Bifid tongue: A rare feature associated with infants of diabetic mother syndrome

Abstract

Infants born to diabetic mothers (IDM) are well documented to have a higher rate of congenital malformations. Sacral agenesis/hypogenesis and caudal dysgenesis are classically linked to maternal diabetes, but many other types of anomalies are more frequent. In this case report, we describe a male infant born to a diabetic mother who in addition to other typical congenital abnormalities was born with an impressive bifid tongue. Accompanying congenital anomalies include unilateral microphthalmia, bilateral microtia, cleft palate, micropenis with unilateral cryptorchidism, bilateral radial hypoplasia, unilateral pre-axial polydactyly, and mid-line central nervous system defects including arhinencephaly and pituitary hypoplasia. Review of the literature reveals an additional case of an infant with a bifid tongue born to a diabetic mother [Comess et al., 1969]. In conclusion, bifid tongue without oral hamartoma, a rare congenital anomaly, can be an associated finding in IDM.

Prevalence of Bifid Tongue and Ankyloglossia in South Indian Population with an Emphasis on its Embryogenesis

“…All these patients did not fissured tongue (0.9%), bifid tongue (0.4%), hairy tongue (0.2%) and macroglossia (0.2%), lingual thyroid nodule (0.1%). Infants born to diabetic mothers are well documented to have a higher rate of congenital malformations (James et al, 2007;Bhuiyan et al, 2009). …”

Bifid tongue, corneal clouding, and Dandy-Walker malformation in a male infant with otopalatodigital syndrome type 2

“…Bifid tongue was seen in 5 out of 16 OFD1 mutation positive patients [Thauvin‐Robinet et al, 2006]. A literature search performed by James et al [2007] revealed that bifid tongue has also been reported in infants of diabetic mothers, hyperimmunoglobulin E syndrome, trisomy 7 mosaicism, short rib‐polydactyly syndrome type IV (Beemer–Langer syndrome [OMIM 269860]), fibrochondrogenesis [OMIM 228520], and in association with cleft palate and Klippel–Feil anomaly. In the OPD2 literature, bifid tongue has only been described once before [Verloes et al, 2000].…”

Isolated congenital bifid tongue

“…[10–13] Bifid tongue has also been reported as a rare feature associated with infants of diabetic mother syndrome. [14] Literature also gives a reference of bifid tongue as a complication of tongue piercing. [15] Associations have also been postulated with cleft palate, mandibular cleft, midline palatomandibular bony fusion and cervical vertebrae.…”