Abstract
Purpose
Our study compared the rates of union achieved with the Ilizarov method in congenital pseudarthrosis of the tibia (CPT) associated with neurofibromatosis type 1 (NF1) or CPT of idiopathic origin in paediatric patients.
Methods
We studied the outcomes of 28 children that were treated for CPT between 2005 and 2013. Group 1 included children (n = 14, mean age = 9.7 years) with CPT associated with NF1 while group 2 were CPT cases that had radiographic confirmation of dysplastic lesions in the tibia but lacked clinical NF1 manifestations (n = 14, mean age = 8.6 years). There was no statistical difference between the groups regarding their age or number of previous operations per patient. Individual technical solutions were planned for each patient but coaptation of bone fragments and autologous local tissue grafting to achieve a greater bone thickness and contact area at the pseudarthrosis level were mainly used. Refracture-free rate after the first operation, number of re-operations per patient, and union rates in the groups were compared.
Results
Bone union and weight bearing were obtained in all the cases after the first operation. Refracture-free rate was 42.86 % in group 1 and 35.71% in group 2 (no statistical difference, p > 0.05). Mean number of re-operations per patient was 1.07 and 0.78 respectively (p > 0.05). Subsequent treatment for refractures with the Ilizarov techniques gained 92.86% of union in both groups at the follow-ups by completion of the study (range, 2–9 years).
Conclusions
The Ilizarov method yields comparable results in the management of CPT associated with NF1 or tibial dysplasia of idiopathic origin in paediatric cases. Further research should focus on the ways to support the Ilizarov method in order to reduce the number of repetitive surgeries or eliminate them.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
References
Andersen KS (1976) Congenital pseudarthrosis of the tibia and neurofibromatosis. Acta Orthop Scand 47(1):108–111
Vander Have KL, Hensinger RN, Caird M, Johnston C, Farley FA (2008) Congenital pseudarthrosis of the tibia. J Am Acad Orthop Surg 16(4):228–236
Hefti F, Bollini G, Dungl P, Fixsen J, Grill F, Ippolito E, Romanus B, Tudisco C, Wientroub S (2000) Congenital pseudarthrosis of the tibia: history, etiology, classification and epidemiologic data. J Pediatr Orthop B 9(1):11–15
Ohnishi I, Sato W, Matsuyama J, Yajima H, Haga N, Kamegaya M, Minami A, Sato M, Yoshino S, Oki T, Nakamura K (2005) Treatment of congenital pseudarthrosis of the tibia: a multicenter study in Japan. J Pediatr Orthop 25(2):219–224
Sakamoto A, Yoshida T, Uchida Y, Kojima T, Kubota H, Iwamoto Y (2008) Long-term follow-up on the use of vascularized fibular graft for the treatment of congenital pseudarthrosis of the tibia. J Orthop Surg Res 3:13
Mathieu L, Vialle R, Thevenin-Lemoine C, Mary P, Damsin JP (2008) Association of Ilizarov’s technique and intramedullary rodding in the treatment of congenital pseudarthrosis of the tibia. J Child Orthop 2(6):449–455
Thabet AM, Paley D, Kocaoglu M, Eralp L, Herzenberg JE, Ergin ON (2008) Periosteal grafting for congenital pseudarthrosis of the tibia: a preliminary report. Clin Orthop 466:2981–2994
Gouron R, Deroussen F, Juvet M, Ursu C, Plancq MC, Collet LM (2011) Early resection of congenital pseudarthrosis of the tibia and successful reconstruction using the Masquelet technique. J Bone Joint Surg (Br) 93(4):552–554
Johnston CE, Birch JG (2008) A tale of two tibias: a review of treatment options for congenital pseudarthrosis of the tibia. J Child Orthop 2:133–149
Grill F, Bollini G, Dungl P, Fixsen J, Hefti F, Ippolito E, Romanus B, Tudisco C, Wientroub S (2000) Treatment approaches for congenital pseudarthrosis of tibia: results of the EPOS multicenter study. European paediatric orthopaedic society (EPOS). J Pediatr Orthop B 9(2):75–89
Ferner RE, Huson SM, Thomas N, Moss C, Willshaw H, Evans DG et al (2007) Guidelines for the diagnosis and management of individuals with neurofibromatosis 1. J Med Genet 44(2):81–88
Crawford AH, Schorry EK (1999) Neurofibromatosis in children: the role of the orthopaedist. J Am Acad Orthop Surg 7(4):217–230
Exner GU, von Hochstetter AR (1995) Fibrous dysplasia and osteofibrous dysplasia. Orthopade 24(1):50–56
Cho TJ, Seo JB, Lee HR, Yoo WJ, Chung CY, Choi IH (2008) Biologic characteristics of fibrous hamartoma from congenital pseudarthrosis of the tibia associated with neurofibromatosis type 1. J Bone Joint Surg Am 90(12):2735–2744
Lee DY, Cho TJ, Lee HR, Lee K, Moon HJ, Park MS, Yoo WJ, Chung CY, Choi IH (2011) Disturbed osteoblastic differentiation of fibrous hamartoma cell from congenital pseudarthrosis of the tibia associated with neurofibromatosis type I. Clin Orthop Surg 3(3):230–237
Riddle ND, Bui MM (2013) Fibrous dysplasia. Arch Pathol Lab Med 137(1):134–138
Sakamoto A, Oda Y, Iwamoto Y, Tsuneyoshi M (2000) A comparative study of fibrous dysplasia and osteofibrous dysplasia with regard to GSa mutation at the Arg201 Codon. J Mol Diagn 2(2):67–72
Ilizarov GA, Gracheva VI (1971) Bloodless treatment of congenital pseudarthrosis of the crus with simultaneous elimination of shortening using dosed distraction. Ortop Travmatol Protez 32(2):42–46
Horn J, Steen H, Terjesen T (2013) Epidemiology and treatment outcome of congenital pseudarthrosis of the tibia. J Child Orthop 7(2):157–166
Khan T, Joseph B (2013) Controversies in the management of congenital pseudarthrosis of the tibia and fibula. Bone Joint J 95-B(8):1027–1034
Cho TJ, Choi IH, Lee SM, Chung CY, Yoo WJ, Lee DY, Lee JW (2008) Refracture after Ilizarov osteosynthesis in atrophic-type congenital pseudarthrosis of the tibia. J Bone Joint Surg (Br) 90(4):488–493
Agashe MV, Song SH, Refai MA, Park KW, Song HR (2012) Congenital pseudarthrosis of the tibia treated with a combination of Ilizarov’s technique and intramedullary rodding. Acta Orthop 83(5):515–522
Pannier S, Pejin Z, Dana C, Masquelet AC, Glorion C (2013) Induced membrane technique for the treatment of congenital pseudarthrosis of the tibia: preliminary results of five cases. J Child Orthop 7(6):477–485
Muhammad Abdul Jamil MK, Abdul Rashid AH, Ibrahim S (2013) Congenital pseudarthrosis of the tibia: healing by lengthening over a rod without compression of the nonunion. a preliminary report. J Pediatr Orthop B 22(3):207–212
Granchi D, Devescovi V, Baglio SR, Magnani M, Donzelli O, Baldini N (2012) A regenerative approach for bone repair in congenital pseudarthrosis of the tibia associated or not associated with type 1 neurofibromatosis: correlation between laboratory findings and clinical outcome. Cytotherapy 14(3):306–314
Das SP, Ganesh S, Pradhan S, Singh D, Mohanty RN (2014) Effectiveness of recombinant human bone morphogenetic protein-7 in the management of congenital pseudarthrosis of the tibia: a randomised controlled trial. Int Orthop 38(9):1987–1992
Dohin B, Kohler R (2012) Masquelet’s procedure and bone morphogenetic protein in congenital pseudarthrosis of the tibia in children: a case series and meta-analysis. J Child Orthop 6(4):297–306. doi:10.1007/s11832-012-0421-3
Petramala L, Giustini S, Zinnamosca L, Marinelli C, Colangelo L, Cilenti G, Formicuccia MC, D’Erasmo E, Calvieri S, Letizia C (2012) Bone mineral metabolism in patients with neurofibromatosis type 1 (von Recklingausen disease). Arch Dermatol Res 304(4):325–331
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest statement
The authors declare that they have no competing interests.
Funding
The authors received no funding for doing this study.
Rights and permissions
About this article
Cite this article
Borzunov, D.Y., Chevardin, A.Y. & Mitrofanov, A.I. Management of congenital pseudarthrosis of the tibia with the Ilizarov method in a paediatric population: influence of aetiological factors. International Orthopaedics (SICOT) 40, 331–339 (2016). https://doi.org/10.1007/s00264-015-3029-7
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00264-015-3029-7