| Literature DB >> 26174613 |
Nir Horesh1, Ron Pery1, Imri Amiel1, Chaya Shwaartz1, Chen Speter1, Larisa Guranda1, Mordechai Gutman1, Aviad Hoffman1.
Abstract
Alpha thalassemia-mental retardation, X-linked (ATR-X) syndrome is a rare genetic disorder with a variety of clinical manifestations. Gastrointestinal symptoms described in this syndrome include difficulties in feeding, regurgitation and vomiting which may lead to aspiration pneumonia, abdominal pain, distention, and constipation. We present a 19-year-old male diagnosed with ATR-X syndrome, who suffered from recurrent colonic volvulus that ultimately led to bowel necrosis with severe septic shock requiring emergent surgical intervention. During 1 year, the patient was readmitted four times due to poor oral intake, dehydration and abdominal distention. Investigation revealed partial small bowel volvulus which resolved with non-operative treatment. Small and large bowel volvulus are uncommon and life-threatening gastrointestinal manifestations of ATR-X patients, which may contribute to the common phenomenon of prolonged food refusal in these patients.Entities:
Keywords: ATR-X syndrome; emergency surgery; intestinal volvulus
Mesh:
Year: 2015 PMID: 26174613 DOI: 10.1002/ajmg.a.37252
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802