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LETTER TO EDITOR
Year : 2010  |  Volume : 58  |  Issue : 6  |  Page : 949-950

Fatal neurosarcoidosis induced by interferon alpha treatment in chronic hepatitis C


1 Department of Neurosurgery, University Hospital of Fez, Morocco; Clinical Neurosciences Laboratory, University Hospital of Fez, Morocco
2 Department of Hepatogastroenterology, University Hospital of Fez, Morocco
3 Clinical Neurosciences Laboratory, University Hospital of Fez, Morocco; Department of Biophysics and Clinical MRI Methods, Faculty of Medicine and Pharmacy of Fez, Morocco

Date of Acceptance02-Aug-2010
Date of Web Publication10-Dec-2010

Correspondence Address:
Sad Boujraf
Clinical Neurosciences Laboratory, University Hospital of Fez, Morocco; Department of Biophysics and Clinical MRI Methods, Faculty of Medicine and Pharmacy of Fez, Morocco

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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.73751

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How to cite this article:
Benzagmout M, Oudghiri B, Boujraf S, Chakour K, Chaoui ME. Fatal neurosarcoidosis induced by interferon alpha treatment in chronic hepatitis C. Neurol India 2010;58:949-50

How to cite this URL:
Benzagmout M, Oudghiri B, Boujraf S, Chakour K, Chaoui ME. Fatal neurosarcoidosis induced by interferon alpha treatment in chronic hepatitis C. Neurol India [serial online] 2010 [cited 2019 Aug 19];58:949-50. Available from: http://www.neurologyindia.com/text.asp?2010/58/6/949/73751


Sir,

The relationship between the occurrence of sarcoidosis and interferon therapy in viral hepatitis C has been suggested in earlier reports. Clinical manifestations include cutaneous sarcoid lesions, pulmonary nodules and peripheral neuropathy. [1],[2] In this communication, we report a case of severe central nervous system (CNS) sarcoidosis secondary to interferon alpha and ribavirin treatment. [3]

A 47-year-old woman, a known case of chronic viral hepatitis C since 2002, had elevated serum levels of alanine and aspartate aminotransferase, 116 IU/l and 98 UI/l, respectively in 2007, and the serum hepatitis C virus (HCV)-RNA was 6.2 × 10 6 copies/ml. However, physical and abdominal ultrasound examinations were normal. Liver biopsy revealed severe hepatic fibrosis. The patient received peginterferon alfa-2a, 180 μg once a week and ribavirin 400 mg twice a day. With two weeks treatment, the viral load had decreased to 4 × 10 6 copies/ml and the alanine and aspartate aminotransferase serum levels were normalized to 28 IU/l and 32 UI/l, respectively. Four weeks later, the patient noticed weight loss of 5 kg associated with dyspnea and progressive appearance of small subcutaneous firm nodules on the extremities. Chest X-ray was normal. However, the computed tomography scan of chest revealed pulmonary nodules associated with bilateral mediastinal lymphadenopathies. Tuberculin skin test was negative. Bronchoalveolar lavage fluid analysis showed elevated lymphocytes and normal eosinophils and neutrophils. Cultures for fungi and tuberculosis were negative. Biopsy of the skin nodules revealed a noncaseating granulomatous lesion. Serum angiotensin-converting enzyme was 130 U/l (normal, <40 U/l). The diagnosis of sarcoidosis was considered and prednisolone was started at a dose of 60 mg per day. After starting corticosteroid therapy, the serum HCV-RNA of the control was 4 × 10 6 copies/ml. Four days later, she developed heaviness of right upper limb, predominant distally, along with right facial paralysis and aphasia of sudden onset. However, she was lethargic, and the ophthalmologic examination was normal. Magnetic resonance imaging brain showed a gyriform and nodular left frontal and parietal subcortical enhancement along with perilesional edema [Figure 1]. Examination of the cerebrospinal fluid revealed protein 260 mg/dl, glucose 25 mg/dl (concomitant blood sugar being 98 mg/dl) and mononuclear pleocytosis 300 leukocytes/mm 3 . The bacteriological examination was negative. Interferon alfa and ribavirin were stopped and intravenous methyl-prednisolone (10 mg/kg/day) was started for three days, followed by prednisone (1 mg/kg/day). Nevertheless, no clinical improvement was noticed. The neurological state of the patient worsened rapidly; she became comatose and required assisted ventilation. She died a week later due to respiratory decompensation.
Figure 1: Cerebral magnetic resonance imaging in the postgadolinium axial T1-weighted image (a) and flair image (b) showing a gyriform and nodular left frontal and parietal subcortical enhancement associated with an important perilesional brain edema

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Sarcoidosis represents a well known but uncommon complication of antiviral treatment. More than 30 cases of sarcoidosis have been reported following treatment of chronic hepatitis C with interferon alfa. [1],[2] Till date only one case of fatal neurosarcoidosis has been described with interferon therapy. [3] Most reported cases occurred in patients who had received combination treatment, interferon alfa or pegylated interferon alfa and ribavirin. [2] Interferon alpha being a immunomodulator has both direct antiviral activity and powerful stimulation of the immune effects, especially on T helper immune response, [4],[5],[6] which is strongly involved in the pathogenesis of sarcoidosis. [7] Most often discontinuing interferon alfa will result in the resolution of sarcoidosis over several months even without adding immunosuppressive treatment. Prognosis is better for peripheral neurosarcoidosis when compared to central nervous system (CNS) sarcoidosis. CNS involvement occurs in the early stages and is associated with high morbidity and mortality. [8] Our patient probably had neurosarcoidosis after introducing interferon alpha and ribavirin therapy.

Considering earlier reports of the heterogeneous spectra of sarcoidosis manifestations, clinicians should be aware of the associated potential complications while evaluating the benefit/risk ratio of the treatment in patients with chronic hepatitis C infection.

 
  References Top

1.Celik G, Sen E, Ulger AF, Kumbasar OO, Bozkaya H, Alper D, et al. Sarcoidosis caused by interferon therapy. Respirology 2005;10:535-40.  Back to cited text no. 1
[PUBMED]    
2.Hurst EA, Mauro T. Sarcoidosis associated with pegylated interferon alfa and ribavirin treatment for chronic hepatitis C: A case report and review of the literature. Arch Dermatol 2005;141:865-8.  Back to cited text no. 2
[PUBMED]  [FULLTEXT]  
3.Miwa H, Furuya T, Tanaka S, Mizuno Y. Neurosarcoidosis associated with interferon therapy. Eur Neurol 2001;45:288-9.  Back to cited text no. 3
    
4.Schattner A. Interferons and autoimmunity. Am J Med Sci 1988;295:532-44.  Back to cited text no. 4
[PUBMED]    
5.Brinkmann V, Geiger T, Alkan S, Heusser CH. Interferon alpha increases the frequency of interferon gamma-producing human CD4+ T cells. J Exp Med 1993;178:1655-63.  Back to cited text no. 5
[PUBMED]  [FULLTEXT]  
6.Hunninghake GW, Crystal RG. Pulmonary sarcoidosis: A disorder mediated by excess helper T-lymphocyte activity at sites of disease activity. N Engl J Med 1981;305:429-34.  Back to cited text no. 6
[PUBMED]  [FULLTEXT]  
7.Fireman EM, Topilsky MR. Sarcoidosis: An organized pattern of reaction from immunology to therapy. Immunol Today 1994;15:199-201.  Back to cited text no. 7
[PUBMED]    
8.Chapelon-Abric C. Neurosarcoidosis. Ann Med Interne 2001;152:113-24.  Back to cited text no. 8
    


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