Abstract
The group of anomalies that affect the tongue, maxilla, and mandible—with or without limb anomalies—comes under the umbrella of the oromandibular limb hypogenesis syndrome. It is sporadically reported and has a very low incidence. There is no sex predilection for this congenital disease. We report a rare subtype of oromandibular limb hypogenesis syndrome in a young female who presented with impaired speech and deformity in the lower jaw.
References
Brockmann K, Backes H, Auber B, Kriebel T, Stellmer F, Zoll B (2009) Overlap of Moebius and oromandibular limb hy-pogenesis syndrome with gastroschisis and pulmonary hy-poplasia. Am J Med Genet A 12:2832–2837
Hall BD (1971) Aglossia- adactylia. Birth Defects Orig Artic Ser 7:233–236
Chicarilli ZN, Polayes IM (1985) Oromandibular limb hypogenesis syndromes. Plast Reconstr Surg 76:13–24
Nevin NC, Burrows D, Allen G, Kernoham DC (1975) Aglossia adactyli syndrome. J Med Genet 12:89–93
Meundi MA, Nair GR, Sreenivasan P, Raj AC (2013) Oromandibular limb hypogenesis syndrome type IIB: case report of hypoglossia-hypodactyly. Case Rep Dentist. https://doi.org/10.1155/2013/370695
Grippaudo FR, Kennedy DC (1998) Oromandibular-limb hypogenesis syndromes: a case of aglossia with an intraoral band. Br J Plast Surg 51:480–483
Sharma G, Vengal M, Pai KM, Nagpal A (2012) Hypoglossia type 1A: report of a case and review of literature with focus on clinical investigations. Indian J Dent Res 23:264–270
Nakajima T, Takahashi M, Tateno S (1979) Subglosso-palatal membrane. Plast Reconstr Surg 63(574):6
Wadhwani P, Mohammad S, Sahu R (2007) Oromandibular limb hypogenesis syndrome, type IIA, hypoglossia-hypodactylia: a case report. J Oral Pathol Med 36:555–557
Oulis CJ, Thornton JB (1982) Severe congenital hypoglossia and micrognathia with other multiple birth defects. Oral Pathol 11:276–282
Cosfunkirat OK, Velidedeoglu HV, Sahin U, Demir Z (1999) An unusual case of hypoglossia- hypodactylia syndrome. Ann Plast Surg 42:333–336
Yamada A, Konno N, Imai Y, Saitou C, Kochi S (2000) Treatment of hypoglossiahypodactyly syndrome without extremity anomalies. Plast Reconstr Surg 106(274):279
Boraz RA (1985) Thomas congenital micrognathia and microglossia? J plast an experimental approach to treatment. ASDC J Dent Child 52(62):64
Khalil KC, Dayal PK, Gopa Kumar R, Prashanth S (1995) Aglossia: a case report. Quintessence Int 26:359–360
Rasool A, Zaroo MI, Wani AH, Darzi MA, Bashir SA, Bijli AH et al (2009) Isolated aglossia in a six year old child presenting with impaired speech. A Case Rep Cases J 2:7926
Sabouri AA, Safari A, Gharechahi J, Esmailzadeh S (2012) Prosthodontic rehabilitation for total glossectomy with a magnetic detachable mandibular tongue prosthesis: a clinical report. J Prosthodont 21:404–407
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Anand, R., Sharma, A. Oromandibular Limb Hypogenesis Syndrome Type IVB with Hypoglossia and Intraoral Band: A Case Report of a Rare Entity. J. Maxillofac. Oral Surg. (2024). https://doi.org/10.1007/s12663-024-02115-6
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DOI: https://doi.org/10.1007/s12663-024-02115-6