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FATAL cryptococcal meningitis in a child with hyper-immunoglobulin M syndrome, with an emphasis on the agent

S M L Suzuki1, F Morelli1, M Negri1

  • 1Section of Medical Mycology, Universidade Estadual de Maringá, Maringá, Brazil.

Journal De Mycologie Medicale
|August 15, 2019
PubMed

Insights

A child with X-linked hyper-immunoglobulin M syndrome (XHIGM) died from Cryptococcus neoformans meningitis. Amphotericin B reduced fungal load in mice, but not in the brain, highlighting treatment challenges.

Area of Science:

  • Mycology
  • Immunology
  • Infectious Diseases

Background:

  • X-linked hyper-immunoglobulin M syndrome (XHIGM) is a primary immunodeficiency increasing susceptibility to opportunistic infections.
  • Cryptococcus neoformans meningitis poses a significant threat, particularly in immunocompromised individuals.

Observation:

  • A fatal case of C. neoformans meningitis occurred in a child with XHIGM.
  • The fungal isolate demonstrated varying in vitro susceptibility to antifungals: amphotericin B (0.5mg/L), fluconazole (4.0mg/L), and voriconazole (0.12mg/L).

Findings:

  • Experimental infection in BALB/c mice showed dissemination of the C. neoformans isolate to the spleen, lungs, and brain.
  • Significant macroscopic organ alterations were observed in infected mice.
  • Amphotericin B treatment reduced fungal burden in the spleen and lungs but was ineffective in the brain.

Implications:

  • This study underscores the challenges in treating cryptococcal meningitis in patients with XHIGM.
  • The findings highlight the need for novel therapeutic strategies targeting fungal dissemination and brain penetration.
Keywords:
Cryptococcus neoformansHyper-IgM syndromeOpportunistic infection

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