Case Report: A Case of Severe Cryptococcal Immune Reconstitution Inflammatory Syndrome Presenting with Brain and Intradural Abscesses in an HIV Patient

Am J Trop Med Hyg. 2020 Aug;103(2):713-718. doi: 10.4269/ajtmh.20-0028. Epub 2020 Apr 23.

Abstract

Clinical worsening or new manifestation of cryptococcal disease following initiation of anti-retroviral therapy (ART) in an HIV patient is a hallmark of cryptococcal immune reconstitution inflammatory syndrome (C-IRIS). However, it can be difficult to distinguish IRIS from worsening or new infection. Here, we present a case of severe C-IRIS involving multiple cerebellar, spinal, and intradural abscesses and spinal arachnoiditis 7 months after ART initiation in an AIDS patient with uncertain prior ART compliance. He had multiple prior episodes of cryptococcal meningitis with complications necessitating ventriculoperitoneal shunt placement and was on suppressive fluconazole when he developed worsening brain manifestations. He received empiric anti-cryptococcal re-induction without improvement. All cerebrospinal fluid cultures remained sterile, with negative Cryptococcus PCR testing, and his condition continued to worsen prior to corticosteroid initiation. Ultimately, C-IRIS was diagnosed by brain biopsy. This case demonstrates an extreme in severity of C-IRIS and in the timeline of presentation after ART initiation.

Publication types

  • Case Reports

MeSH terms

  • Acquired Immunodeficiency Syndrome / drug therapy*
  • Amphotericin B / therapeutic use
  • Anti-Inflammatory Agents / therapeutic use
  • Anti-Retroviral Agents / therapeutic use*
  • Antifungal Agents / therapeutic use*
  • Arachnoid / pathology
  • Arachnoiditis / congenital
  • Arachnoiditis / diagnostic imaging
  • Arachnoiditis / drug therapy
  • Biopsy
  • Brain / pathology
  • Brain Abscess / diagnostic imaging*
  • Brain Abscess / drug therapy
  • Brain Edema / diagnostic imaging
  • Cerebellar Diseases / diagnostic imaging
  • Cerebellar Diseases / drug therapy
  • Empyema, Subdural / diagnostic imaging*
  • Empyema, Subdural / drug therapy
  • Fluconazole / therapeutic use
  • Flucytosine / therapeutic use
  • HIV Infections / drug therapy
  • Humans
  • Immune Reconstitution Inflammatory Syndrome / diagnostic imaging*
  • Immune Reconstitution Inflammatory Syndrome / drug therapy
  • Magnetic Resonance Imaging
  • Male
  • Meningitis, Cryptococcal / drug therapy*
  • Middle Aged
  • Prednisone / therapeutic use
  • Recurrence

Substances

  • Anti-Inflammatory Agents
  • Anti-Retroviral Agents
  • Antifungal Agents
  • Amphotericin B
  • Fluconazole
  • Flucytosine
  • Prednisone

Supplementary concepts

  • Familial spinal arachnoiditis