title: Abnormal intrinsic esophageal innervation in congenital diaphragmatic hernia: a likely cause of motor dysfunction.
abstract: OBJECTIVE
  Patients with congenital diaphragmatic hernia (CDH) often have dilated esophagus and gastroesophageal reflux. Sparse intrinsic esophageal innervation has been described in rats with CDH, but this issue has not been investigated in patients with CDH. The present study tests the hypothesis that innervatory anomalies could account for motor dysfunction in human CDH.
  
  
  METHODS
  The esophagi of CDH (n = 6) and control babies dead of other causes (n = 6) were included in paraffin, transversally sectioned, and immunostained with antineurofilament and anti-S-100 antibodies. The proportion of the section surface occupied by neural structures, the ganglionar surface, and the number of neurons per ganglion were measured in 2 to 5 low-power fields from the proximal and distal esophagus with the assistance of image analysis software. Mann-Whitney tests were used for comparing the results using a threshold of significance of P < .05.
  
  
  RESULTS
  The percentage of neural/muscle surface was similar in the upper esophagus in both groups, but it was significantly decreased in the lower esophagus of patients with CDH in comparison with controls. There was a relative scarcity of neural tissue in the intermuscular plexus of the lower esophagus. On the other hand, the ganglionar surface and the number of neurons per ganglion were identical in both groups. These results were similar with both immunostainings.
  
  
  CONCLUSIONS
  Intrinsic innervation of the lower esophagus in CDH is abnormal in terms of decreased density of neural structures in the intermuscular plexus. These neural crest-derived anomalies could explain in part the esophageal dysfunction in survivors of CDH.
authors.affiliation: Department of Pediatric Surgery, Hospital Universitario La Paz, 28046 Madrid, Spain.
authors.forename: Federica
authors.initials: F
authors.lastname: Pederiva
authors.forename: Jose I
authors.initials: JI
authors.lastname: Rodriguez
authors.forename: Elena
authors.initials: E
authors.lastname: Ruiz-Bravo
authors.forename: Leopoldo
authors.initials: L
authors.lastname: Martinez
authors.forename: Juan A
authors.initials: JA
authors.lastname: Tovar
mesh_terms: D015154:Esophageal Motility Disorders / Q000209:etiology / Q000503:physiopathology*; D004947:Esophagus / Q000294:innervation*; D005260:Female; D006548:Hernia, Diaphragmatic / Q000150:complications / Q000473:pathology / Q000503:physiopathology*; D065630:Hernias, Diaphragmatic, Congenital; D006801:Humans; D007231:Infant, Newborn; D008297:Male