Cargando…
Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases
Tumor-induced osteomalacia (TIO) is a rare and largely underdiagnosed paraneoplastic condition. Previous reviews often reported incomplete data on clinical aspects, diagnosis or prognosis. The aim of this study was to present a systematic clinical review of all published cases of TIO. A search was c...
Autores principales: | , , , , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Springer US
2022
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9474374/ https://www.ncbi.nlm.nih.gov/pubmed/35857061 http://dx.doi.org/10.1007/s00223-022-01005-8 |
_version_ | 1784789702842253312 |
---|---|
author | Bosman, Ariadne Palermo, Andrea Vanderhulst, Julien De Beur, Suzanne M. Jan Fukumoto, Seiji Minisola, Salvatore Xia, Weibo Body, Jean-Jacques Zillikens, M. Carola |
author_facet | Bosman, Ariadne Palermo, Andrea Vanderhulst, Julien De Beur, Suzanne M. Jan Fukumoto, Seiji Minisola, Salvatore Xia, Weibo Body, Jean-Jacques Zillikens, M. Carola |
author_sort | Bosman, Ariadne |
collection | PubMed |
description | Tumor-induced osteomalacia (TIO) is a rare and largely underdiagnosed paraneoplastic condition. Previous reviews often reported incomplete data on clinical aspects, diagnosis or prognosis. The aim of this study was to present a systematic clinical review of all published cases of TIO. A search was conducted in Pubmed, Embase, Web of Science from inception until April 23rd, 2020. We selected case reports and case series of patients diagnosed with TIO, with information on tumor localization and serum phosphate concentration. Two reviewers independently extracted data on biochemical and clinical characteristics including bone involvement, tumor localization and treatment. 468 articles with 895 unique TIO cases were included. Median age was 46 years (range 9 months–90 years) and 58.3% were males. Hypophosphatemia and inappropriately low or normal 1,25-dihydroxyvitamin D levels, characteristic for TIO, were present in 98% of cases. Median tumor size was 2.7 cm (range 0.5 to 25.0 cm). Serum fibroblast growth factor 23 was related to tumor size (r = 0.344, P < 0.001). In 32% of the cases the tumor was detected by physical examination. Data on bone phenotype confirmed skeletal involvement: 62% of cases with BMD data had a T-score of the lumbar spine ≤ − 2.5 (n = 61/99) and a fracture was reported in at least 39% of all cases (n = 346/895). Diagnostic delay was longer than 2 years in more than 80% of cases. 10% were reported to be malignant at histology. In conclusion, TIO is a debilitating disease characterized by a long diagnostic delay leading to metabolic disturbances and skeletal impairment. Increasing awareness of TIO should decrease its diagnostic delay and the clinical consequences. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s00223-022-01005-8. |
format | Online Article Text |
id | pubmed-9474374 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Springer US |
record_format | MEDLINE/PubMed |
spelling | pubmed-94743742022-09-16 Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases Bosman, Ariadne Palermo, Andrea Vanderhulst, Julien De Beur, Suzanne M. Jan Fukumoto, Seiji Minisola, Salvatore Xia, Weibo Body, Jean-Jacques Zillikens, M. Carola Calcif Tissue Int Review Article Tumor-induced osteomalacia (TIO) is a rare and largely underdiagnosed paraneoplastic condition. Previous reviews often reported incomplete data on clinical aspects, diagnosis or prognosis. The aim of this study was to present a systematic clinical review of all published cases of TIO. A search was conducted in Pubmed, Embase, Web of Science from inception until April 23rd, 2020. We selected case reports and case series of patients diagnosed with TIO, with information on tumor localization and serum phosphate concentration. Two reviewers independently extracted data on biochemical and clinical characteristics including bone involvement, tumor localization and treatment. 468 articles with 895 unique TIO cases were included. Median age was 46 years (range 9 months–90 years) and 58.3% were males. Hypophosphatemia and inappropriately low or normal 1,25-dihydroxyvitamin D levels, characteristic for TIO, were present in 98% of cases. Median tumor size was 2.7 cm (range 0.5 to 25.0 cm). Serum fibroblast growth factor 23 was related to tumor size (r = 0.344, P < 0.001). In 32% of the cases the tumor was detected by physical examination. Data on bone phenotype confirmed skeletal involvement: 62% of cases with BMD data had a T-score of the lumbar spine ≤ − 2.5 (n = 61/99) and a fracture was reported in at least 39% of all cases (n = 346/895). Diagnostic delay was longer than 2 years in more than 80% of cases. 10% were reported to be malignant at histology. In conclusion, TIO is a debilitating disease characterized by a long diagnostic delay leading to metabolic disturbances and skeletal impairment. Increasing awareness of TIO should decrease its diagnostic delay and the clinical consequences. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s00223-022-01005-8. Springer US 2022-07-20 2022 /pmc/articles/PMC9474374/ /pubmed/35857061 http://dx.doi.org/10.1007/s00223-022-01005-8 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . |
spellingShingle | Review Article Bosman, Ariadne Palermo, Andrea Vanderhulst, Julien De Beur, Suzanne M. Jan Fukumoto, Seiji Minisola, Salvatore Xia, Weibo Body, Jean-Jacques Zillikens, M. Carola Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases |
title | Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases |
title_full | Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases |
title_fullStr | Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases |
title_full_unstemmed | Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases |
title_short | Tumor-Induced Osteomalacia: A Systematic Clinical Review of 895 Cases |
title_sort | tumor-induced osteomalacia: a systematic clinical review of 895 cases |
topic | Review Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9474374/ https://www.ncbi.nlm.nih.gov/pubmed/35857061 http://dx.doi.org/10.1007/s00223-022-01005-8 |
work_keys_str_mv | AT bosmanariadne tumorinducedosteomalaciaasystematicclinicalreviewof895cases AT palermoandrea tumorinducedosteomalaciaasystematicclinicalreviewof895cases AT vanderhulstjulien tumorinducedosteomalaciaasystematicclinicalreviewof895cases AT debeursuzannemjan tumorinducedosteomalaciaasystematicclinicalreviewof895cases AT fukumotoseiji tumorinducedosteomalaciaasystematicclinicalreviewof895cases AT minisolasalvatore tumorinducedosteomalaciaasystematicclinicalreviewof895cases AT xiaweibo tumorinducedosteomalaciaasystematicclinicalreviewof895cases AT bodyjeanjacques tumorinducedosteomalaciaasystematicclinicalreviewof895cases AT zillikensmcarola tumorinducedosteomalaciaasystematicclinicalreviewof895cases |