Literature DB >> 12528952

Neurofibromatosis type 1: a clinicopathological study of the orofacial manifestations in 6 pediatric patients.

R Sigillo1, H Rivera, N G Nikitakis, J J Sauk.   

Abstract

Neurofibromatosis type 1 (NF1) is a relatively frequent mucocutaneous syndrome, which is transmitted as an autosomal dominant trait or which may represent neomutation. It is characterized by a variety of clinical manifestations, including multiple neurofibromas that are associated with a high risk of sarcomatous transformation. The aim of this report was to elucidate the orofacial manifestations observed in 6 pediatric patients (between 4 and 15 years of age) diagnosed with NF1. Physical, clinical, radiological, histological, and immunohistochemical studies were performed. Orofacial lesions were observed in all studied patients, located either in the soft tissues (4 cases) or centrally in the jaws (2 cases). All cases showed facial asymmetry, one of them exhibiting marked facial hemihypertrophy. All cases with soft tissue involvement were plexiform neurofibromas, while the intraosseous cases were diagnosed as solitary neurofibromas. Knowledge of the variability of presentation of orofacial soft tissue and bone manifestations of NF1 in children is necessary for prompt diagnosis.

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Year:  2002        PMID: 12528952

Source DB:  PubMed          Journal:  Pediatr Dent        ISSN: 0164-1263            Impact factor:   1.874


  4 in total

1.  ATF4 mediation of NF1 functions in osteoblast reveals a nutritional basis for congenital skeletal dysplasiae.

Authors:  Florent Elefteriou; M Douglas Benson; Hideaki Sowa; Michael Starbuck; Xiuyun Liu; David Ron; Luis F Parada; Gerard Karsenty
Journal:  Cell Metab       Date:  2006-12       Impact factor: 27.287

Review 2.  Solitary submucous neurofibroma of the mandible: review of the literature and report of a rare case.

Authors:  Rita Depprich; Daman D Singh; Petra Reinecke; Norbert R Kübler; Jörg Handschel
Journal:  Head Face Med       Date:  2009-11-13       Impact factor: 2.151

Review 3.  Update on 13 Syndromes Affecting Craniofacial and Dental Structures.

Authors:  Theodosia N Bartzela; Carine Carels; Jaap C Maltha
Journal:  Front Physiol       Date:  2017-12-14       Impact factor: 4.566

4.  Multifocal Head and Neck Neurofibromas with Osseous Abnormalities and Muscular Hypoplasia in a Child with Neurofibromatosis: Type I.

Authors:  Rachna Rath; Sheetal Kaur; Shadab Ali Baig; Punyashlok Pati; Sonalisa Sahoo
Journal:  Case Rep Radiol       Date:  2016-06-12
  4 in total

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