Background. The orofaciodigital syndromes (OFDS) are a heterogeneous group of syndromes that affect the face, oral cavity, and the digits. OFDS type IV (OMIM %258860) is rare and characterized by broad nasal root and tip, orbital hypertelorism or telecanthus, micrognathia, hypoplastic mandible, and low-set ears. Oral symptoms may include cleft lip, cleft or highly arched palate, bifid uvula, cleft or hypoplastic maxillary and mandibular alveolar ridge, oral frenula, lingual hamartoma, and absent or hypoplastic epiglottis. Dental anomalies are common and generally include disturbances in the number of teeth. Case Report. This report presents a six-year-old girl, referred with the chief complaint of missing teeth. She was diagnosed as having OFDS type IV based on clinical findings. Her parents reported three deceased children and two fetuses that had the same phenotype. She was the seventh child of consanguineous parents who were first cousins. Conclusion. This is a very rare syndrome. Many reported OFDS type IV cases have consanguineous parents, consistent with an autosomal recessive trait. Manifestation of cleft palate in the healthy sibling may be mild expression of the disorder or an unrelated isolated cleft.
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http://dx.doi.org/10.1155/2014/605892 | DOI Listing |
Phys Med Biol
October 2024
Patrick G. Johnston Centre for Cancer Research, Queen's University Belfast, Belfast BT9 7AE, United Kingdom.
. Optical fibre dosimeters (OFDs) offer great promise for real-timedose measurement in radiation-based treatment modalities such as radiotherapy and brachytherapy. This is attributed to their many useful qualities such as high spatial resolution and sensitivity.
View Article and Find Full Text PDFInt J Clin Pediatr Dent
February 2024
Department of Oral and Maxillofacial Pathology, Kalinga Institute of Dental Sciences (KIDS), KIIT Deemed to be University, Bhubaneswar, Odisha, India.
Retina
January 2023
NIHR Biomedical Research Centre, Moorfields Eye Hospital, NHS Foundation Trust, London, United Kingdom.
Purpose: To report characteristics of outer foveal defects (OFDs) in type-2 macular telangiectasia (MacTel) on spectral domain optical coherence tomography.
Methods: This was a single-center observational study. From a registry of 745 patients with MacTel, patients with OFDs were characterized.
J Craniofac Surg
November 2022
Department for Cleft Lip and Palate Treatment, Plastic Surgery Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China.
Orofaciodigital syndrome type 1 (OFDS1) is a genetic disorder characterized by specific oral, facial, and limb malformations. A 14-month-old girl with congenital cleft palate, lower lip midline cleft, and digital anomalies admitted to our hospital was preliminarily diagnosed with OFDS1. Genetic analysis revealed that she carried a heterozygous variant of OFD1 at locus Xp22.
View Article and Find Full Text PDFNat Commun
October 2022
Department of Biochemistry and Molecular Biology, The University of Texas Medical Branch at Galveston, Galveston, TX, 77550, USA.
Oral-facial-digital (OFD) syndromes are a heterogeneous group of congenital disorders characterized by malformations of the face and oral cavity, and digit anomalies. Mutations within 12 cilia-related genes have been identified that cause several types of OFD, suggesting that OFDs constitute a subgroup of developmental ciliopathies. Through homozygosity mapping and exome sequencing of two families with variable OFD type 2, we identified distinct germline variants in INTS13, a subunit of the Integrator complex.
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