A novel mutation in a Fijian boy with Shwachman-Diamond syndrome

J Pediatr Hematol Oncol. 2009 Nov;31(11):847-9. doi: 10.1097/MPH.0b013e3181b9c949.

Abstract

Shwachman-Diamond syndrome (SDS) is an autosomal recessive disorder characterized by pancreatic insufficiency, bone marrow dysfunction, and metaphyseal chondrodysplasia. SDS is associated with mutations in the Shwachman-Bodian-Diamond Syndrome gene, with 90% of reported mutations in exon 2. We present a Fijian boy with SDS who has a novel A>G substitution in exon 1 of the Shwachman-Bodian-Diamond Syndrome gene that has not been reported in the literature. This patient's unique clinical course includes the presence of a cleft lip and episodic hypoglycemia. SDS lacks a clear genotype-phenotype correlation, as is showed by the heterogeneity in its clinical presentation.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / pathology
  • Fiji
  • Humans
  • Infant, Newborn
  • Male
  • Mutation, Missense*
  • Proteins / genetics*

Substances

  • Proteins
  • SBDS protein, human