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Emergency surgery in chronic intestinal pseudo-obstruction due to mitochondrial neurogastrointestinal encephalomyopathy: case reports.
Granero Castro, Pablo; Fernández Arias, Sebastián; Moreno Gijón, María; Alvarez Martínez, Paloma; Granero Trancón, José; Álvarez Pérez, Jose Antonio; Lamamie Clairac, Eduardo; González González, Juan José.
Afiliación
  • Granero Castro P; Department of General Surgery, Hospital Universitario Central de Asturias, Oviedo, Spain. pgranerocastro@aecirujanos.es.
Int Arch Med ; 3: 35, 2010 Dec 08.
Article en En | MEDLINE | ID: mdl-21143863
Chronic intestinal pseudo-obstruction (CIPO) is a syndrome characterized by recurrent clinical episodes of intestinal obstruction in the absence of any mechanical cause occluding the gut. There are multiple causes related to this rare syndrome. Mitochondrial neurogastrointestinal encephalomyopathy (MNGIE) is one of the causes related to primary CIPO. MNGIE is caused by mutations in the gene encoding thymidine phosphorylase. These mutations lead to an accumulation of thymidine and deoxyuridine in blood and tissues of these patients. Toxic levels of these nucleosides induce mitochondrial DNA abnormalities leading to an abnormal intestinal motility.Herein, we described two rare cases of MNGIE syndrome associated with CIPO, which needed surgical treatment for gastrointestinal complications. In one patient, intra-abdominal hypertension and compartment syndrome generated as a result of the colonic distension forced to perform emergency surgery. In the other patient, a perforated duodenal diverticulum was the cause that forced to perform surgery. There is not a definitive treatment for MNGIE syndrome and survival does not exceed 40 years of age. Surgery only should be considered in some selected patients.

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: Int Arch Med Año: 2010 Tipo del documento: Article País de afiliación: España Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Idioma: En Revista: Int Arch Med Año: 2010 Tipo del documento: Article País de afiliación: España Pais de publicación: Reino Unido