Literature DB >> 26174613

Volvulus and bowel obstruction in ATR-X syndrome-clinical report and review of literature.

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.
© 2015 Wiley Periodicals, Inc.

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


  2 in total

1.  Different clinicopathological features of non-elderly sigmoid volvulus patients.

Authors:  Ufuk Uylas; Cuneyt Kayaalp
Journal:  Int J Colorectal Dis       Date:  2020-07-13       Impact factor: 2.571

2.  Syndromic disorder of sex development due to a novel hemizygous mutation in the carboxyl-terminal domain of ATRX.

Authors:  Masaki Takagi; Hiroko Yagi; Ryuji Fukuzawa; Satoshi Narumi; Tomonobu Hasegawa
Journal:  Hum Genome Var       Date:  2017-04-13
  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.