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Surgical Outcome and Oncological Survival of Osteofibrous Dysplasia-Like and Classic Adamantinomas: An International Multicenter Study of 318 Cases.
Schutgens, E M; Picci, P; Baumhoer, D; Pollock, R; Bovée, J V M G; Hogendoorn, P C W; Dijkstra, P D S; Rueten-Budde, A J; Jutte, P C; Traub, F; Leithner, A; Tunn, P-U; Funovics, P; Sys, G; San-Julian, M; Schaap, G R; Dürr, H R; Hardes, J; Healey, J; Capanna, R; Biau, D; Gomez-Brouchet, A; Wunder, J; Cosker, T D A; Laitinen, M K; Niu, X; Kostiuk, V; van de Sande, M A J.
Afiliación
  • Schutgens EM; Departments of Orthopedic Surgery (E.M.S., P.D.S.D., and M.A.J.v.d.S.), Histopathology (J.V.M.G.B.), and Pathology (P.C.W.H.), Leiden University Medical Center, Leiden, the Netherlands.
  • Picci P; London Sarcoma Service, Royal National Orthopaedic Hospital, Stanmore, United Kingdom.
  • Baumhoer D; Medical Oncology, Musculoskeletal Oncology Department, IRCCS Istituto Ortopedico Rizzoli, Bologna, Italy.
  • Pollock R; Bone Tumour Reference Centre, Institute of Pathology, University Hospital and University of Basel, Basel, Switzerland.
  • Bovée JVMG; London Sarcoma Service, Royal National Orthopaedic Hospital, Stanmore, United Kingdom.
  • Hogendoorn PCW; Departments of Orthopedic Surgery (E.M.S., P.D.S.D., and M.A.J.v.d.S.), Histopathology (J.V.M.G.B.), and Pathology (P.C.W.H.), Leiden University Medical Center, Leiden, the Netherlands.
  • Dijkstra PDS; Departments of Orthopedic Surgery (E.M.S., P.D.S.D., and M.A.J.v.d.S.), Histopathology (J.V.M.G.B.), and Pathology (P.C.W.H.), Leiden University Medical Center, Leiden, the Netherlands.
  • Rueten-Budde AJ; Departments of Orthopedic Surgery (E.M.S., P.D.S.D., and M.A.J.v.d.S.), Histopathology (J.V.M.G.B.), and Pathology (P.C.W.H.), Leiden University Medical Center, Leiden, the Netherlands.
  • Jutte PC; Mathematical Institute, Leiden University, Leiden, the Netherlands.
  • Traub F; Department of Orthopedics, University Medical Center Groningen, University of Groningen, Groningen, the Netherlands.
  • Leithner A; Orthopedic Surgery, University of Tübingen, Tübingen, Germany.
  • Tunn PU; Department of Orthopedics and Trauma, Medical University of Graz, Graz, Austria.
  • Funovics P; Orthopedic Surgery, Helios-Clinics, Berlin, Germany.
  • Sys G; Orthopedic Surgery, Medical University of Vienna, Vienna, Austria.
  • San-Julian M; Orthopedic Surgery, Ghent University Hospital, Ghent, Belgium.
  • Schaap GR; Orthopedic Surgery, University of Navarra, Pamplona, Spain.
  • Dürr HR; Orthopedic Surgery, Academic Medical Center, Amsterdam, the Netherlands.
  • Hardes J; Department of Tumor Orthopedics and Sarcoma Surgery, University Hospital Essen, Essen, Germany.
  • Healey J; Musculoskeletal Oncology, Department of Orthopedic Surgery, Physical Medicine and Rehabilitation, University Hospital, LMU Munich, Munich, Germany.
  • Capanna R; Orthopedic Surgery, Memorial Sloan Kettering Cancer Center, New York, NY.
  • Biau D; Department of Orthopaedics, S. Chiara University Hospital, University of Pisa, Italy.
  • Gomez-Brouchet A; Orthopedic Surgery, Cochin Hospital, Paris, France.
  • Wunder J; Department of Histopathology, University Medical Center, Toulouse, France.
  • Cosker TDA; University Musculoskeletal Oncology Unit, Mount Sinai Hospital, Toronto, Ontario, Canada.
  • Laitinen MK; Orthopedic Surgery, Nuffield Orthopedic Center, Oxford, United Kingdom.
  • Niu X; Orthopedic Surgery, Helsinki University Hospital, Helsinki, Finland.
  • Kostiuk V; Department of Orthopedic Oncology, Beijing Jishuitan Hospital, Beijing, People's Republic of China.
  • van de Sande MAJ; Orthopedic Surgery, National Cancer Institute Ukraine, Kiev, Ukraine.
J Bone Joint Surg Am ; 102(19): 1703-1713, 2020 10 07.
Article en En | MEDLINE | ID: mdl-33027124
BACKGROUND: Osteofibrous dysplasia-like adamantinoma (OFD-AD) and classic adamantinoma (AD) are rare, neoplastic diseases with only limited data supporting current treatment protocols. We believe that our retrospective multicenter cohort study is the largest analysis of patients with adamantinoma to date. The primary purpose of this study was to describe the disease characteristics and evaluate the oncological outcomes. The secondary purpose was to identify risk factors for local recurrence after surgical treatment and propose treatment guidelines. METHODS: Three hundred and eighteen confirmed cases of OFD-AD and AD for which primary treatment was carried out between 1985 and 2015 were submitted by 22 tertiary bone tumor centers. Proposed clinical risk factors for local recurrence such as size, type, and margins were analyzed using univariable and multivariate Cox regression analysis. RESULTS: Of the 318 cases, 128 were OFD-AD and 190 were AD. The mean age at diagnosis was 17 years (median, 14.5 years) for OFD-AD and 32 years (median, 28 years) for AD; 53% of the patients were female. The mean tumor size in the OFD-AD and AD groups combined was 7.8 cm, measured histologically. Sixteen percent of the patients sustained a pathological fracture prior to treatment. Local recurrence was recorded in 22% of the OFD-AD cases and 24% of the AD cases. None of the recurrences in the OFD-AD group progressed to AD. Metastatic disease was found in 18% of the AD cases and fatal disease, in 11% of the AD cases. No metastatic or fatal disease was reported in the OFD-AD group. Multivariate Cox regression analysis demonstrated that uncontaminated resection margins (hazard ratio [HR] = 0.164, 95% confidence interval [CI] = 0.092 to 0.290, p < 0.001), pathological fracture (HR = 1.968, 95% CI = 1.076 to 3.600, p = 0.028), and sex (female versus male: HR = 0.535, 95% CI = 0.300 to 0.952, p = 0.033) impacted the risk of local recurrence. CONCLUSIONS: OFD-AD and AD are parts of a disease spectrum but should be regarded as different entities. Our results support reclassification of OFD-AD into the intermediate locally aggressive category, based on the local recurrence rate of 22% and absence of metastases. In our study, metastatic disease was restricted to the AD group (an 18% rate). We advocate wide resection with uncontaminated margins including bone and involved periosteum for both OFD-AD and AD. LEVEL OF EVIDENCE: Prognostic Level IV. See Instructions for Authors for a complete description of levels of evidence.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Enfermedades del Desarrollo Óseo / Neoplasias Óseas / Adamantinoma Tipo de estudio: Clinical_trials / Etiology_studies / Guideline / Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Adolescent / Adult / Female / Humans / Male Idioma: En Revista: J Bone Joint Surg Am Año: 2020 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Enfermedades del Desarrollo Óseo / Neoplasias Óseas / Adamantinoma Tipo de estudio: Clinical_trials / Etiology_studies / Guideline / Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Adolescent / Adult / Female / Humans / Male Idioma: En Revista: J Bone Joint Surg Am Año: 2020 Tipo del documento: Article País de afiliación: Países Bajos Pais de publicación: Estados Unidos