Abstract

IntroductionChronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids is a brainstem disorder characterized by perivascular pathologic reaction with lymphocyte infiltration and leading to diplopia, facial palsy, dysarthria, and gait ataxia. It was thought to be an autoimmune disorder without distinct pathogenesis. Chronic hepatitis B virus infection has been proposed in correlation with autoimmune diseases, including central nervous system demyelinating disease. Patients with chronic hepatitis B infection may develop the syndrome of chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids.Case presentationA 34-year-old Taiwanese man who had been a hepatitis B virus carrier for a decade presented to our emergency room. He had influenza symptoms and progressive symptoms of left hemifacial numbness, double vision, and an unsteady gait of 2 days’ duration. Chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids was diagnosed, with increased hepatitis B viral load at the same time. He had no past history of similar neurologic deficits, and his liver function tests had been within normal limits before this episode. After corticosteroid and entecavir treatments, his neurological deficits and neuroimaging anomalies improved and his serum hepatitis B virus DNA viral load normalized.ConclusionsHepatitis B virus infection may induce central nervous system autoimmune reactions, including chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids. In such cases, concomitant administration of corticosteroids and antiviral agent was helpful. We suggest further investigations in patients with regulatory T cell dysfunction, which may assist in clarifying a loss of immune tolerance in patients with such disorders.

Highlights

  • Chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids is a brainstem disorder characterized by perivascular pathologic reaction with lymphocyte infiltration and leading to diplopia, facial palsy, dysarthria, and gait ataxia

  • We suggest further investigations in patients with regulatory T cell dysfunction, which may assist in clarifying a loss of immune tolerance in patients with such disorders

  • Impaired regulatory T cell (Treg) function in peripheral blood caused by complex genetic defects or environmental factors may play a role in HBVrelated, cell-mediated autoimmunity and may have contributed to the increased hepatitis B virus (HBV) DNA viral load in our patient

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Summary

Conclusions

To the best of our knowledge, we report the first case associated with HBV infection. Long-term corticosteroid use had been recommended for CLIPPERS because of the relapsing-remitting character of the disease. When this disorder is accompanied by chronic HBV infection, antiviral therapy is strongly recommended. Consent Written informed consent was obtained from the patient for publication of this case report and any accompanying images. Author details 1Department of Gerontology and Geriatrics, National Taiwan University Hospital, Taipei, Taiwan. Author details 1Department of Gerontology and Geriatrics, National Taiwan University Hospital, Taipei, Taiwan. 2Department of Internal Medicine, National Taiwan University Hospital, Taipei, Taiwan. 3Department of Medical Imaging, National Taiwan University Hospital, Taipei, Taiwan. 4Department of Neurology, National Taiwan University Hospital, No., Changde Street, Zhongzheng District, Taipei City 10048, Taiwan

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