Late-onset dyshormonogenic goitrous hypothyroidism due to a homozygous mutation of the SLC26A7 gene: a case report

Ital J Pediatr. 2024 May 29;50(1):106. doi: 10.1186/s13052-024-01672-3.

Abstract

Background: In this study, we used targeted next-generation sequencing (NGS) to investigate the genetic basis of congenital hypothyroidism (CH) in a 19-year-old Tunisian man who presented with severe hypothyroidism and goiter.

Case presentation: The propositus reported the appearance of goiter when he was 18. Importantly, he did not show signs of mental retardation, and his growth was proportionate. A partial organification defect was detected through the perchlorate-induced iodide discharge test. NGS identified a novel homozygous mutation in exon 18 of the SLC26A7 gene (P628Qfs*11), which encodes for a new iodide transporter. This variant is predicted to result in a truncated protein. Notably, the patient's euthyroid brother was heterozygous for the same mutation. No renal acid-base abnormalities were found and the administration of 1 mg of iodine failed to correct hypothyroidism.

Conclusions: We described the first case of goitrous CH due to a homozygous mutation of the SLC26A7 gene diagnosed during late adolescence.

Keywords: SLC26A7 gene; Case report; Congenital hypothyroidism; Dyshormonogenic goiter; Genetic analysis.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Antiporters
  • Congenital Hypothyroidism* / diagnosis
  • Congenital Hypothyroidism* / genetics
  • Goiter / genetics
  • Homozygote*
  • Humans
  • Male
  • Mutation*
  • Sulfate Transporters* / genetics

Substances

  • Antiporters
  • SLC26A7 protein, human
  • Sulfate Transporters