Report on a case of Rothmund-Thomson syndrome associated with esophageal stenosis

Dis Esophagus. 2011 Nov;24(8):E41-4. doi: 10.1111/j.1442-2050.2011.01260.x. Epub 2011 Sep 23.

Abstract

Rothmund-Thomson syndrome (RTS) is a rare autosomal recessive genodermatosis. While its incidence is unknown, approximately 300 cases have been reported in the literature. The syndrome typically presents with a characteristic facial rash (poikiloderma), its diagnostic hallmark, and heterogeneous clinical features including congenital skeletal abnormalities, sparse hair distribution, juvenile cataracts, and a predisposition to osteosarcoma. Gastrointestinal symptoms, such as pyloric stenosis, anal atresia, annular pancreas, and rectovaginal fistula, have also been reported sporadically. This is a report describing a patient diagnosed with RTS referred to us because of dysphagia caused by esophageal stenosis. Long-term results of endoscopic dilation are also presented.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Adult
  • Catheterization*
  • Esophageal Stenosis / complications*
  • Esophageal Stenosis / pathology
  • Esophageal Stenosis / therapy*
  • Female
  • Humans
  • Hypothyroidism / complications
  • Hypothyroidism / drug therapy
  • Infant
  • Rothmund-Thomson Syndrome / complications*
  • Young Adult