Literature DB >> 32099307

A rare case of pycnodysostosis: Technical difficulties in managing long bone fractures.

M Romans1, Balaji Sambandam1, Justin Moses1, M Pragash1.   

Abstract

Pycnodysostosis is a rare inherited disorder of autosomal recessive trait causing cathepsin K deficiency, leading to failure of osteoclastic activity. Brittle and sclerotic bones which are prone for frequent fractures is the characteristic feature of this congenital disorder. Despite good healing potential there are few issues in the management of fractures in pycnodysostosis patients. In this article we report the challenges faced in managing a fracture of the femoral shaft in a 12 year old girl with pycnodysostosis. For early rehabilitation and to avoid deformity and shortening, we opted for surgical fixation over conservative treatment. Narrow medullary canal ruled out the option for titanium elastic nail fixation. 4.5mm dynamic compression plate was used to fix the fracture. Sclerotic bone made drilling extremely difficult. Deformed femoral shaft allowed plating over the anterior surface only, instead of the routine lateral surface plating. Postoperative fracture healing was satisfactory. Implant was removed after 18 months.
© 2018.

Entities:  

Keywords:  Osteosclerosis; Pycnodysostosis; Short stature

Year:  2018        PMID: 32099307      PMCID: PMC7026621          DOI: 10.1016/j.jcot.2018.09.012

Source DB:  PubMed          Journal:  J Clin Orthop Trauma        ISSN: 0976-5662


  6 in total

1.  Current research on pycnodysostosis.

Authors:  Serap Turan
Journal:  Intractable Rare Dis Res       Date:  2014-08

2.  A challenging paediatric pathological femur fracture in pyknodysostosis (osteopetrosis acro-osteolytica): lessons learnt.

Authors:  Hosam E Matar; Leroy A James
Journal:  BMJ Case Rep       Date:  2014-11-20

3.  Metachronous bilateral subtrochanteric fracture of femur in an osteopetrotic bone: A case report with technical note.

Authors:  Dharmendra Kumar; Vijay Kumar Jain; Hitesh Lal; Rajinder Kumar Arya; Skand Sinha
Journal:  J Clin Orthop Trauma       Date:  2012-09-20

Review 4.  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

5.  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

6.  A case report of pycnodysostosis with atypical femur fracture diagnosed by next-generation sequencing of candidate genes.

Authors:  Hyung Keun Song; Young Bae Sohn; Yong Jun Choi; Yoon-Sok Chung; Ja-Hyun Jang
Journal:  Medicine (Baltimore)       Date:  2017-03       Impact factor: 1.889

  6 in total
  1 in total

Review 1.  Orthopedic Treatment of Pycnodysostosis: A Systematic Review.

Authors:  Taha M Taka; Brandon Lung; Hayk Stepanyan; David So; Steven Yang
Journal:  Cureus       Date:  2022-04-19
  1 in total

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