Major congenital synechiae of the oral cavity constitute a clinically confusing spectrum of abnormalities. On the basis of clinical data, we propose two categories: 1) abnormalities secondary to persistence of the buccopharyngeal membrane and 2) abnormalities secondary to formation of ectopic membranes. An ectopic membrane results from abnormal fusion and can be subclassified as a subglossopalatal membrane, glossopalatal ankylosis, or syngnathia. This classification is supported by embryologic studies and is used to reclassify all cases reported since 1900. Distinct differences, such as the presence of associated limb anomalies, emerge; these are reviewed and add support to the proposed classification.
WatanabeKSadakiFTakahamaH. The ultrastructure of oral (buccopharyngeal) membrane formation and rupture in the anuran embryo. Anat Rec1984; 210: 513–24.
2.
DaviesJ. Embryology and anatomy of the head, neck, face, palate, nose, and paranasal sinuses. In: PaparellaMMShumrickDA, eds. Otolaryngology. Vol 1. Philadelphia, Pa: WB Saunders, 1980: 63–123.
3.
FridenbergP. Congenital detachment of faucial pillar and isolation of palato-glossus muscle. Laryngoscope1908; 18: 567–72.
4.
LongacreJJ. Congenital atresia of the oropharynx. Plast Reconstr Surg1951; 8: 341–8.
LamotheD. Un cas d'absence congénitale de la langue avec persistance de la membrane orale. Ann Mai Oreil Larynx1930; 49: 717–9.
11.
KeithA. Concerning the origin and nature of certain malformations of the face, head, and foot. Br J Surg1940; 28: 173–92.
12.
FarringtonRK. Aglossia congenita. NC Med J1947; 8: 24–6.
13.
KouyoumdjianAOMcDonaldJJ. Association of congenital adrenal neuroblastoma with multiple anomalies, including an unusual oropharyngeal cavity (imperforate buccopharyngeal membrane?). Cancer1951; 4: 784–8.
14.
HaywardJRAveryJK. A variation in cleft palate. J Oral Surg1957; 15: 320–4.
15.
BerendesJ. Angeborene Synechie zwischen der Mund-Boden Schleimhaut und den Oberkieferfort-satzen am Rande einer Gaumensplate. Z Hals Nasen Ohrenheilkd1960–1961; 9: 180.
FuhrmannWKochFSchweckendiekW. Autosomal dominant Vererbung von Gaumenspalte und Synechien zwischen Gaumen und Mundboden oder Zunge. Humangenetik1972; 14: 196–203.
ZalzalGHBratcherGOCottonRT. Subglossopalatal membrane. Arch Otolaryngol Head Neck Surg1986; 112: 1101–3.
20.
LegiusEMoermanPFrynsJPVandenbergheKEggermontE. Holzgreve-Wagner-Rehder syndrome: Potter sequence associated with persistent buccopharyngeal membrane. Am J Med Genet1988; 31: 269–72.
21.
KramerW. Zur Entstehung der angeborenen Gaumenspalte. Zentralbl Chir1911; 38: 385–7.
22.
PhelipJA. Ankyloglosse supérieur congénital. Arch Med Enfants1920; 23: 243–4.
JurgenssenOJ. Seltener fall einer Zungerverwachsung. Oesterr Z Kinderheilkd Kinderfuersorge1947; 1: 118–20.
25.
CosackG. Die angeborene Zungen-Munddach Verwachsung als Leitmotiv eines Komplexes van multiplen Abartungen (zur Genese des Ankyloglossum Superius). Z Kinderheilkd1952–1953; 72: 240–9.
26.
BunnigeM. Angeborene Zungen-Munddach-Verwachsung (Ankyloglossum superius congenitum). Ann Paediatr1960; 194: 173–80.
27.
WilsonRAKlimanMEHardymentAF. Ankyloglossia superior (palato-glossal adhesion in the newborn infant). Pediatrics1963; 31: 1051–4.
Kettner. Kongenitaler Zungendefekt. Dtsch Med Wochenschr1907; 33: 532–4.
30.
DavisWB. Congenital deformities of the face. Surg Gynecol Obstet1935; 61: 201–9.
31.
BurketLW. Congenital bony temporomandibular ankylosis and facial hemiatrophy. Review of the literature and report of a case. J Am Med Assoc1936; 106: 1719–22.
32.
SzelelyF. Atresia oris. Budapesti Orvosi Ujsag1941; 39: 15–6.
33.
HochstetterF. Uber zwei Falle epithelialer Syngnathie bei menschlichen Keimlingen. Dtsch Med Wochenschr1948; 108: 1.
34.
MathisH. Uber einen Fall von Ernahrungschwierigkeit bei connataler Syngnathie. Dtsch Zahnaerztl Z1962; 16: 1167–72.
35.
SallehNM. Congenital partial fusion of the mandible and maxilla. Report of a case. Oral Surg Oral Med Oral Pathol1965; 20: 74–6.
36.
PetterssonG. Aglossia congenita with bony fusion of the jaws. Report of one case. Acta Chir Scand1961; 122: 93–5.
37.
SnijmanPCPrinslooJG. Congenital fusion of the gums. Case report. Am J Dis Child1966; 112: 593–5.
38.
HogginsGS. Aglossia congenita with bony fusion of the jaws. Br J Oral Surg1969; 7: 63–5.
PillaiKGKamathVVKumarGSNagamaniN. Persistent buccopharyngeal membrane with cleft palate. A case report. Oral Surg Oral Med Oral Pathol1990; 69: 164–6.
48.
NeumanZShulmanJ. Congenital sinuses of the lower lip. Oral Surg Oral Med Oral Pathol1961; 14: 1415–20.
49.
ShawWCSimpsonJP. Oral adhesions associated with cleft lip and palate and lip fistulae. Cleft Palate J1980; 17: 127–31.
50.
SternbergNSagherVGolanJEidelmanAIBenhurN. Congenital fusion of the gums with bilateral fusion of the temporomandibular joints. Plast Reconstr Surg1983; 72: 385–7.
51.
TraaholtLM. Cleft lip and palate associated with alveolar synechia and filiform palpebral synechia. Plast Reconstr Surg1990; 86: 337–9.
52.
PoyntorFJHigginsTT. Persistent pharyngeal rudiment. Proc R Soc Med1915; 8: 60.
53.
KulievAMGrinbergKNKukharenkoVIKulazenkoVPBogomazovEA. Monosomy 21 in a human spontaneous abortus. Morphogenetic disturbances and phenotype at the cellular level. Hum Genet1977; 38: 137–45.
54.
BrechtKJohnsonCMIII. Complete mandibular agenesis. Report of a case. Arch Otolaryngol1985; 111: 132–4.
55.
GossANBodnerJWAveryJK. Fusion capability of rat embryonic oral tissue in vitro. Arch Oral Biol1970; 15: 797–804.
56.
BodnerJCheneyCAveryJK. Embryonic movements in the rat during palatal closure [16-mm film]. Ann Arbor, Mich: University of Michigan, School of Dentistry, 1969.
57.
KingCTG. Teratogenic effects of meclizine hydrochloride on the rat. Science1963; 141: 353–5.
58.
WarkanyJNelsonRCSchraffenbergerE. Congenital malformations induced in rats by maternal nutritional deficiency. Am J Dis Child1943; 65: 882.
59.
WraggLEDiewertVMKleinM. Spatial relations in the oral cavity and the mechanism of secondary palate closure in the rat. Arch Oral Biol1972; 17: 683–90.
60.
GreeneRMKochharDM. Palatal closure in the mouse as demonstrated in frozen sections. Am J Anat1973; 137: 477–82.
61.
WalkerBE. Effects on palate development of mechanical interference with the fetal environment. Science1959; 130: 981.
62.
GorlinRJPindborgJJ. Cleft lip-palate, congenital lip fistulas, and anomalies of the extremities. In: GorlinRJPindborgJJ, eds. Syndromes of the head and neck. New York, NY: McGraw-Hill, 1964: 117–21.
