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Pain, Physical and Social Functioning, and Quality of Life in Individuals with Multiple Hereditary Exostoses in the NetherlandsA National Cohort Study
A.L. Goud, MD1; J. de Lange, MSc2; V.A.B. Scholtes, PhD1; S.K. Bulstra, MD, PhD3; S.J. Ham, MD, PhD1
1 Department of Orthopedic Surgery, Onze Lieve Vrouwe Gasthuis, P.O. Box 95500, 1090 HM Amsterdam, The Netherlands. E-mail address for A.L. Goud: annemariegoud@hotmail.com
2 MHE-MO Patient Coalition, P. O. Box 95500, 1090 HM, Amsterdam, The Netherlands
3 Department of Orthopedic Surgery, University Hospital, Groningen, P.O. Box 30.001, 9700 RB, Groningen, The Netherlands
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Investigation performed at the Department of Orthopedic Surgery, Onze Lieve Vrouwe Gasthuis, Amsterdam, and the Department of Orthopedic Surgery, University Hospital, Groningen, The Netherlands

A commentary by Alexandre Arkader, MD, is linked to the online version of this article at jbjs.org.



Disclosure: None of the authors received payments or services, either directly or indirectly (i.e., via his or her institution), from a third party in support of any aspect of this work. None of the authors, or their institution(s), have had any financial relationship, in the thirty-six months prior to submission of this work, with any entity in the biomedical arena that could be perceived to influence or have the potential to influence what is written in this work. Also, no author has had any other relationships, or has engaged in any other activities, that could be perceived to influence or have the potential to influence what is written in this work. The complete Disclosures of Potential Conflicts of Interest submitted by authors are always provided with the online version of the article.

Copyright © 2012 by The Journal of Bone and Joint Surgery, Inc.
J Bone Joint Surg Am, 2012 Jun 06;94(11):1013-1020. doi: 10.2106/JBJS.K.00406
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Abstract

Background: 

This study aimed to assess pain and quality of life in a large cohort of patients with multiple hereditary exostoses.

Methods: 

All 322 known patients with multiple hereditary exostoses in the Netherlands were asked to participate. An age-specific questionnaire was sent to children (less than eighteen years old) and adults. The questionnaire focused on pain, daily activities, and school and/or professional situation. Adults also filled out the RAND-36 questionnaire. Results were statistically analyzed with use of the SPSS 15.0 software and with the chi-square test and multiple logistic regression. A p value of <0.05 was regarded as significant.

Results: 

Two hundred and eighty-three patients (88%), including 184 adults (65%) and ninety-nine children (35%), completed the questionnaire. Multiple hereditary exostoses resulted in various physical and social consequences. The majority of adults (119) were employed; however, thirty-three (28%) had changed jobs because of the symptoms of multiple hereditary exostoses and twenty-five (21%) required adjustments in their working environment. Of the sixty-five adults who were not employed, thirteen were medically unfit to work. Of eighty-five children attending school, forty-five (53%) experienced problems at school. The symptoms of multiple hereditary exostoses caused twenty-seven children (27%) and eighty-five adults (46%) to stop participating in sporting activities. Pain was the greatest problem, with sixty-two children (63%) and 152 adults (83%) who reported recent pain. On multivariate analysis, pain in adults was correlated most significantly with age and problems at work, and pain in children was correlated with the perception of the disease and problems at school. Adult patients with multiple hereditary exostoses had a lower quality of life than the Dutch reference groups, with lower scores on six of eight RAND-36 subscales.

Conclusions: 

Our study confirms that multiple hereditary exostoses is a chronic disease causing a profound impact on quality of life. The results suggest that pain is not the only problem associated with multiple hereditary exostoses, as it has an extensive influence on daily activities, as well as on social and psychological well-being, causing significant disability.

Level of Evidence: 

Prognostic Level IV. See Instructions for Authors for a complete description of levels of evidence.

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    References

    Accreditation Statement
    These activities have been planned and implemented in accordance with the Essential Areas and policies of the Accreditation Council for Continuing Medical Education (ACCME) through the joint sponsorship of the American Academy of Orthopaedic Surgeons and The Journal of Bone and Joint Surgery, Inc. The American Academy of Orthopaedic Surgeons is accredited by the ACCME to provide continuing medical education for physicians.
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