Fabry Disease with Aseptic Meningitis: A Case Series and Literature Review of an Underestimated Clinical Presentation

Curr Med Sci. 2022 Apr;42(2):274-279. doi: 10.1007/s11596-022-2578-4. Epub 2022 Apr 13.

Abstract

Objective: Fabry disease (FD) is an X-linked lysosomal storage disease caused by the mutation in the α-galactosidase A gene that leads to a consequently decreased α-galactosidase A enzyme activity and a series of clinical presentations. However, FD accompanied with aseptic meningitis can be relatively scarce and rarely reported, which leads to significant clinical misdiagnosis of this disease.

Methods: Sixteen patients diagnosed with FD based on a decreased activity of α-galactosidase A enzyme and/or genetic screening were identified through a 6-year retrospective chart review of a tertiary hospital. Clinical presentations, brain magnetic resonance imaging, cerebrospinal fluid analysis, treatment and outcome data were analyzed in cases of aseptic meningitis associated with FD.

Results: Three out of 16 cases exhibited aseptic meningitis associated with FD. There was one female and two male patients with a mean age of 33.3 years. A family history of renal failure or hypertrophic cardiomyopathy was found in 3 cases. All cases presented with a persistent or intermittent headache and recurrent ischemic stroke. The cerebrospinal fluid analyses showed mild pleocytosis in 2 patients and an elevated level of protein in all patients. Cerebrospinal fluid cytology revealed activated lymphocytes, suggesting the existence of aseptic meningitis. In the literature review, up to 9 cases presenting with FD and aseptic meningitis were found, which bore a resemblance to our patients in demographic and clinical characteristics.

Conclusion: Our cases suggested that aseptic meningitis in FD might be under-detected and easily misdiagnosed, and should be more thoroughly examined in further cases.

Keywords: Fabry disease; aseptic meningitis; case series; headache; ischemic stroke.

Publication types

  • Review

MeSH terms

  • Adult
  • Fabry Disease* / complications
  • Fabry Disease* / diagnosis
  • Fabry Disease* / genetics
  • Female
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Meningitis, Aseptic* / diagnosis
  • Meningitis, Aseptic* / drug therapy
  • Meningitis, Aseptic* / etiology
  • Retrospective Studies
  • alpha-Galactosidase / therapeutic use

Substances

  • alpha-Galactosidase