{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T01:57:04Z","timestamp":1760147824354,"version":"build-2065373602"},"reference-count":22,"publisher":"MDPI AG","issue":"3","license":[{"start":{"date-parts":[[2023,3,3]],"date-time":"2023-03-03T00:00:00Z","timestamp":1677801600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Children"],"abstract":"<jats:p>Background: Congenital pseudoarthrosis of the tibia (CPT) remains a challenge for physicians. Several treatment options have been proposed, but the standard of care remains inconclusive. In this study, we present three patients for whom the failure of prior treatments was managed with a contralateral vascularized fibular bone graft (VFG) and an anatomic distal tibial locking plate. Methods: Between 2017 and 2021, three patients were referred for failed treatment of CPT. All patients had undergone multiple prior surgeries, including tumor excision and fixation with ring external fixators, plates, and screws. We performed radical tumor resection and reconstruction of bone defects with a VFG. The construct was fixed with an anatomic locking plate, and the patients were followed up for a mean of 45.7 months. Results: All three patients were able to obtain graft union at 19.3 weeks. At the final follow-up, all grafts achieved bony hypertrophy without evidence of bone resorption or local tumor recurrence. There was a mean leg length difference of 8.5 cm preoperatively, compared with 6.3 cm postoperatively. The average lower leg angulation was 7.4 degrees and the average ankle range of motion was 58.3 degrees. The mean VAS score was 0 and the mean AOFAS score was 88.3. No significant complications were noted. Conclusions: Implantation of a VFG and an anatomic distal tibia locking plate can be considered an option for treatment-refractory CPT. Patients can expect to achieve bone consolidation, ambulate as tolerated, and have a low complication rate.<\/jats:p>","DOI":"10.3390\/children10030503","type":"journal-article","created":{"date-parts":[[2023,3,3]],"date-time":"2023-03-03T02:56:50Z","timestamp":1677812210000},"page":"503","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Treatment of Refractory Congenital Pseudoarthrosis of Tibia with Contralateral Vascularized Fibular Bone Graft and Anatomic Distal Tibial Locking Plate: A Case Series and Literature Review"],"prefix":"10.3390","volume":"10","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1255-7020","authenticated-orcid":false,"given":"Te-Feng Arthur","family":"Chou","sequence":"first","affiliation":[{"name":"Department of Orthopaedics, Kuang Tien General Hospital, Taichung 433401, Taiwan"},{"name":"Department of Orthopaedics and Traumatology, Taipei Veterans General Hospital, Taipei 112201, Taiwan"},{"name":"Department of Orthopaedic Surgery, Montefiore Medical Center, Albert Einstein College of Medicine, The Bronx, NY 10461, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ting-Yu","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Orthopaedics, Kuang Tien General Hospital, Taichung 433401, Taiwan"},{"name":"Department of Orthopaedics and Traumatology, Taipei Veterans General Hospital, Taipei 112201, Taiwan"},{"name":"Department of Nursing, Hungkuang University, Taichung 433304, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Matthew N.","family":"Wang","sequence":"additional","affiliation":[{"name":"Department of Orthopaedics, Kuang Tien General Hospital, Taichung 433401, Taiwan"},{"name":"Department of Nursing, Hungkuang University, Taichung 433304, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6395-1749","authenticated-orcid":false,"given":"Chen-Yuan","family":"Yang","sequence":"additional","affiliation":[{"name":"Department of Orthopaedics, Kuang Tien General Hospital, Taichung 433401, Taiwan"},{"name":"Department of Nursing, Hungkuang University, Taichung 433304, Taiwan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2023,3,3]]},"reference":[{"key":"ref_1","first-page":"e3","article-title":"Congenital Pseudarthrosis of the Tibia","volume":"5","author":"Foster","year":"2017","journal-title":"JBJS Rev."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"11","DOI":"10.1097\/01202412-200001000-00003","article-title":"Congenital pseudarthrosis of the tibia: History, etiology, classification, and epidemiologic data","volume":"9","author":"Hefti","year":"2000","journal-title":"J. Pediatr. Orthop. B"},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"1027","DOI":"10.1302\/0301-620X.95B8.31434","article-title":"Controversies in the management of congenital pseudarthrosis of the tibia and fibula","volume":"95-B","author":"Khan","year":"2013","journal-title":"Bone Jt. J."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"120","DOI":"10.1302\/1863-2548.13.180147","article-title":"Congenital pseudarthrosis of the tibia: Biological and biomechanical considerations to achieve union and prevent refracture","volume":"13","author":"Paley","year":"2019","journal-title":"J. Child Orthop."},{"key":"ref_5","first-page":"89","article-title":"Congenital pseudarthrosis of the tibia: Treatment with vascularized autogenous fibular grafts. A preliminary report","volume":"147","author":"Weiland","year":"1980","journal-title":"Johns Hopkins Med. J."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"654","DOI":"10.2106\/00004623-199072050-00003","article-title":"Vascularized fibular grafts in the treatment of congenital pseudarthrosis of the tibia","volume":"72","author":"Weiland","year":"1990","journal-title":"J. Bone Jt. Surg. Am."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"90","DOI":"10.1097\/01202412-200004000-00003","article-title":"Free vascular fibular transfer in congenital pseudoarthrosis of the tibia: Results of the EPOS multicenter study. European Paediatric Orthopaedic Society (EPOS)","volume":"9","author":"Romanus","year":"2000","journal-title":"J. Pediatr. Orthop. B"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"13","DOI":"10.1186\/1749-799X-3-13","article-title":"Long-term follow-up on the use of vascularized fibular graft for the treatment of congenital pseudarthrosis of the tibia","volume":"3","author":"Sakamoto","year":"2008","journal-title":"J. Orthop. Surg. Res."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"747","DOI":"10.1016\/j.otsr.2017.05.005","article-title":"Vascularised fibula or induced membrane to treat congenital pseudarthrosis of the Tibia: A multicentre study of 18 patients with a mean 9.5-year follow-up","volume":"103","author":"Vigouroux","year":"2017","journal-title":"Orthop. Traumatol. Surg. Res."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"468","DOI":"10.4103\/0970-0358.105951","article-title":"Experience with free fibula transfer with screw fixation as a primary modality of treatment for congenital pseudarthosis of tibia in children\u2014Series of 26 cases","volume":"45","author":"Kalra","year":"2012","journal-title":"Indian J. Plast. Surg."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"30","DOI":"10.1186\/s13018-018-1037-1","article-title":"Comparison of intramedullary nailing and plate fixation in distal tibial fractures with metaphyseal damage: A meta-analysis of randomized controlled trials","volume":"14","author":"Hu","year":"2019","journal-title":"J. Orthop. Surg. Res."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"34","DOI":"10.1016\/j.jmbbm.2015.10.029","article-title":"Biomechanical evaluation of intramedullary nail and bone plate for the fixation of distal metaphyseal fractures","volume":"56","author":"Nourisa","year":"2016","journal-title":"J. Mech. Behav. Biomed. Mater."},{"key":"ref_13","first-page":"1","article-title":"Neurofibromatosis in children","volume":"218","author":"Crawford","year":"1986","journal-title":"Acta Orthop. Scand. Suppl."},{"key":"ref_14","first-page":"37","article-title":"Congenital pseudarthrosis of the tibia. Long-term followup of 29 cases treated by microvascular bone transfer","volume":"314","author":"Gilbert","year":"1995","journal-title":"Clin. Orthop. Relat. Res."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"736","DOI":"10.1097\/BOT.0b013e318213f709","article-title":"Randomized, prospective comparison of plate versus intramedullary nail fixation for distal tibia shaft fractures","volume":"25","author":"Vallier","year":"2011","journal-title":"J. Orthop. Trauma"},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"1590","DOI":"10.1177\/0363546516631745","article-title":"Plate Versus Intramedullary Nail Fixation of Anterior Tibial Stress Fractures: A Biomechanical Study","volume":"44","author":"Markolf","year":"2016","journal-title":"Am. J. Sports Med."},{"key":"ref_17","first-page":"17","article-title":"Congenital anterior bowing of the tibia. The significance of the different types in relation to pseudarthrosis","volume":"51","author":"Hardinge","year":"1972","journal-title":"Ann. R. Coll. Surg. Engl."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"151","DOI":"10.1016\/j.cjtee.2017.04.001","article-title":"Comparative prospective study between medial and lateral distal tibial locking compression plates for distal third tibial fractures","volume":"20","author":"Garg","year":"2017","journal-title":"Chin. J. Traumatol."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"650","DOI":"10.1177\/1071100717695348","article-title":"Evaluation of Fibular Fracture Type vs Location of Tibial Fixation of Pilon Fractures","volume":"38","author":"Busel","year":"2017","journal-title":"Foot Ankle Int."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"409","DOI":"10.1016\/j.injury.2017.12.015","article-title":"Anterolateral distal tibia locking plate osteosynthesis and their ability to capture OTAC3 pilon fragments","volume":"49","author":"Aneja","year":"2018","journal-title":"Injury"},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"393","DOI":"10.1097\/01.bpo.0000217716.64986.f0","article-title":"Residual deformities following successful initial bone union in congenital pseudoarthrosis of the tibia","volume":"26","author":"Inan","year":"2006","journal-title":"J. Pediatr. Orthop."},{"key":"ref_22","first-page":"346","article-title":"[Reconstruction of the long bones by the induced membrane and spongy autograft]","volume":"45","author":"Masquelet","year":"2000","journal-title":"Ann. Chir. Plast. Esthet."}],"container-title":["Children"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2227-9067\/10\/3\/503\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T18:46:51Z","timestamp":1760122011000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2227-9067\/10\/3\/503"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,3,3]]},"references-count":22,"journal-issue":{"issue":"3","published-online":{"date-parts":[[2023,3]]}},"alternative-id":["children10030503"],"URL":"https:\/\/doi.org\/10.3390\/children10030503","relation":{},"ISSN":["2227-9067"],"issn-type":[{"type":"electronic","value":"2227-9067"}],"subject":[],"published":{"date-parts":[[2023,3,3]]}}}