Risk of fracture in patients with Charcot-Marie-Tooth disease

Sander Pouwels; Anthonius de Boer; Hubert G M Leufkens; Wim E J Weber; Cyrus Cooper; Frank de Vries

doi:10.1002/mus.24240

Risk of fracture in patients with Charcot-Marie-Tooth disease

Sander Pouwels, Anthonius de Boer, Hubert G M Leufkens, Wim E J Weber, Cyrus Cooper, Frank de Vries

Research output: Contribution to journal › Article › Academic › peer-review

Abstract

INTRODUCTION: In this study we evaluated fracture risk in patients with Charcot-Marie-Tooth (CMT) disease.

METHODS: We conducted a retrospective cohort study using the UK Clinical Practice Research Datalink (1987-2012). Each patient with CMT disease was matched with up to 6 patients without a history of CMT disease. The outcome measure was fractures.

RESULTS: The risk of non-osteoporotic fracture was statistically significantly increased [adjusted hazard ratio (AHR) 1.47, 95% confidence interval (CI) 1.01-2.14], whereas risk of any and osteoporotic fracture did not reach statistical significance compared with control patients [AHR 1.31 (95% CI 0.98-1.74) and AHR 1.10 (95% CI 0.69-1.74), respectively].

CONCLUSIONS: CMT patients have a 1.5-fold increased risk for non-osteoporotic fracture. Studies with larger numbers of CMT patients and with additional data on CMT subtype, bone mineral density, and functional status should be performed to confirm a true association between CMT and an increased risk of fracture.

Original language	English
Pages (from-to)	919-24
Number of pages	6
Journal	Muscle & Nerve
Volume	50
Issue number	6
DOIs	https://doi.org/10.1002/mus.24240
Publication status	Published - Dec 2014

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title = "Risk of fracture in patients with Charcot-Marie-Tooth disease",

abstract = "INTRODUCTION: In this study we evaluated fracture risk in patients with Charcot-Marie-Tooth (CMT) disease.METHODS: We conducted a retrospective cohort study using the UK Clinical Practice Research Datalink (1987-2012). Each patient with CMT disease was matched with up to 6 patients without a history of CMT disease. The outcome measure was fractures.RESULTS: The risk of non-osteoporotic fracture was statistically significantly increased [adjusted hazard ratio (AHR) 1.47, 95% confidence interval (CI) 1.01-2.14], whereas risk of any and osteoporotic fracture did not reach statistical significance compared with control patients [AHR 1.31 (95% CI 0.98-1.74) and AHR 1.10 (95% CI 0.69-1.74), respectively].CONCLUSIONS: CMT patients have a 1.5-fold increased risk for non-osteoporotic fracture. Studies with larger numbers of CMT patients and with additional data on CMT subtype, bone mineral density, and functional status should be performed to confirm a true association between CMT and an increased risk of fracture.",

author = "Sander Pouwels and {de Boer}, Anthonius and Leufkens, {Hubert G M} and Weber, {Wim E J} and Cyrus Cooper and {de Vries}, Frank",

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doi = "10.1002/mus.24240",

language = "English",

volume = "50",

pages = "919--24",

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TY - JOUR

T1 - Risk of fracture in patients with Charcot-Marie-Tooth disease

AU - Pouwels, Sander

AU - de Boer, Anthonius

AU - Leufkens, Hubert G M

AU - Weber, Wim E J

AU - Cooper, Cyrus

AU - de Vries, Frank

PY - 2014/12

Y1 - 2014/12

N2 - INTRODUCTION: In this study we evaluated fracture risk in patients with Charcot-Marie-Tooth (CMT) disease.METHODS: We conducted a retrospective cohort study using the UK Clinical Practice Research Datalink (1987-2012). Each patient with CMT disease was matched with up to 6 patients without a history of CMT disease. The outcome measure was fractures.RESULTS: The risk of non-osteoporotic fracture was statistically significantly increased [adjusted hazard ratio (AHR) 1.47, 95% confidence interval (CI) 1.01-2.14], whereas risk of any and osteoporotic fracture did not reach statistical significance compared with control patients [AHR 1.31 (95% CI 0.98-1.74) and AHR 1.10 (95% CI 0.69-1.74), respectively].CONCLUSIONS: CMT patients have a 1.5-fold increased risk for non-osteoporotic fracture. Studies with larger numbers of CMT patients and with additional data on CMT subtype, bone mineral density, and functional status should be performed to confirm a true association between CMT and an increased risk of fracture.

AB - INTRODUCTION: In this study we evaluated fracture risk in patients with Charcot-Marie-Tooth (CMT) disease.METHODS: We conducted a retrospective cohort study using the UK Clinical Practice Research Datalink (1987-2012). Each patient with CMT disease was matched with up to 6 patients without a history of CMT disease. The outcome measure was fractures.RESULTS: The risk of non-osteoporotic fracture was statistically significantly increased [adjusted hazard ratio (AHR) 1.47, 95% confidence interval (CI) 1.01-2.14], whereas risk of any and osteoporotic fracture did not reach statistical significance compared with control patients [AHR 1.31 (95% CI 0.98-1.74) and AHR 1.10 (95% CI 0.69-1.74), respectively].CONCLUSIONS: CMT patients have a 1.5-fold increased risk for non-osteoporotic fracture. Studies with larger numbers of CMT patients and with additional data on CMT subtype, bone mineral density, and functional status should be performed to confirm a true association between CMT and an increased risk of fracture.

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DO - 10.1002/mus.24240

M3 - Article

C2 - 24634316

SN - 0148-639X

VL - 50

SP - 919

EP - 924

JO - Muscle & Nerve

JF - Muscle & Nerve

IS - 6

ER -

Risk of fracture in patients with Charcot-Marie-Tooth disease

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