Ultrastructural deposits appearing as "zebra bodies" in renal biopsy: Fabry disease?- comparative case reports

BMC Nephrol. 2017 May 12;18(1):157. doi: 10.1186/s12882-017-0571-0.

Abstract

Background: Fabry Disease (FD) is a genetic disorder caused by alpha-galactosidase A deficiency. Certain drugs, such as hydroxychloroquine, can produce renal deposits that mimic morphological findings seen in FD, characterizing a type of drug-induced renal phospholipidosis.

Case presentation: Case 1: A 28-year-old female patient with systemic lupus erythematosus who had been using hydroxychloroquine for 14 months presented subnephrotic proteinuria. Renal biopsy showed deposits compatible with FD. Neither activity analysis of alpha-galactosidase A nor genetic analysis were available and were not performed. These deposits were not detected in a subsequent renal biopsy three years after withdrawal of the medication, characterizing a possible hydroxychloroquine-induced renal phospholipidosis. Case 2: A 29-year-old male patient presented with acroparesthesia, angiokeratomas, cornea verticillata and subnephrotic proteinuria. Deposits compatible with FD were detected upon renal biopsy. The evaluation of alpha-galactosidase A showed no activity in both blood and leukocytes. Genetic analysis identified an M284 T mutation in exon 6, and such mutation was also found in other family members.

Conclusion: Clinical investigation is necessary in suspected cases of Fabry Disease upon renal biopsy in order to confirm diagnosis. Drug-induced renal phospholipidosis should be considered in differential diagnosis in cases with intracellular osmiophilic, lamellar inclusions in electron microscopy.

Keywords: Fabry Disease; Hydroxychloroquine; Lysosomal Storage Disorder; Renal Biopsy; Systemic Lupus Erythematosus.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Biopsy / methods*
  • Diagnosis, Differential
  • Fabry Disease / pathology*
  • Female
  • Humans
  • Kidney / pathology*
  • Kidney / ultrastructure
  • Kidney Diseases / pathology*
  • Lipidoses / pathology*
  • Male