Craniomaxillofacial morphology alterations in children, adolescents and adults with neurofibromatosis 1: A cone beam computed tomography analysis of a Brazilian sample.
Eloá-Borges Luna,Maria-Elisa-Rangel Janini,Flávia Lima,Raquel-Richelieu-de Andrade Pontes,Fábio-Ribeiro Guedes,Mauro Geller,Licínio Esmeraldo da Silva,Alexandre-Trindade Motta,Karin Soares Gonçalves Cunha +8 more
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TLDR
Brazilian children, adolescents and adults with NF1 have short mandible, maxilla and skull base, and children and adolescents present mandibular retrusion.Abstract:
BACKGROUND Oral manifestations are common in neurofibromatosis 1 (NF1), and include jaws and teeth alterations. Our aim was to investigate the craniomaxillofacial morphology of Brazilian children, adolescents and adults with NF1 using cone beam computed tomography. MATERIAL AND METHODS This study was conducted with 36 Brazilian individuals with NF1 with ages ranging from 4 to 75. The participants were submitted to anamnesis, extra and intraoral exam and cephalometric analysis using cone beam computed tomography. Height of the NF1 individuals was compared to the length of jaws and skull base. The results of the cephalometric measurements of the NF1 group were compared with a control group paired by age, gender and skin color. RESULTS Individuals with NF1 had lower maxillary length (p<0.0001), lower mandibular length (p<0.0001), lower skull base length (p<0.0001). In children and adolescents, the mandible was more posteriorly positioned (p=0.01), when compared with the control group. There was no association between jaws and skull base length with the height of the individuals with NF1. CONCLUSIONS Brazilian children, adolescents and adults with NF1 have short mandible, maxilla and skull base. Moreover, children and adolescents present mandibular retrusion.read more
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Craniofacial and oral alterations in patients with Neurofibromatosis 1
TL;DR: To conclude, the awareness of craniofacial alterations common in NF1 would help avoiding unnecessary and even harmful involvement, e.g. of periapical cemental dysplasia or enlarged mandibular canal which do not require treatment.
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Increased extracellular matrix deposition during chondrogenic differentiation of dental pulp stem cells from individuals with neurofibromatosis type 1: an in vitro 2D and 3D study
Paula Nascimento Almeida,Deuilton do Nascimento Barboza,Eloá Borges Luna,Maria Clara de Macena Correia,Rhayra B. Dias,Ana Caroline Siquara de Sousa,Maria Eugenia Leite Duarte,Maria Isabel D. Rossi,Karin Soares Gonçalves Cunha +8 more
TL;DR: NF1 DPSCs presented increased extracellular matrix deposition during chondrogenic differentiation, which could be related to skeletal changes in individuals with NF1.
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Unilateral Creeping Destruction of Deformed Mandibular Ramus and Angle Associated with Extensive Facial Plexiform Neurofibroma in Neurofibromatosis Type 1: A Case Report with Analysis of the Literature for Diagnosing Osteolytic Events of the Mandible in Tumor-suppressor Gene Syndrome.
TL;DR: This report describes the rare occurrence of slowly advancing, unilateral destruction of proportions of the mandible in NF1, with the affected bone segment completely surrounded by a PNF.
Journal ArticleDOI
Craniofacial bone alterations in patients with neurofibromatosis type 1
J. Chauvel-Picard,Laurence Lion-Francois,Pierre-Aurélien Beuriat,Christian Paulus,Alexandru Szathmari,Carmine Mottolese,Arnaud Gleizal,Federico Di Rocco +7 more
TL;DR: The aim of this paper is to review the neurosurgical and craniofacial surgical modalities to prevent the further progression of the disease by “reconstructing” the normal relationship of the orbit and the skull.
Journal ArticleDOI
Maxillofacial manifestations in children and adolescents with neurofibromatosis 1
TL;DR: The described clinical case demonstrates the difficulties in diagnostic of NFI in children in the absence of mutilating plexiform neurofibromas and provoked significant functional disorders in maxillofacial area which resulted not only in lower quality of life but also mimicked iatrogenic complications of routine dental treatment.
References
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Journal Article
Clinical applications of cone-beam computed tomography in dental practice.
TL;DR: An overview of currently available maxillofacial CBCT systems is provided and the specific application of various CBCT display modes to clinical dental practice is reviewed.
Journal ArticleDOI
Image analysis and superimposition of 3-dimensional cone-beam computed tomography models
TL;DR: Findings such as resorption, hyperplasic growth, displacement, shape anomalies of mandibular condyles, and morphological differences between the right and left sides emphasize the diagnostic value of computed tomography acquisitions.
Journal ArticleDOI
Skeletal abnormalities in neurofibromatosis type 1 : approaches to therapeutic options
Florent Elefteriou,Mateusz Kolanczyk,Aaron Schindeler,David Viskochil,David Viskochil,Janet M. Hock,Elizabeth K. Schorry,Alvin H. Crawford,Jan M. Friedman,David G. Little,Juha Peltonen,John C. Carey,John C. Carey,David S. Feldman,Xijie Yu,Linlea Armstrong,Patricia Birch,David L. Kendler,Stefan Mundlos,Feng Chun Yang,Gina Agiostratidou,Kim Hunter-Schaedle,David A. Stevenson,David A. Stevenson +23 more
TL;DR: This report reviews the clinical skeletal manifestations and available preclinical mouse models and summarizes key issues that present barriers to optimal clinical management of skeletal abnormalities in neurofibromatosis type 1 and addresses major difficulties encountered for the design of clinical trials.
Journal ArticleDOI
Neurofibro matosis: Oral and radiographic manifestations
Steven D. Shapiro,Steven D. Shapiro,Kenneth Abramovitch,Kenneth Abramovitch,Margot L. Van Dis,Margot L. Van Dis,Larry J. Skoczylas,Larry J. Skoczylas,Robert P. Langlais,Robert P. Langlais,Ronald J. Jorgenson,Ronald J. Jorgenson,Robert S. Young,Robert S. Young,Vincent M. Riccardi,Vincent M. Riccardi +15 more
TL;DR: The prevalence of oral and radiographic findings in this sample was 72%, which is much higher than previously reported and the five most common findings are oral neurofibromas, enlarged fungiform papillae, intrabony lesions, wide inferior alveolar canals, and enlarged mandibular foramina.
Journal ArticleDOI
Multiple roles for neurofibromin in skeletal development and growth
Mateusz Kolanczyk,Nadine Kossler,Jirko Kühnisch,Liron Lavitas,Sigmar Stricker,Ulrich Wilkening,Inderchand Manjubala,Peter Fratzl,Ralf Spörle,Bernhard G. Herrmann,Luis F. Parada,Uwe Kornak,Stefan Mundlos +12 more
TL;DR: Nf1 has an important role in the development of joints, as shown by fusion of the hip joints and other joint abnormalities, which are not observed in neurofibromatosis type I, and has multiple essential roles in skeletal development and growth.