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Case Reports in Dentistry
Volume 2013 (2013), Article ID 370695, 4 pages
http://dx.doi.org/10.1155/2013/370695
Case Report

Oromandibular Limb Hypogenesis Syndrome Type IIB: Case Report of Hypoglossia-Hypodactyly

1Department of Oral Medicine, Diagnosis and Radiology, Dayananda Sagar College of Dental Sciences, Shavige Malleshwara Hills, Kumaraswamy Layout, Bangalore 560078, India
2Department of Oral Medicine, Diagnosis and Radiology, New Horizon Dental College & Research institute, Sakri, Bilaspur 495001, India
3Department of Oral Medicine, Diagnosis and Radiology, Kannur Dental College, Anjarakkandy post, Kannur 670612, India
4Department of Oral Medicine, Diagnosis and Radiology, KVG Dental College and Hospital, Vidhyanagar post, Kurunjibagh, Sullia 574327, India

Received 26 September 2012; Accepted 27 December 2012

Academic Editors: N. Brezniak and C. Ledesma-Montes

Copyright © 2013 Manasa Anand Meundi et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Hypoglossia-hypodactyly is a rare congenital anomaly affecting the tongue and the limbs. Hall in 1971 classified it under a complex group of disorders called oromandibular limb hypogenesis syndromes. It is an extremely rare condition with around 40 cases reported in the world literature. The cause of the syndrome is unknown. Some type of intrauterine trauma is the most widely accepted etiology. The characteristic features of the syndrome are hypoglossia, limb anomalies of variable degree, and micrognathia of the mandible. This unique case report of hypoglossia-hypodactyly was observed in a patient with normal mandible. In addition, patient also had pulmonary regurgitation. His parents and other siblings were normal. Positive prenatal history of maternal hyperthermia was obtained suspecting it to be the cause of the syndrome.