Tophaceous gout in a female premenopausal patient with an unexpected diagnosis of glycogen storage disease type Ia: a case report and literature review

Clin Rheumatol. 2016 Nov;35(11):2851-2856. doi: 10.1007/s10067-016-3290-1. Epub 2016 May 2.

Abstract

A young female with recurrent tophaceous gout and infertility presented to our clinic. On clinical evaluation, hypoglycaemia, hypertriglyceridaemia, lactic acidosis, and hepatomegaly were noted. Targeted gene sequencing revealed a novel composite heterozygous c.190G>T/c.508C>T mutation in the G6PC gene of the patient, leading to a diagnosis of glycogen storage disease type Ia. Her father possessed a heterozygous c.190G>T mutation, and her mother possessed a heterozygous c.508C>T mutation. A search of the previous literature revealed 16 reported cases of glycogen storage disease type Ia with gout. Here, we describe a female patient with gout, review previous cases, and discuss the mechanisms of gout and hyperuricaemia in glycogen storage disease type Ia.

Keywords: Glycogen storage disease type Ia; Gout; Hyperuricaemia.

Publication types

  • Case Reports
  • Review

MeSH terms

  • DNA Mutational Analysis
  • Female
  • Glucose-6-Phosphatase / genetics
  • Glycogen Storage Disease Type I / complications
  • Glycogen Storage Disease Type I / diagnosis*
  • Glycogen Storage Disease Type I / genetics
  • Gout / complications*
  • Gout / genetics
  • Heterozygote
  • Humans
  • Mutation

Substances

  • Glucose-6-Phosphatase
  • G6PC2 protein, human