Literature DB >> 20728312

Orthognathic surgery in pycnodysostosis: a case report.

F Hernández-Alfaro1, J Arenaz Búa, M Serra Serrat, J Mareque Bueno.   

Abstract

Pycnodysostosis is an extremely rare genetic osteosclerosis caused by cathepsin K deficiency. It is a human autosomal recessive genetic disorder characterized mainly by osteosclerosis of the skeleton due to decreased bone turnover. It is characterized by short stature, brachycephaly, short and stubby fingers, open cranial sutures and fontanelle, and diffuse osteosclerosis. Multiple fractures of the long bones and osteomyelitis of the jaw are frequent complications. The authors describe an 18-year-old girl with a clinical and radiological diagnosis of pycnodysostosis and the ortho-surgical treatment undertaken. Bimaxillary orthognathic surgery was carried out using rigid fixation and bone grafts. The authors recommend bimaxillary orthognathic surgery as a choice for treating the dentofacial deformities of pycnodysostosis, emphasizing the good and stable results obtained in terms of facial aesthetics and occlusion.
Copyright © 2010 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

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Year:  2010        PMID: 20728312     DOI: 10.1016/j.ijom.2010.07.006

Source DB:  PubMed          Journal:  Int J Oral Maxillofac Surg        ISSN: 0901-5027            Impact factor:   2.789


  5 in total

Review 1.  Clinical and animal research findings in pycnodysostosis and gene mutations of cathepsin K from 1996 to 2011.

Authors:  Yang Xue; Tao Cai; Songtao Shi; Weiguang Wang; Yanli Zhang; Tianqiu Mao; Xiaohong Duan
Journal:  Orphanet J Rare Dis       Date:  2011-05-10       Impact factor: 4.123

2.  Clinical and radiographic features of pycnodysostosis: A case report.

Authors:  Cleomar Rodrigues; Fernando-Antônio Gomes; José-Alcides Arruda; Luciano Silva; Pâmella Álvares; Priscila da Fonte; Ana-Paula Sobral; Marcia Silveira
Journal:  J Clin Exp Dent       Date:  2017-10-01

3.  Clinical and Radiographic Features of Pycnodysostosis with Emphasis on Dentofacial Problems.

Authors:  Hossein Aghili; Seyed Mohammad Ali Tabatabaei; Mahdjoube Goldani Moghadam
Journal:  Case Rep Dent       Date:  2017-11-26

4.  Clinical and cephalometric analysis of three cases with pycnodysostosis: case reports.

Authors:  Eser Capan; Serap Turan; Hulya Kilicoglu
Journal:  J Istanb Univ Fac Dent       Date:  2015-01-31

5.  Pycnodysostosis with Special Emphasis on Dentofacial Characteristics.

Authors:  Aisha Khoja; Mubassar Fida; Attiya Shaikh
Journal:  Case Rep Dent       Date:  2015-11-16
  5 in total

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