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Pain, Physical and Social Functioning, and Quality of Life in Individuals with Multiple Hereditary Exostoses in the Netherlands: A National Cohort Study
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 th...
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| Published in: | Journal of bone and joint surgery. American volume 2012-06, Vol.94 (11), p.1013-1020 |
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| container_issue | 11 |
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| container_title | Journal of bone and joint surgery. American volume |
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| creator | Goud, A.L de Lange, J Scholtes, V.A.B Bulstra, S.K Ham, S.J |
| description | 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 |
| doi_str_mv | 10.2106/JBJS.K.00406 |
| format | article |
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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.</description><identifier>ISSN: 0021-9355</identifier><identifier>EISSN: 1535-1386</identifier><identifier>DOI: 10.2106/JBJS.K.00406</identifier><identifier>PMID: 22637207</identifier><identifier>CODEN: JBJSA3</identifier><language>eng</language><publisher>Boston, MA: Copyright by The Journal of Bone and Joint Surgery, Incorporated</publisher><subject>Activities of Daily Living ; Adaptation, Psychological ; Adolescent ; Adult ; Age Factors ; Aged ; Aged, 80 and over ; Biological and medical sciences ; Child ; Child, Preschool ; Chronic Disease ; Cohort Studies ; Diseases of the osteoarticular system ; Exostoses, Multiple Hereditary - diagnosis ; Exostoses, Multiple Hereditary - psychology ; Exostoses, Multiple Hereditary - therapy ; Female ; Humans ; Interpersonal Relations ; Male ; Malformations and congenital and or hereditary diseases involving bones. Joint deformations ; Medical sciences ; Middle Aged ; Miscellaneous. Osteoarticular involvement in other diseases ; Motor Activity - physiology ; Netherlands ; Orthopedic surgery ; Pain, Intractable - diagnosis ; Pain, Intractable - epidemiology ; Pain, Intractable - therapy ; Quality of Life ; Risk Assessment ; Sex Factors ; Sickness Impact Profile ; Stress, Psychological ; Surgery (general aspects). Transplantations, organ and tissue grafts. Graft diseases ; Surveys and Questionnaires ; Young Adult</subject><ispartof>Journal of bone and joint surgery. American volume, 2012-06, Vol.94 (11), p.1013-1020</ispartof><rights>Copyright 2012 by The Journal of Bone and Joint Surgery, Incorporated</rights><rights>2015 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c2386-1d159355ee7d3927c92f69947217aa22a245f89415a5b0036a3d4c927789022e3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=26002958$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/22637207$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Goud, A.L</creatorcontrib><creatorcontrib>de Lange, J</creatorcontrib><creatorcontrib>Scholtes, V.A.B</creatorcontrib><creatorcontrib>Bulstra, S.K</creatorcontrib><creatorcontrib>Ham, S.J</creatorcontrib><title>Pain, Physical and Social Functioning, and Quality of Life in Individuals with Multiple Hereditary Exostoses in the Netherlands: A National Cohort Study</title><title>Journal of bone and joint surgery. American volume</title><addtitle>J Bone Joint Surg Am</addtitle><description>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.</description><subject>Activities of Daily Living</subject><subject>Adaptation, Psychological</subject><subject>Adolescent</subject><subject>Adult</subject><subject>Age Factors</subject><subject>Aged</subject><subject>Aged, 80 and over</subject><subject>Biological and medical sciences</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Chronic Disease</subject><subject>Cohort Studies</subject><subject>Diseases of the osteoarticular system</subject><subject>Exostoses, Multiple Hereditary - diagnosis</subject><subject>Exostoses, Multiple Hereditary - psychology</subject><subject>Exostoses, Multiple Hereditary - therapy</subject><subject>Female</subject><subject>Humans</subject><subject>Interpersonal Relations</subject><subject>Male</subject><subject>Malformations and congenital and or hereditary diseases involving bones. Joint deformations</subject><subject>Medical sciences</subject><subject>Middle Aged</subject><subject>Miscellaneous. Osteoarticular involvement in other diseases</subject><subject>Motor Activity - physiology</subject><subject>Netherlands</subject><subject>Orthopedic surgery</subject><subject>Pain, Intractable - diagnosis</subject><subject>Pain, Intractable - epidemiology</subject><subject>Pain, Intractable - therapy</subject><subject>Quality of Life</subject><subject>Risk Assessment</subject><subject>Sex Factors</subject><subject>Sickness Impact Profile</subject><subject>Stress, Psychological</subject><subject>Surgery (general aspects). Transplantations, organ and tissue grafts. Graft diseases</subject><subject>Surveys and Questionnaires</subject><subject>Young Adult</subject><issn>0021-9355</issn><issn>1535-1386</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2012</creationdate><recordtype>article</recordtype><recordid>eNpFkV-PEyEUxYnRuHX1zWfDi4kPnQqXATq-7Tb7v65rqs8TdoZx0OlQgbHbb-LH9c62akiAXH6cw-UQ8pqzGXCm3l-fXq9mNzPGcqaekAmXQmZczNVTMmEMeFYIKY_Iixi_M2Rypp-TIwAlNDA9Ib_vjOun9K7dRVeZjpq-pitfOdyeD32VnO9d_236WP88mM6lHfUNXbrGUtfTq752v1yNB5FuXWrpx6FLbtNZemmDrV0yYUfPHnxMPto43kitpbcW59ChZvxAT-itGW3QceFbHxJdpaHevSTPGlS1rw7rMfl6fvZlcZktP11cLU6WWQXYZMZrLscOrdW1KEBXBTSqKHINXBsDYCCXzbzIuTTynjGhjKhzhLSeFwzAimPybq-7Cf7nYGMq1y5WtsPXWT_EkjOuFQATDNHpHq2CjzHYptwEt8YOESrHLMoxi_KmfMwC8TcH5eF-bet_8N_PR-DtATAR_74Jpq9c_M8pzK-Qc-TyPbf1XbIh_uiGrQ1la02XWjRDOwUiA8aBKRzZWFLiD3P_oFM</recordid><startdate>20120606</startdate><enddate>20120606</enddate><creator>Goud, A.L</creator><creator>de Lange, J</creator><creator>Scholtes, V.A.B</creator><creator>Bulstra, S.K</creator><creator>Ham, S.J</creator><general>Copyright by The Journal of Bone and Joint Surgery, Incorporated</general><general>Journal of Bone and Joint Surgery Incorporated</general><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope></search><sort><creationdate>20120606</creationdate><title>Pain, Physical and Social Functioning, and Quality of Life in Individuals with Multiple Hereditary Exostoses in the Netherlands: A National Cohort Study</title><author>Goud, A.L ; de Lange, J ; Scholtes, V.A.B ; Bulstra, S.K ; Ham, S.J</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c2386-1d159355ee7d3927c92f69947217aa22a245f89415a5b0036a3d4c927789022e3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2012</creationdate><topic>Activities of Daily Living</topic><topic>Adaptation, Psychological</topic><topic>Adolescent</topic><topic>Adult</topic><topic>Age Factors</topic><topic>Aged</topic><topic>Aged, 80 and over</topic><topic>Biological and medical sciences</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Chronic Disease</topic><topic>Cohort Studies</topic><topic>Diseases of the osteoarticular system</topic><topic>Exostoses, Multiple Hereditary - diagnosis</topic><topic>Exostoses, Multiple Hereditary - psychology</topic><topic>Exostoses, Multiple Hereditary - therapy</topic><topic>Female</topic><topic>Humans</topic><topic>Interpersonal Relations</topic><topic>Male</topic><topic>Malformations and congenital and or hereditary diseases involving bones. Joint deformations</topic><topic>Medical sciences</topic><topic>Middle Aged</topic><topic>Miscellaneous. Osteoarticular involvement in other diseases</topic><topic>Motor Activity - physiology</topic><topic>Netherlands</topic><topic>Orthopedic surgery</topic><topic>Pain, Intractable - diagnosis</topic><topic>Pain, Intractable - epidemiology</topic><topic>Pain, Intractable - therapy</topic><topic>Quality of Life</topic><topic>Risk Assessment</topic><topic>Sex Factors</topic><topic>Sickness Impact Profile</topic><topic>Stress, Psychological</topic><topic>Surgery (general aspects). Transplantations, organ and tissue grafts. Graft diseases</topic><topic>Surveys and Questionnaires</topic><topic>Young Adult</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Goud, A.L</creatorcontrib><creatorcontrib>de Lange, J</creatorcontrib><creatorcontrib>Scholtes, V.A.B</creatorcontrib><creatorcontrib>Bulstra, S.K</creatorcontrib><creatorcontrib>Ham, S.J</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Journal of bone and joint surgery. American volume</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Goud, A.L</au><au>de Lange, J</au><au>Scholtes, V.A.B</au><au>Bulstra, S.K</au><au>Ham, S.J</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Pain, Physical and Social Functioning, and Quality of Life in Individuals with Multiple Hereditary Exostoses in the Netherlands: A National Cohort Study</atitle><jtitle>Journal of bone and joint surgery. American volume</jtitle><addtitle>J Bone Joint Surg Am</addtitle><date>2012-06-06</date><risdate>2012</risdate><volume>94</volume><issue>11</issue><spage>1013</spage><epage>1020</epage><pages>1013-1020</pages><issn>0021-9355</issn><eissn>1535-1386</eissn><coden>JBJSA3</coden><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.</abstract><cop>Boston, MA</cop><pub>Copyright by The Journal of Bone and Joint Surgery, Incorporated</pub><pmid>22637207</pmid><doi>10.2106/JBJS.K.00406</doi><tpages>8</tpages></addata></record> |
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| subjects | Activities of Daily Living Adaptation, Psychological Adolescent Adult Age Factors Aged Aged, 80 and over Biological and medical sciences Child Child, Preschool Chronic Disease Cohort Studies Diseases of the osteoarticular system Exostoses, Multiple Hereditary - diagnosis Exostoses, Multiple Hereditary - psychology Exostoses, Multiple Hereditary - therapy Female Humans Interpersonal Relations Male Malformations and congenital and or hereditary diseases involving bones. Joint deformations Medical sciences Middle Aged Miscellaneous. Osteoarticular involvement in other diseases Motor Activity - physiology Netherlands Orthopedic surgery Pain, Intractable - diagnosis Pain, Intractable - epidemiology Pain, Intractable - therapy Quality of Life Risk Assessment Sex Factors Sickness Impact Profile Stress, Psychological Surgery (general aspects). Transplantations, organ and tissue grafts. Graft diseases Surveys and Questionnaires Young Adult |
| title | Pain, Physical and Social Functioning, and Quality of Life in Individuals with Multiple Hereditary Exostoses in the Netherlands: A National Cohort Study |
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