Literature DB >> 10661860

Ondine's curse and Hirschsprung's disease: neurocristopathic syndrome.

N Urushihara1, Y Nakagawa, N Tanaka, N Uemura, A Yoshida.   

Abstract

We report a female newborn with Ondine's curse and Hirschsprung's disease--neurocristopathic syndrome. The female infant required endotracheal intubation and mechanical ventilation due to apnea which developed soon after birth. She had abdominal distension with bilious vomiting. A barium enema revealed a caliber change at the rectum and rectal biopsies showed no ganglion cells. Colostomy was performed at the age of 17 days. Hypoxemia with hypercapnia was noted during her sleep, and tracheostomy was performed at the age of 55 days. In addition, deafness and pupillary autonomic dysfunction were observed. The definitive surgery for Hirschsprung's disease was performed at the age of 4 months. She is now 2 years old with normal growth but needs ventilator support at home. In this case, we detected no mutation in the RET gene and EDNRB gene.

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Year:  1999        PMID: 10661860     DOI: 10.1055/s-2008-1072300

Source DB:  PubMed          Journal:  Eur J Pediatr Surg        ISSN: 0939-7248            Impact factor:   2.191


  1 in total

1.  Haddad syndrome--congenital central hypoventilation associated with Hirschsprung's disease.

Authors:  Susan D'Souza; R P Khubchandani
Journal:  Indian J Pediatr       Date:  2003-07       Impact factor: 1.967

  1 in total

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