The in utero repair of Tessier number 7 lateral facial clefts created by amniotic band-like compression

J Craniofac Surg. 1998 Nov;9(6):557-62; discussion 563. doi: 10.1097/00001665-199811000-00012.

Abstract

The purpose of this study was to determine the effectiveness of intrauterine reconstruction of lateral facial clefts caused by surgically simulated amniotic band compression. The authors hypothesized that intrauterine lysis of these bands could: 1) prevent the progression of cleft formation; 2) normalize facial development; and 3) allow for the scarless repair of these lateral atypical facial clefts. In a prospective randomized trial, eight 65-day gestational fetal lambs (term, 140 days) were exposed via a maternal hysterotomy. A Tessier 7 facial cleft was made by applying a 2.0 nylon suture as a constriction band to the growing face. The sutures were attached to the zygomatic arch and looped circumferentially into the oral commissure. Two weeks after surgery, four of eight animals were re-exposed. All four animals developed phenotypic changes consistent with Tessier 7 lateral facial clefts. These animals were treated in utero by 1) excising the synthetic constricting band, and 2) opening the epithelialized edges of the lateral facial clefts and performing a primary repair. At birth, the bands on the four untreated animals were cut. The lambs developed to 3 months of age, at which time they were evaluated anthropomorphically for changes in soft tissue measurements, and histologically using hematoxylin and eosin and Mason trichrome stain for the degree of scar at the repair site. Tessier 7 lateral facial clefts were produced in all the untreated animals. Each developed macrostomia with an average 2.3-cm lateral displacement of the oral commissure and partial maxillary clefting induced by the pressure of the restriction band. No evidence of tissue necrosis, maceration, or ulceration was noted. In contrast, animals treated in utero were phenotypically normal. There was no evidence of macrostomia and all anthropomorphic measurements were symmetrical. No bony clefts were seen on gross examination. Histologically, there was no evidence of scar formation at the site of the cleft repair. These findings suggest that like the standard cleft lip, atypical lateral facial clefts can be repaired successfully in utero without scarring. Furthermore, lysis of a restriction band in utero can prevent the progression of this facial deformity, leading to normal development of the face.

MeSH terms

  • Amniotic Band Syndrome / complications
  • Amniotic Band Syndrome / surgery*
  • Animals
  • Disease Models, Animal
  • Facial Bones / abnormalities
  • Facial Bones / surgery*
  • Female
  • Fetal Diseases / pathology
  • Fetal Diseases / physiopathology
  • Fetal Diseases / surgery*
  • Humans
  • Infant, Newborn
  • Maxillofacial Abnormalities / etiology
  • Maxillofacial Abnormalities / surgery*
  • Pregnancy
  • Prospective Studies
  • Sheep