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CASE REPORT |
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| Year : 2011 | Volume
: 56
| Issue : 1 | Page : 82-83 |
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| Scedosporium infection in a patient with anti-tnfα therapy |
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Caroline T Nguyen, Siba P Raychaudhuri
Department of Medicine, VA Medical Center Sacramento and UC Davis School of Medicine, Division of Rheumatology, Allergy and Clinical Immunology , Hospital Way, Mather, CA 95655, USA
| Date of Web Publication | 10-Mar-2011 |
Correspondence Address:
Siba P Raychaudhuri
Department of Medicine, UC Davis School of Medicine and VA Sacramento Medical Center, Division of Rheumatology, 10535, Hospital Way, Mather, CA 95655
USA
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0019-5154.77560
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 Abstract | | |
Patients on anti-TNFα therapy are at increased risk for rare opportunistic infections. Here we are reporting a case of Scedosporium apiospermum infection in a patient treated with anti-TNF for 5 years. Patients on anti-TNFα need close follow-up and clinicians should be suspicious for atypical infections in these immunocompromised hosts.
Keywords: Scedosporium, anti-TNF therapy, immunocompromised
How to cite this article:
Nguyen CT, Raychaudhuri SP. Scedosporium infection in a patient with anti-tnfα therapy. Indian J Dermatol 2011;56:82-3 |
| Introduction | |  |
Currently anti-TNF agents are widely used for cutaneous and systemic autoimmune diseases. For dermatologists most common indications for anti-TNF use are psoriasis and psoriatic arthritis. We describe a case of Scedosporium apiospermum infection in a patient receiving long-term treatment with etanercept, a TNF-α inhibitor. S. apiospermum is a ubiquitous filamentous fungus found in soil, polluted water, and contaminated ambient air in hospital isolation rooms. In immunocompromised patients, S. apiospermum can lead to life-threatening pulmonary or disseminated infections. Increasing reports of S. apiospermum infection in the past few years suggests that S. apiospermum is an emergent opportunistic pathogen. [1]
TNF-α inhibitors have had a significant impact in the treatment of inflammatory rheumatologic disease. [2] However, these drugs have immunosuppressive effects. Infectious diseases including tuberculosis, atypical mycobacteria, histoplasmosis, coccidioidomycosis, and various opportunistic infections in patients receiving TNF-a inhibitors have been reported in the literature and postmarketing surveillance. [3],[4] A PUBMED search resulted in one report of Scedosporium infection as a complication of infliximab therapy for ankylosing spondylitis. [5] To the best of our knowledge, this is the first reported case of S. apiospermum infection in a patient receiving etanercept.
| Case Report | | |
Our patient is a 64-year-old man with spondyloarthropathy of undetermined origin. The patient did not have peripheral arthritis. Neither he had active psoriasis lesions, but he had pitting and ridging on nails. He had sacroiliitis and ankylosis of cervical vertebrae. After extensive workup, we narrowed our differential diagnosis to ankylosing spondylitis and psoriatic arthritis. The patient was previously treated with sulfasalazine and NSAID. In 2002, he was started on methotrexate because of continued neck and lower back pain with morning stiffness. In 2004, etanercept was initiated at the dose of 50 mg/week and sulfasalazine was discontinued. He did remarkably well with etanercept in respect to back pain and morning stiffness. In 2008 he presented with increasing and persistent pain in the left orbit. A CT showed chronic left ethmoid sinus disease and sphenoid sinus opacification. He was started on moxifloxacin for 3 weeks with little improvement of symptoms. As he was receiving an anti-TNF agent, we actively pursued investigation for opportunistic and rare infectious agents. A flexible endoscopic examination of the sinuses demonstrated mucopurulent ethmoid disease. He underwent surgery for bilateral sinus sphenoidotomies and left ethmoidectomy. Tissue specimens of the right and left nasal turbinates and nasal washes were sent for evaluation and culture. The tissue isolates showed chronic inflammatory edematous respiratory tissue without any significant pathology in the underlying bones and GMS stains showed no evidence of fungus. However, cultures of the specimen from the sphenoid sinuses returned positive for S. apiospermum. A MRI of head and sinuses did not demonstrate any evidence for invasive rhinocerebral fungal disease. We consulted our infectious disease colleagues and the patient was started on voriconazole 200 mg BID. He completed a 10-week course of antibiotic therapy. His sinus symptoms completely resolved after surgery and antibiotic treatment. Repeat endoscopic examination demonstrated that the ethmoidectomies and sphenoidotomies were patent and clean. Follow-up MRI and CT of the brain and sinuses did not show any evidence for sinusitis or invasion of the adjacent anatomical structures. As there was no histopathological or radiological evidence for invasive fungal infection we felt comfortable to continue etanercept. The patient has continued to take both etanercept and methotrexate for more than 1 year with no signs of re-infection or complication.
| Discussion | | |
Scedosporium apiospermum has been associated with mycetoma, keratitis, endophthalmitis, osteomyelitis, and brain abscesses. S. apiospermum conidia enter via the respiratory tract and germinate with hyphae invasion. A wide range of pulmonary manifestations exists, ranging from simple colonization as seen in patients with cystic fibrosis to fungus ball formation in patients with cavitary lesions and invasive disease, simulating aspergillosis. [5],[6] Sinusitis may present in both immunocompetent and immunocompromised patients with variable severity. [1] As the clinical presentation of Scedosporium infection, including fever, cough, and dyspnea is nonspecific, diagnosis can be based on cytology, histopathology, and isolation of the fungus in culture. Culture confirmation is important as the histological appearance and clinical presentation of S. apiospermum are difficult to distinguish from that of the Aspergillus species on pathological examination. In a case report of three lung transplant patients, mean-time from specimen collection to positive-culture identification for sputum culture was 4.5 days compared to 9.5 days with bronchoalveolar lavage culture. In our patient, positive culture was identified in 10 days. [7]
Treatment for Scedosporium infection continues to be challenging. S. apiospermum has been shown to have intrinsic resistance to many anti-fungal agents, including fluconazole and amphotericin B, the traditional antifungal of choice for disseminated hyalohyphomycoses. [8] Much of the data on the treatment of S. apiospermum pertains to the use of voriconazole. A retrospective review of 107 patients treated for Scedosporium infection with voriconazole showed 57% of patients achieved a successful response after a median of 103 days of therapy. [9] In vitro studies have demonstrated that voriconazole is more active against S. apiospermum than either itraconazole or amphotericin B. [6] The optimal choice and duration of therapy remain unknown. Surgical debridement or drainage for limited disease in combination with antifungal therapy, as was done successfully with our patient, is recommended now. [5],[6],[10]
| References | | |
| 1. | Cortez KJ, Roilides E, Quiroz-Telles F, Meletiadis J, Antachopoulos C, Knudsen T, et al. Infections caused by Scedosporium. Clin Microbiol Rev 2008;21:157-97. 
[PUBMED] [FULLTEXT] |
| 2. | Fleischmann R, Iqbal I. Risk: Benefit profile of etanercept in elderly patients with rheumatoid arthritis, ankylosing spondylitis, or psoriatic arthritis. Drugs Aging 2007;24:239-54.
[PUBMED] [FULLTEXT] |
| 3. | Raychaudhuri SP, Nguyen CT, Raychaudhuri SK, Gershwin ME. Incidence and nature of infectious disease in patients treated with anti-TNF agents. Autoimmun Rev 2009;9:67-81.
[PUBMED] [FULLTEXT] |
| 4. | Wood KL, Hage CA, Knox KS, Kleiman MB, Sannuti A, Day RB, et al. Histoplasmosis after treatment with anti-tumor necrosis factor-a therapy. Am J Respir Crit Care Med 2003;167:1279-82.
[PUBMED] [FULLTEXT] |
| 5. | Ngai JC, Lam R, Ko FW, To KW, Hui DS. Pulmonary Scedosporium infection as a complication of infliximab therapy for ankylosing spondylitis. Thorax 2009;64:184.
[PUBMED] [FULLTEXT] |
| 6. | Koga T, Kitajima T, Tanaka R, Hirokawa H, Ichiki M, Rikimaru T, et al. Chronic pulmonary scedosporiosis simulating aspergillosis. Respirology 2005;10:682-4.
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| 7. | Sheu R, Bricker A, Sahi H, Mohammed T. Psudallescheria boydii (Scedosporium species) in 3 lung transplant recipients: Computed tomography findings and literature review. J Comput Assist Tomogr 2009;33:247-52.
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| 8. | Johnson EM, Szekely A, Warnock DW. In-vitro activity of voriconazole, itraconozole and amphotericin B against filamentous fungi. J Antimicrob Chemother 1998;42:741-5.
[PUBMED] [FULLTEXT] |
| 9. | Troke P, Aguirrebengoa K, Artega C, Ellis D, Heath CH, Lutsar I, et al. Treatment of Scedosporiosis with voriconazole: Clinical experience with 107 patients. Antimicrob Agents Chemother 2008;52:1743-50.
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| 10. | Raj R, Frost AE. Scedosporium apiospermum fungemia in a lung transplant recipient. Chest 2002;121:1714-6.
[PUBMED] [FULLTEXT] |
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