{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,15]],"date-time":"2026-09-15T19:03:21Z","timestamp":1789499001991,"version":"build-2803163510"},"reference-count":27,"publisher":"Ovid Technologies (Wolters Kluwer Health)","content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"abstract":"\n Background:<\/jats:title>\n Existing classifications for forearm deformity in multiple hereditary exostoses (MHE) have demonstrated low interrater reliability and limited clinical applicability. Our primary research question was whether a Delphi consensus process could generate a more reliable and clinically useful classification for longitudinal surveillance of MHE-related forearm deformities.<\/jats:p>\n <\/jats:sec>\n \n Methods:<\/jats:title>\n Fifteen pediatric hand surgeons from the Congenital Upper Limb Difference (CoULD) Study Group participated in 7 rounds of online Delphi surveys (June 2022 to September 2023). An investigator team facilitated the process with anonymized feedback. Round 1 identified key clinical and radiographic features. Subsequent rounds refined these features, determined categorical structure, and piloted the system with representative radiographs. Consensus was defined as \u226570% agreement. After final consensus, interrater reliability was tested by 4 independent surgeons who classified 101 radiographs from 67 patients with forearm MHE. Reliability was assessed with kappa and concordance coefficients and compared with the Masada, Gottschalk, and Jo classifications.<\/jats:p>\n <\/jats:sec>\n \n Results:<\/jats:title>\n The Delphi process identified radiographic predictors that included ulnar variance, the radial articular angle (RAA), radial head alignment, and osteochondroma size. Consensus favored a 3-tier classification: Type 1, mild; Type 2, moderate; and Type 3, severe. Closing consensus was achieved, with 93% agreement. Reliability testing showed moderate agreement on the first reading (kappa = 0.55), significantly higher than for the Masada (kappa = 0.21 [original classification], kappa = 0.35 [modified version]), Gottschalk (kappa = 0.43), and Jo (kappa = 0.43) classifications. After refinement that incorporated osteochondroma size and the radial head subtypes (3A, subluxation, and 3B, dislocation), agreement improved to substantial (kappa = 0.64) (p < 0.001), with high concordance (0.82).<\/jats:p>\n <\/jats:sec>\n \n Conclusions:<\/jats:title>\n The CoULD MHE Classification, developed through expert consensus, integrates validated radiographic predictors with defined thresholds. It demonstrated significantly improved reliability over prior systems and provides a practical framework for longitudinal monitoring of forearm deformities in patients with MHE.<\/jats:p>\n <\/jats:sec>\n \n Level of Evidence:<\/jats:title>\n \n Diagnostic\n Level V<\/jats:underline>\n . See Instructions for Authors for a complete description of levels of evidence.\n <\/jats:p>\n <\/jats:sec>","DOI":"10.2106\/jbjs.25.01593","type":"journal-article","created":{"date-parts":[[2026,9,15]],"date-time":"2026-09-15T18:08:36Z","timestamp":1789495716000},"update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":0,"title":["Classification of Forearm Involvement in Multiple Hereditary Exostoses"],"prefix":"10.2106","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7390-384X","authenticated-orcid":false,"given":"Carley","family":"Vuillermin","sequence":"first","affiliation":[{"name":"Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6718-3580","authenticated-orcid":false,"given":"Maria F.","family":"Canizares","sequence":"additional","affiliation":[{"name":"Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0141-717X","authenticated-orcid":false,"given":"Patricia E.","family":"Miller","sequence":"additional","affiliation":[{"name":"Boston Children\u2019s Hospital, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lindley B.","family":"Wall","sequence":"additional","affiliation":[{"name":"St. Louis Children\u2019s Hospital, Washington University School of Medicine, St. Louis, Missouri"},{"name":"Shriners Hospitals for Children\u2013St. Louis, St. Louis, Missouri"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1663-1777","authenticated-orcid":false,"given":"Suzanne E.","family":"Steinman","sequence":"additional","affiliation":[{"name":"Seattle Children\u2019s Hospital, Seattle, Washington"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8064-5508","authenticated-orcid":false,"given":"Ann","family":"Van Heest","sequence":"additional","affiliation":[{"name":"Gillette Children\u2019s Specialty Healthcare, St. Paul, Minnesota"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"on behalf of the CoULD Study Group","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2026,9,15]]},"reference":[{"key":"bib1-20260915","doi-asserted-by":"crossref","first-page":"700","DOI":"10.1302\/0301-620X.43B4.700","article-title":"Bone growth in diaphysial aclasis","volume":"43-B","author":"Solomon","year":"1961","journal-title":"J Bone Joint Surg Br."},{"issue":"6A","key":"bib2-20260915","doi-asserted-by":"crossref","first-page":"815","DOI":"10.2106\/00004623-197961060-00003","article-title":"Hereditary multiple exostoses. Anthropometric, roentgenographic, and clinical aspects","volume":"61","author":"Shapiro","year":"1979","journal-title":"J Bone Joint Surg Am."},{"issue":"7","key":"bib3-20260915","doi-asserted-by":"crossref","first-page":"986","DOI":"10.2106\/00004623-199407000-00005","article-title":"The natural history of hereditary multiple exostoses","volume":"76","author":"Schmale","year":"1994","journal-title":"J Bone Joint Surg Am."},{"issue":"6","key":"bib4-20260915","first-page":"748","article-title":"Manifestations and management of osteochondromas: a retrospective analysis of 382 patients","volume":"72","author":"Saglik","year":"2006","journal-title":"Acta Orthop Belg."},{"issue":"11","key":"bib5-20260915","doi-asserted-by":"crossref","first-page":"2286","DOI":"10.1016\/j.jhsa.2012.07.033","article-title":"Effect of osteochondroma location on forearm deformity in patients with multiple hereditary osteochondromatosis","volume":"37","author":"Gottschalk","year":"2012","journal-title":"J Hand Surg Am."},{"issue":"17","key":"bib6-20260915","doi-asserted-by":"crossref","first-page":"1586","DOI":"10.2106\/JBJS.L.00736","article-title":"Forearm deformity in patients with hereditary multiple exostoses: factors associated with range of motion and radial head dislocation","volume":"95","author":"Clement","year":"2013","journal-title":"J Bone Joint Surg Am."},{"issue":"1","key":"bib7-20260915","doi-asserted-by":"crossref","first-page":"24","DOI":"10.1302\/0301-620X.71B1.2914999","article-title":"Operations for forearm deformity caused by multiple osteochondromas","volume":"71","author":"Masada","year":"1989","journal-title":"J Bone Joint Surg Br."},{"issue":"4","key":"bib8-20260915","doi-asserted-by":"crossref","first-page":"292.e1","DOI":"10.1016\/j.jhsa.2017.01.010","article-title":"An Evaluation of Forearm Deformities in Hereditary Multiple Exostoses: Factors Associated With Radial Head Dislocation and Comprehensive Classification","volume":"42","author":"Jo","year":"2017","journal-title":"J Hand Surg Am."},{"issue":"2","key":"bib9-20260915","doi-asserted-by":"crossref","first-page":"110","DOI":"10.5435\/00124635-200503000-00004","article-title":"Manifestations of hereditary multiple exostoses","volume":"13","author":"Stieber","year":"2005","journal-title":"J Am Acad Orthop Surg."},{"issue":"1","key":"bib10-20260915","doi-asserted-by":"crossref","first-page":"43","DOI":"10.1002\/ajmg.1320550113","article-title":"Natural history study of hereditary multiple exostoses","volume":"55","author":"Wicklund","year":"1995","journal-title":"Am J Med Genet."},{"issue":"3","key":"bib11-20260915","doi-asserted-by":"crossref","first-page":"397","DOI":"10.2106\/00004623-200203000-00010","article-title":"Evaluation of the forearm in untreated adult subjects with multiple hereditary osteochondromatosis","volume":"84","author":"Noonan","year":"2002","journal-title":"J Bone Joint Surg Am."},{"issue":"3","key":"bib12-20260915","doi-asserted-by":"crossref","first-page":"169","DOI":"10.1007\/s11751-016-0267-1","article-title":"Multiple osteochondromas (MO) in the forearm: a 12-year single-centre experience","volume":"11","author":"Ham","year":"2016","journal-title":"Strategies Trauma Limb Reconstr."},{"issue":"1","key":"bib13-20260915","doi-asserted-by":"crossref","first-page":"e60","DOI":"10.1097\/BPO.0000000000002287","article-title":"Reliability of the Masada Classification for Forearm Involvement in Patients With Hereditary Multiple Osteochondromas (HMO)","volume":"43","author":"Canizares","year":"2023","journal-title":"J Pediatr Orthop."},{"issue":"8","key":"bib14-20260915","doi-asserted-by":"crossref","first-page":"1006.e1","DOI":"10.1016\/j.jhsa.2024.07.010","article-title":"A New Classification System for Forearm Deformities Caused by Hereditary Multiple Osteochondromas","volume":"50","author":"Chan","year":"2025","journal-title":"J Hand Surg Am."},{"issue":"4","key":"bib15-20260915","doi-asserted-by":"crossref","first-page":"438","DOI":"10.1007\/s11552-012-9440-2","article-title":"Carpal tunnel syndrome caused by a large osteochondroma","volume":"7","author":"Wong","year":"2012","journal-title":"Hand (N Y)."},{"issue":"1","key":"bib16-20260915","doi-asserted-by":"crossref","first-page":"92","DOI":"10.1097\/01241398-199401000-00019","article-title":"Deformities and problems of the forearm in children with multiple hereditary osteochondromata","volume":"14","author":"Peterson","year":"1994","journal-title":"J Pediatr Orthop."},{"issue":"1","key":"bib17-20260915","doi-asserted-by":"crossref","first-page":"69","DOI":"10.1097\/BPO.0000000000000732","article-title":"Intraobserver and Interobserver Reliability of the Oberg-Manske-Tonkin (OMT) Classification: Establishing a Registry on Congenital Upper Limb Differences","volume":"38","author":"Bae","year":"2018","journal-title":"J Pediatr Orthop."},{"issue":"12","key":"bib18-20260915","doi-asserted-by":"crossref","first-page":"2066","DOI":"10.1016\/j.jhsa.2010.09.031","article-title":"Developmental biology and classification of congenital anomalies of the hand and upper extremity","volume":"35","author":"Oberg","year":"2010","journal-title":"J Hand Surg Am."},{"issue":"7","key":"bib19-20260915","doi-asserted-by":"crossref","first-page":"e32829","DOI":"10.1097\/MD.0000000000032829","article-title":"Use of Delphi in health sciences research: A narrative review","volume":"102","author":"Shang","year":"2023","journal-title":"Medicine (Baltimore)."},{"issue":"4","key":"bib20-20260915","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1016\/j.jclinepi.2013.12.002","article-title":"Defining consensus: a systematic review recommends methodologic criteria for reporting of Delphi studies","volume":"67","author":"Diamond","year":"2014","journal-title":"J Clin Epidemiol."},{"issue":"1","key":"bib21-20260915","doi-asserted-by":"crossref","first-page":"13","DOI":"10.2106\/00004623-199301000-00003","article-title":"Deformities of the forearm in patients who have multiple cartilaginous exostosis","volume":"75","author":"Burgess","year":"1993","journal-title":"J Bone Joint Surg Am."},{"issue":"1","key":"bib23-20260915","doi-asserted-by":"crossref","first-page":"159","DOI":"10.2307\/2529310","article-title":"The measurement of observer agreement for categorical data","volume":"33","author":"Landis","year":"1977","journal-title":"Biometrics."},{"issue":"4","key":"bib24-20260915","doi-asserted-by":"crossref","first-page":"568","DOI":"10.2106\/00004623-199604000-00010","article-title":"Function of the upper extremities in hereditary multiple exostoses","volume":"78","author":"Stanton","year":"1996","journal-title":"J Bone Joint Surg Am."},{"issue":"7","key":"bib25-20260915","doi-asserted-by":"crossref","first-page":"e656","DOI":"10.1097\/BPO.0000000000001515","article-title":"Predicting Radial Head Instability in Multiple Hereditary Exostoses (MHE): A Multicenter Analysis of Risk Factors","volume":"40","author":"Meza","year":"2020","journal-title":"J Pediatr Orthop."},{"issue":"5","key":"bib26-20260915","doi-asserted-by":"crossref","first-page":"103445","DOI":"10.1016\/j.otsr.2022.103445","article-title":"Forearm deformity in multiple hereditary exostosis. Radiologic predictors of radial head dislocation","volume":"109","author":"Bom","year":"2023","journal-title":"Orthop Traumatol Surg Res."},{"issue":"2","key":"bib27-20260915","doi-asserted-by":"crossref","first-page":"246","DOI":"10.1097\/01241398-200003000-00022","article-title":"Clinical and radiographic analysis of osteochondromas and growth disturbance in hereditary multiple exostoses","volume":"20","author":"Porter","year":"2000","journal-title":"J Pediatr Orthop."},{"issue":"8","key":"bib28-20260915","doi-asserted-by":"crossref","first-page":"427","DOI":"10.1007\/s002560050412","article-title":"MR imaging of symptomatic osteochondromas with pathological correlation","volume":"27","author":"Mehta","year":"1998","journal-title":"Skeletal Radiol."}],"container-title":["Journal of Bone and Joint Surgery"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.2106\/JBJS.25.01593","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,15]],"date-time":"2026-09-15T18:08:41Z","timestamp":1789495721000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.2106\/JBJS.25.01593"}},"subtitle":["Consensus from the CoULD Study Group"],"short-title":[],"issued":{"date-parts":[[2026,9,15]]},"references-count":27,"URL":"https:\/\/doi.org\/10.2106\/jbjs.25.01593","relation":{},"ISSN":["0021-9355","1535-1386"],"issn-type":[{"value":"0021-9355","type":"print"},{"value":"1535-1386","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,9,15]]}}}