Bisphosphonate Use and Fractures in Adults with Hypophosphatasia
ABSTRACT Adults with hypophosphatasia (HPP) may suffer femoral fractures resembling the atypical femoral fractures that can occur with long‐term bisphosphonate treatment, and there is an emerging consensus that bisphosphonates should not be used in adults with HPP and low bone mass. However, the spe...
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doaj-15ba7041856a4875a37d6d33958927bc2021-05-02T03:47:16ZengWileyJBMR Plus2473-40392019-10-01310n/an/a10.1002/jbm4.10223Bisphosphonate Use and Fractures in Adults with HypophosphatasiaKate Rassie0Michael Dray1Toshimi Michigami2Tim Cundy3Department of Endocrinology Greenlane Clinical Centre Auckland New ZealandDepartment of Pathology Waikato Hospital Hamilton New ZealandDepartment of Bone & Mineral Research Osaka Women's & Children's Hospital Isumi JapanDepartment of Endocrinology Greenlane Clinical Centre Auckland New ZealandABSTRACT Adults with hypophosphatasia (HPP) may suffer femoral fractures resembling the atypical femoral fractures that can occur with long‐term bisphosphonate treatment, and there is an emerging consensus that bisphosphonates should not be used in adults with HPP and low bone mass. However, the spectrum of HPP in adults is wide: ranging from the severely affected—who commonly have osteomalacia—through to the minimally affected. The former typically have biallelic and the latter, heterozygous ALPL mutations. We have reviewed reports of fractures in adults with genetically proven HPP which suggest that the risk of fracture is at least 200‐fold greater in those with biallelic mutations. We also discuss two cases of postmenopausal women with heterozygous ALPL mutations. One had fractures and severe osteoporosis, but histology revealed no evidence of osteomalacia. The second had taken alendronate for 8 years, but despite profound suppression of bone turnover, histology again revealed no evidence of osteomalacia. The management of adults with HPP who have coexisting osteoporosis is challenging. More data are clearly needed, but we suggest that the risks of bisphosphonate therapy may be relatively low in patients who have heterozygous mutations and no histological evidence of osteomalacia. © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.https://doi.org/10.1002/jbm4.10223ALPLATYPICAL FEMORAL FRACTURESGENE MUTATIONSHYPOPHOSPHATASIAOSTEOMALACIAOSTEOPOROSIS |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Kate Rassie Michael Dray Toshimi Michigami Tim Cundy |
spellingShingle |
Kate Rassie Michael Dray Toshimi Michigami Tim Cundy Bisphosphonate Use and Fractures in Adults with Hypophosphatasia JBMR Plus ALPL ATYPICAL FEMORAL FRACTURES GENE MUTATIONS HYPOPHOSPHATASIA OSTEOMALACIA OSTEOPOROSIS |
author_facet |
Kate Rassie Michael Dray Toshimi Michigami Tim Cundy |
author_sort |
Kate Rassie |
title |
Bisphosphonate Use and Fractures in Adults with Hypophosphatasia |
title_short |
Bisphosphonate Use and Fractures in Adults with Hypophosphatasia |
title_full |
Bisphosphonate Use and Fractures in Adults with Hypophosphatasia |
title_fullStr |
Bisphosphonate Use and Fractures in Adults with Hypophosphatasia |
title_full_unstemmed |
Bisphosphonate Use and Fractures in Adults with Hypophosphatasia |
title_sort |
bisphosphonate use and fractures in adults with hypophosphatasia |
publisher |
Wiley |
series |
JBMR Plus |
issn |
2473-4039 |
publishDate |
2019-10-01 |
description |
ABSTRACT Adults with hypophosphatasia (HPP) may suffer femoral fractures resembling the atypical femoral fractures that can occur with long‐term bisphosphonate treatment, and there is an emerging consensus that bisphosphonates should not be used in adults with HPP and low bone mass. However, the spectrum of HPP in adults is wide: ranging from the severely affected—who commonly have osteomalacia—through to the minimally affected. The former typically have biallelic and the latter, heterozygous ALPL mutations. We have reviewed reports of fractures in adults with genetically proven HPP which suggest that the risk of fracture is at least 200‐fold greater in those with biallelic mutations. We also discuss two cases of postmenopausal women with heterozygous ALPL mutations. One had fractures and severe osteoporosis, but histology revealed no evidence of osteomalacia. The second had taken alendronate for 8 years, but despite profound suppression of bone turnover, histology again revealed no evidence of osteomalacia. The management of adults with HPP who have coexisting osteoporosis is challenging. More data are clearly needed, but we suggest that the risks of bisphosphonate therapy may be relatively low in patients who have heterozygous mutations and no histological evidence of osteomalacia. © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research. |
topic |
ALPL ATYPICAL FEMORAL FRACTURES GENE MUTATIONS HYPOPHOSPHATASIA OSTEOMALACIA OSTEOPOROSIS |
url |
https://doi.org/10.1002/jbm4.10223 |
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AT katerassie bisphosphonateuseandfracturesinadultswithhypophosphatasia AT michaeldray bisphosphonateuseandfracturesinadultswithhypophosphatasia AT toshimimichigami bisphosphonateuseandfracturesinadultswithhypophosphatasia AT timcundy bisphosphonateuseandfracturesinadultswithhypophosphatasia |
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1721495481336463360 |