infect Dis Clin Pract 2007, 15:345-347.
• [22]Saeed MU, Dacuycuy MA, Kennedy DJ: Cryptococcal Epidural Abscess with Bone Involvement: Case Report and Review of Literature. Infect Dis Clin Pract 2009, 17:193-195.
• [23]Burton R, Gogela N, Rebe K, McNally M, Meintjes G: Cryptococcal immune reconstitution inflammatory syndrome presenting with erosive bone lesions, arthritis and subcutaneous abscesses. AIDS 2009, 23:2371-2373.
• [24]Geller DS, Pope JB, Thornhill BA, Dorfman HD: Cryptococcal pyarthrosis and sarcoidosis. Skeletal Radiol 2009, 38:721-727.
• [25]Kiertiburanakul S, Wirojtananugoon S, Pracharktam R, Sungkanuparph S: Cryptococcosis in human immunodeficiency virus-negative patients. Int J Infect Dis 2006, 10(1):72-78.
• [26]Chuang YM, Ho YC, Chang HT, Yu CJ, Yang PC, Hsueh PR: Disseminated cryptococcosis in HIV-uninfected patients. Eur J Clin Microbiol Infect Dis 2008, 27(4):307-310.
• [27]Smith DK, Neal JJ, Holmberg SD: Unexplained opportunistic infections and CD4+ T-lymphocytopenia without HIV infection. An investigation of Cases in the United States. N Eng J Med 1993, 328:373-379.
• [28]Shu Shun L, Christopher H, Mody : Cryptococcus. Proc Am Thorac Soc 2010, 7:186-196.
• [29]Emmons CW: Saprophytic sources of Cryptococcus neoformans associated with the pigeon (Columba livia). Am J Hyg 1955, 62:227-232.
• [30]Escandón P, Sánchez A, Martínez M, Meyer W, Castañeda E: Molecular epidemiology of clinical and environmental isolates of the Cryptococcus neoformans species complex reveals a high genetic diversity and the presence of the molecular type VGII mating type a in Colombia. FEMS Yeast Res 2006, 6:625-635.
• [31]Datta K, Bartlett KH, Baer R, Byrnes E, Galanis E, Heitman J, Hoang L, Leslie MJ, MacDougall L, Magill SS, Morshed MG, Marr KA: Cryptococcus gattii Working Group of the Pacific Northwest: Spread of Cryptococcus gattii into Pacific Northwest region of the United States. Emerg Infect Dis 2009, 15:1185-1191.
• [32]Fraser JA, Giles SS, Wenink EC, Geunes-Boyer SG, Wright JR, Diezmann S, Allen A, Stajich JE, Dietrich FS, Perfect JR, Heitman J: Same-sex mating and the origin of the Vancouver Island Cryptococcus gattii outbreak. Nature 2005, 437:1360-1364.
• [33]McTaggart L, Richardson SE, Seah C, Hoang L, Fothergill A, Zhang SX: Rapid identification of Cryptococcus neoformans var. grubii, C. neoformans var. neoformans, and C. gattii by use of rapid biochemical tests, differential media, and DNA sequencing. J Clin Microbiol 2011, 49(7):2522-7.
• [34]Gurevitz O, Goldschmied-Reuven A, Block C, Kopolovic J, Farfel Z, Hassin D: Cryptococcus neoformans vertebral osteomyelitis. J Med Vet Mycol 1994, 32:315-318.
• [35]Frazier DD, Campbell DR, Garvey TA, Wiesel S, Bohlman HH, Eismont FJ: Fungal Infections of the Spine: Report of Eleven Patients with Long-Term Follow-up. J Bone Joint Surg Am 2001, 83:560-565.
• [36]Hickie JB, Walker T: Cryptococcosis (torulosis): Some problems in diagnosis and management. Australas Ann Med 1964, 13:229-39.
• [37]Saag MS, Graybill RJ, Larsen RA, Pappas PG, Perfect JR, Powderly WG, Sobel JD, Dismukes WE: Practice guidelines for the management of cryptococcal disease. Clin Infect Dis 2000, 30:710-718.