A tale of 2 cousins: An atypical and a typical case of abetalipoproteinemia

J Clin Lipidol. 2016 Jul-Aug;10(4):1030-1034. doi: 10.1016/j.jacl.2016.01.003. Epub 2016 Feb 13.

Abstract

Abetalipoproteinemia (ABL) is a rare recessive genetic disease caused by mutations of the MTTP gene. This disease is characterised by a defect in the lipidation of APO B and the absence of VLDL and chylomicron production. Patients affected by ABL present neurological, hemalogical and gastro-intestinal symptoms due to deficiency in lipophilic vitamins and fat malabsorption. We herein report the case of two cousins, one presenting classical symptoms of abetalipoproteinemia and one presenting a much attenuated phenotype. The proband carried a novel combination of MTTP mutations, the 1867+1G>A and the R540C mutations. This patient never received any vitamin supplements and was relatively free of symptoms despite an undetectable APO B concentration. Her cousin was homozygous for 1867+1G>A MTTP mutation and presented most of the classical symptoms of ABL. In conclusion we report a very unusual kindred where on affected member is strongly symptomatic of ABL whereas the other presents very mostly asymptomatic disease suggesting that ABL can present itself with a very incomplete clinical penetrance.

Keywords: Abetalipoproteinemia; Apo B; Low-density lipoprotein; MTTP; Vitamin A; Vitamin E.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Abetalipoproteinemia / genetics*
  • Abetalipoproteinemia / metabolism
  • Adult
  • Carrier Proteins / genetics
  • Female
  • Humans
  • Male
  • Mutation
  • Pedigree
  • Phenotype
  • Young Adult

Substances

  • Carrier Proteins
  • microsomal triglyceride transfer protein