Subglossopalatal Synechia in Association with Cardiac and Digital Anomalies

Abstract

A 1-day-old boy in respiratory distress had a midline soft tissue band between the floor of the mouth and the posterior edge of the hard palate. There was also a soft palatal cleft, cardiac anomalies, and a hypoplastic right fifth finger and toe. Although his airway initially improved following urgent excision of the subglossopalatal band, he continued to have episodic desaturations. A tongue–lip adhesion opened his airway, and he subsequently underwent resection of juxtaductal aortic coarctation and ligation of patent ductus arteriosus and left superior vena cava. Congenital oral synechiae are uncommon. Affected infants often require prompt intervention secondary to respiratory distress and feeding difficulty. Review of the literature indicates that midline subglossopalatal synechia with cardiac and digital anomalies may be in the oromandibular–limb hypogenesis spectrum.

Subglossopalatal Membrane With Associated Cleft Palate, Cardiovascular, and Neurologic Anomalies

“…Subglossopalatal membrane is a rare congenital oral synechia. It has been described in the literature only a handful of times 1–8 since Nakajima et al 1 first defined it in 1979 (Supplementary Digital Content, Table 1, http://links.lww.com/SCS/D433). Delineation between SGPM and BPM remnants based on embryologic anatomy and associated anomalies have been proposed 2,9 …”

“…It was first described in the literature by Nakajima et al 1 The etiology is unknown, and whereas it has been categorized as a buccopharyngeal membrane (BPM) remnant, Zalzal et al 2 suggests that it may be due to an abnormal fusion event in the 6th or 7th week of gestation. Eight cases in the literature describe subglossopalatal membranes, [1][2][3][4][5][6][7][8] with 5 of them being associated with additional congenital anomalies (Supplementary Digital Content, Table 1, http://links.lww.com/ SCS/D433). Here, we present a case of subglossopalatal membrane with associated anomalies that was treated with surgical excision.…”

Oral Synechia with Epithelial Cyst in Neonate with Cleft Palate: A Case Report

“…Of the cases reviewed, only seven were medially located, and 11 were singular cordlike adhesions, as opposed to multiple adhesions. Midline subglossopalatal synechia has also been described in conjunction with cardiac and digital anomalies, representing a manifestation of oromandibular-limb hypogenesis spectrum (Oh et al, 2008).…”

Intra Oral Epithelial Bands in a New Born: Report of Two Cases and Review of Literature

“…Donepudi et al reported two cases of cleft palate lateral synechia (CPLS) syndrome in a single family and described surgical management of the synechia [13]. Albert K. Oh reported subglossopalatal synechia in association with cardiac and digital anomalies [14]. Snijman et al reported congenital fusion of gums in a 3 week old child along with temporomandibular joint ankylosis [15].…”