ArticleJournal of oral and maxillofacial pathology : JOMFP2012
Isolated aglossia congenita: A rare case of oromandibular limb hypogenesis syndrome type I B.
Article in Journal of oral and maxillofacial pathology : JOMFP, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.
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7 citing papers in PubMed, 9 citations in OpenAlex.
- Congenital Hypoglossia With Craniofacial Anomalies Without Limb Defects: A Rare Phenotypic Variant of Oromandibular-Limb Hypogenesis Syndrome.Clinical case reports · 2026Article
- A rare variant of oromandibular limb hypogenesis syndrome: a case report of glossopalatal ankylosis.Frontiers in dental medicine · 2026Article
- The Role of Genetics in Human Oral Health: A Systematic-Narrative Review.Dentistry journal · 2025Review
- Isolated hypoglossia: Oromandibular Limb Hypogenesis Syndrome Type 1 A - A Rare Case Report.Journal of clinical and experimental dentistry · 2024Article
- Oligodontia in the Clinical Spectrum of Syndromes: A Systematic Review.Dentistry journal · 2023Review
- Randomized testing of taste discrimination in a case of congenital aglossia.Journal of oral biology and craniofacial researchArticle
- A Rare Clinical Variant of Oromandibular Limb Hypogenesis Syndrome Type I B.International journal of clinical pediatric dentistryArticle
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1 author at 1 institution in 1 country.
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No grant is acknowledged in the PubMed record.
Abstract
Aglossia congenita (AC), congenital total absence of the tongue, is a very rare midline developmental anomaly, hypothesized to be associated with vascular disruption between the fourth and eighth week of gestation. It was classified by Hall (1971) as part of oromandibular limb hypogenesis syndrome (OLHS) type I B. Most of the cases reported with OLHS are actually hypoglossia with limb abnormalities whereas isolated aglossia is an extremely rare entity. A case of isolated AC is presented in a 28-year-old Indian male. He had long narrow face, tapering chin, low set ears, and microstomia. Intraorally, he had narrow palatal vault, constricted oropharyngeal isthmus, oligodontia, and maxillo-mandibular hypoplasia. Interestingly, the patient showed a median palatal groove, which has not been reported before. He also had an unusual acquired adaptive mechanism to compensate for aglossia. This report presents the manifestations of this rare syndrome, its complications, differential diagnosis, and rehabilitation strategies.
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