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Pleconaril-resistant chronic parechovirus-associated enteropathy in agammaglobulinaemia

Annick A J M van de Ven1, Jan Willem Douma, Carin Rademaker

  • 1Department of Pediatric Immunology and Infectious Diseases, University Medical Center Utrecht/Wilhelmina Children's Hospital, Utrecht, the Netherlands.

Antiviral Therapy
|June 21, 2011
PubMed

Insights

Common variable immunodeficiency (CVID) patients can develop severe protein-losing enteropathy due to chronic viral infections. Immunosuppressive therapy, not antiviral agents, improved symptoms, suggesting immune dysregulation.

Area of Science:

  • Immunology
  • Virology
  • Gastroenterology

Background:

  • Common variable immunodeficiency (CVID) is a primary immunodeficiency characterized by low immunoglobulin levels and impaired B-cell function.
  • Protein-losing enteropathy (PLE) is a rare but serious complication associated with various gastrointestinal disorders, including immunodeficiencies.

Observation:

  • A 14-year-old patient with CVID presented with severe PLE.
  • Chronic enteral infection with human parechovirus type 1 and norovirus was identified as the likely cause of PLE.

Findings:

  • Standard treatment strategies, including supportive care and the antipicornavirus agent pleconaril, were ineffective in eradicating the viral infection or resolving PLE.
  • The patient's symptoms significantly improved upon initiation of immunosuppressive therapy.

Implications:

  • This case suggests that in immunocompromised hosts with CVID, chronic viral infections can trigger severe gastrointestinal manifestations like PLE.
  • Immune dysregulation, rather than direct viral replication, may play a crucial role in the pathogenesis of PLE in this context.
  • Further research is warranted to explore the complex interplay between CVID, viral infections, and gastrointestinal complications.

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