Persistence of Müllerian derivatives and intestinal lymphangiectasis in two newborn brothers: confirmation of the Urioste syndrome

Am J Med Genet. 2001 Nov 15;104(1):69-74. doi: 10.1002/ajmg.1599.

Abstract

We describe two newborn brothers with a pattern of malformation characterized by the persistence of Müllerian duct derivatives, intestinal lymphangiectasia, hypertrophied alveolar ridges, and early death. Postmortem examination showed the presence of a rudimentary uterus, fallopian tubes, the upper third of a vagina, a prostate of normal shape, a dilated colon, and generalized intestinal and pulmonary lymphangiectasia. The syndrome was first delineated by Urioste and co-workers [1993: Am J Med Genet 47:494-503]. These cases confirm the existence of a definite and distinct entity.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / pathology
  • Adult
  • Fatal Outcome
  • Female
  • Humans
  • Infant, Newborn
  • Lymphangiectasis, Intestinal / genetics*
  • Lymphangiectasis, Intestinal / pathology
  • Mullerian Ducts / abnormalities*
  • Syndrome