Cutis laxa and fatal pulmonary hypertension: a newly recognized syndrome?

Clin Dysmorphol. 2011 Apr;20(2):77-81. doi: 10.1097/MCD.0b013e3283439676.

Abstract

Cutis laxa is a connective tissue disorder with distinctive lax, redundant, and inelastic skin. It is a genetically heterogenous disorder with autosomal dominant and recessive patterns of inheritance. We report a patient with cutis laxa supported by clinical, microscopic, and ultrastructural findings. Molecular analysis of fibulin-4 and -5, of the α2 subunit of the V-type H+ ATPase, and of the component of the oligomeric Golgi complex 7 (COG7) genes excluded the type I and type II autosomal recessive forms of cutis laxa, and congenital disorders of glycosylation associated with cutis laxa. Remarkably, our patient also presented severe and lethal pulmonary hypertension as a newborn. This case with cutis laxa, severe pulmonary hypertension, and no detectable mutations in fibulin-4 and -5 genes may represent a previously unrecognized syndrome.

Publication types

  • Case Reports

MeSH terms

  • Abnormalities, Multiple*
  • Cryptorchidism
  • Cutis Laxa / congenital*
  • Euthanasia, Passive
  • Fatal Outcome
  • Female
  • Fetal Growth Retardation
  • Hernia, Umbilical
  • Humans
  • Hypertension, Pulmonary / congenital*
  • Infant, Newborn
  • Male
  • Oligohydramnios
  • Pregnancy
  • Syndrome
  • Withholding Treatment