Bone Health Determinants in Ambulant Prepubertal Boys With Duchenne Muscular Dystrophy Treated With Deflazacort: Findings From a 3‐Year Study
ABSTRACT Introduction/Aims Duchenne muscular dystrophy (DMD) is complicated by bone fragility. This study aimed to elucidate changes in bone mineral density (BMD) and body composition over time and to explore associations with adiposity measures in DMD. Methods A three‐year follow‐up analysis was pe...
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| creator | Panicucci, Chiara Casalini, Sara Angelelli, Alessia Brolatti, Noemi Pedemonte, Marina Patti, Giuseppa Maghnie, Mohamad Bruno, Claudio Di Iorgi, Natascia |
| description | ABSTRACT
Introduction/Aims
Duchenne muscular dystrophy (DMD) is complicated by bone fragility. This study aimed to elucidate changes in bone mineral density (BMD) and body composition over time and to explore associations with adiposity measures in DMD.
Methods
A three‐year follow‐up analysis was performed of total body (TB) and lumbar spine (LS) dual‐energy x‐ray absorptiometry (DXA) measurements, anthropometric measures, Tanner stage and bone turnover biomarkers assessments, and the incidence of fragility fractures in 26 ambulant prepubertal DMD patients treated with deflazacort (DFZ).
Results
Age at baseline was 7.7 years (interquartile range: 6–9.2). The TB BMD Z‐score declined over time and was negatively related to the TB fat mass percentage and fat mass index (p |
| doi_str_mv | 10.1002/mus.28309 |
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Introduction/Aims
Duchenne muscular dystrophy (DMD) is complicated by bone fragility. This study aimed to elucidate changes in bone mineral density (BMD) and body composition over time and to explore associations with adiposity measures in DMD.
Methods
A three‐year follow‐up analysis was performed of total body (TB) and lumbar spine (LS) dual‐energy x‐ray absorptiometry (DXA) measurements, anthropometric measures, Tanner stage and bone turnover biomarkers assessments, and the incidence of fragility fractures in 26 ambulant prepubertal DMD patients treated with deflazacort (DFZ).
Results
Age at baseline was 7.7 years (interquartile range: 6–9.2). The TB BMD Z‐score declined over time and was negatively related to the TB fat mass percentage and fat mass index (p < 0.05), but not to body mass index (BMI) standard deviation score (SDS). In contrast LS bone mineral apparent density (BMAD) Z‐score remained stable and normal. The cumulative incidence of fragility fractures was 19.2%; DMD boys with fractures displayed a 1.5‐fold higher decline of TB BMD Z‐score/year (p < 0.05) and a worse adiposity profile compared to fracture‐free patients. No difference was found in DFZ dose or duration between the two groups.
Discussion
We observed a high incidence of fragility fractures, and identified fat tissue as a potential detrimental factor for bone health, suggesting a need for monitoring in DMD patients with excessive adiposity. Fat mass measures assessed by DXA could help to identify those at risk, enabling targeted interventions for better bone health. The co‐occurrence of multiple glucocorticoid side effects might characterize patients at higher risk of fractures.
This study identified fat tissue as a potential detrimental factor for bone health, suggesting a need for monitoring in DMD patients with excessive adiposity. The graphical was created with BioRender.com.</description><identifier>ISSN: 0148-639X</identifier><identifier>ISSN: 1097-4598</identifier><identifier>EISSN: 1097-4598</identifier><identifier>DOI: 10.1002/mus.28309</identifier><identifier>PMID: 39648958</identifier><language>eng</language><publisher>Hoboken, USA: John Wiley & Sons, Inc</publisher><subject>Absorptiometry, Photon ; Adipose tissue ; Biomarkers ; Body composition ; Body Composition - drug effects ; Body fat ; Body mass index ; Body size ; Bone composition ; Bone Density - drug effects ; Bone Density Conservation Agents - therapeutic use ; bone fragility ; Bone mass ; Bone mineral density ; Bone turnover ; Bulk density ; Child ; Clinical ; Dual energy X-ray absorptiometry ; Duchenne muscular dystrophy ; Duchenne's muscular dystrophy ; DXA bone mineral density ; Dystrophy ; Follow-Up Studies ; Fractures ; Fractures, Bone - chemically induced ; Fractures, Bone - epidemiology ; Fractures, Bone - etiology ; Fragility ; Glucocorticoids ; Humans ; Male ; Muscular dystrophy ; Muscular Dystrophy, Duchenne - complications ; Muscular Dystrophy, Duchenne - drug therapy ; osteoporosis ; Pregnenediones - adverse effects ; Pregnenediones - therapeutic use ; Side effects ; Spine ; Spine (lumbar) ; Time measurement</subject><ispartof>Muscle & nerve, 2025-02, Vol.71 (2), p.191-199</ispartof><rights>2024 The Author(s). published by Wiley Periodicals LLC.</rights><rights>2024 The Author(s). Muscle & Nerve published by Wiley Periodicals LLC.</rights><rights>2024. This article is published under http://creativecommons.org/licenses/by-nc-nd/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><cites>FETCH-LOGICAL-c2999-f7178fa8d25f2a3e7b5fd0e64d5d06f50d061807af560c0f5638845620a3ce643</cites><orcidid>0000-0003-4571-611X</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1002%2Fmus.28309$$EPDF$$P50$$Gwiley$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1002%2Fmus.28309$$EHTML$$P50$$Gwiley$$Hfree_for_read</linktohtml><link.rule.ids>230,314,776,780,881,1411,27901,27902,45550,45551</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/39648958$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Panicucci, Chiara</creatorcontrib><creatorcontrib>Casalini, Sara</creatorcontrib><creatorcontrib>Angelelli, Alessia</creatorcontrib><creatorcontrib>Brolatti, Noemi</creatorcontrib><creatorcontrib>Pedemonte, Marina</creatorcontrib><creatorcontrib>Patti, Giuseppa</creatorcontrib><creatorcontrib>Maghnie, Mohamad</creatorcontrib><creatorcontrib>Bruno, Claudio</creatorcontrib><creatorcontrib>Di Iorgi, Natascia</creatorcontrib><title>Bone Health Determinants in Ambulant Prepubertal Boys With Duchenne Muscular Dystrophy Treated With Deflazacort: Findings From a 3‐Year Study</title><title>Muscle & nerve</title><addtitle>Muscle Nerve</addtitle><description>ABSTRACT
Introduction/Aims
Duchenne muscular dystrophy (DMD) is complicated by bone fragility. This study aimed to elucidate changes in bone mineral density (BMD) and body composition over time and to explore associations with adiposity measures in DMD.
Methods
A three‐year follow‐up analysis was performed of total body (TB) and lumbar spine (LS) dual‐energy x‐ray absorptiometry (DXA) measurements, anthropometric measures, Tanner stage and bone turnover biomarkers assessments, and the incidence of fragility fractures in 26 ambulant prepubertal DMD patients treated with deflazacort (DFZ).
Results
Age at baseline was 7.7 years (interquartile range: 6–9.2). The TB BMD Z‐score declined over time and was negatively related to the TB fat mass percentage and fat mass index (p < 0.05), but not to body mass index (BMI) standard deviation score (SDS). In contrast LS bone mineral apparent density (BMAD) Z‐score remained stable and normal. The cumulative incidence of fragility fractures was 19.2%; DMD boys with fractures displayed a 1.5‐fold higher decline of TB BMD Z‐score/year (p < 0.05) and a worse adiposity profile compared to fracture‐free patients. No difference was found in DFZ dose or duration between the two groups.
Discussion
We observed a high incidence of fragility fractures, and identified fat tissue as a potential detrimental factor for bone health, suggesting a need for monitoring in DMD patients with excessive adiposity. Fat mass measures assessed by DXA could help to identify those at risk, enabling targeted interventions for better bone health. The co‐occurrence of multiple glucocorticoid side effects might characterize patients at higher risk of fractures.
This study identified fat tissue as a potential detrimental factor for bone health, suggesting a need for monitoring in DMD patients with excessive adiposity. The graphical was created with BioRender.com.</description><subject>Absorptiometry, Photon</subject><subject>Adipose tissue</subject><subject>Biomarkers</subject><subject>Body composition</subject><subject>Body Composition - drug effects</subject><subject>Body fat</subject><subject>Body mass index</subject><subject>Body size</subject><subject>Bone composition</subject><subject>Bone Density - drug effects</subject><subject>Bone Density Conservation Agents - therapeutic use</subject><subject>bone fragility</subject><subject>Bone mass</subject><subject>Bone mineral density</subject><subject>Bone turnover</subject><subject>Bulk density</subject><subject>Child</subject><subject>Clinical</subject><subject>Dual energy X-ray absorptiometry</subject><subject>Duchenne muscular dystrophy</subject><subject>Duchenne's muscular dystrophy</subject><subject>DXA bone mineral density</subject><subject>Dystrophy</subject><subject>Follow-Up Studies</subject><subject>Fractures</subject><subject>Fractures, Bone - chemically induced</subject><subject>Fractures, Bone - epidemiology</subject><subject>Fractures, Bone - etiology</subject><subject>Fragility</subject><subject>Glucocorticoids</subject><subject>Humans</subject><subject>Male</subject><subject>Muscular dystrophy</subject><subject>Muscular Dystrophy, Duchenne - complications</subject><subject>Muscular Dystrophy, Duchenne - drug therapy</subject><subject>osteoporosis</subject><subject>Pregnenediones - adverse effects</subject><subject>Pregnenediones - therapeutic use</subject><subject>Side effects</subject><subject>Spine</subject><subject>Spine (lumbar)</subject><subject>Time measurement</subject><issn>0148-639X</issn><issn>1097-4598</issn><issn>1097-4598</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2025</creationdate><recordtype>article</recordtype><sourceid>24P</sourceid><sourceid>EIF</sourceid><recordid>eNp1kc9u1DAQhy0EokvhwAsgS1zgkHacxI7DpeofliK1AqmtgJPlTSZdV4m92E6rcOIN4Bl5Elx2qQCJy1iWv_k04x8hTxnsMIB8dxjDTi4LqO-RGYO6ykpey_tkBqyUmSjqj1vkUQhXAMCkqB6SraIWpay5nJFvB84iPUbdxyU9woh-MFbbGKixdH9YjH260PceV-MCfdQ9PXBToB_MLT42S7Sp_XQMTQI9PZpC9G61nOi5Rx2x3YDY9fqLbpyPr-jc2NbYy0Dn3g1U0-LH1--fMDWfxbGdHpMHne4DPtmc2-Ri_vr88Dg7effm7eH-SdbkdV1nXcUq2WnZ5rzLdYHVgnctoChb3oLoOKTKJFS64wIaSLWQsuQiB100CSu2yd7am_YasG3QRq97tfJm0H5SThv194s1S3XprhVjFciyFMnwYmPw7vOIIarBhAb79GHoxqAKVgouQQqZ0Of_oFdu9Dbtlyie86TkkKiXa6rxLgSP3d00DNRtzirlrH7lnNhnf45_R_4ONgG7a-DG9Dj936ROL87Wyp8kcLTb</recordid><startdate>202502</startdate><enddate>202502</enddate><creator>Panicucci, Chiara</creator><creator>Casalini, Sara</creator><creator>Angelelli, Alessia</creator><creator>Brolatti, Noemi</creator><creator>Pedemonte, Marina</creator><creator>Patti, Giuseppa</creator><creator>Maghnie, Mohamad</creator><creator>Bruno, Claudio</creator><creator>Di Iorgi, Natascia</creator><general>John Wiley & Sons, Inc</general><general>Wiley Subscription Services, Inc</general><scope>24P</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>7T5</scope><scope>7TK</scope><scope>7TM</scope><scope>7TS</scope><scope>7U7</scope><scope>7U9</scope><scope>C1K</scope><scope>H94</scope><scope>K9.</scope><scope>NAPCQ</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0003-4571-611X</orcidid></search><sort><creationdate>202502</creationdate><title>Bone Health Determinants in Ambulant Prepubertal Boys With Duchenne Muscular Dystrophy Treated With Deflazacort: Findings From a 3‐Year Study</title><author>Panicucci, Chiara ; Casalini, Sara ; Angelelli, Alessia ; Brolatti, Noemi ; Pedemonte, Marina ; Patti, Giuseppa ; Maghnie, Mohamad ; Bruno, Claudio ; Di Iorgi, Natascia</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c2999-f7178fa8d25f2a3e7b5fd0e64d5d06f50d061807af560c0f5638845620a3ce643</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2025</creationdate><topic>Absorptiometry, Photon</topic><topic>Adipose tissue</topic><topic>Biomarkers</topic><topic>Body composition</topic><topic>Body Composition - drug effects</topic><topic>Body fat</topic><topic>Body mass index</topic><topic>Body size</topic><topic>Bone composition</topic><topic>Bone Density - drug effects</topic><topic>Bone Density Conservation Agents - therapeutic use</topic><topic>bone fragility</topic><topic>Bone mass</topic><topic>Bone mineral density</topic><topic>Bone turnover</topic><topic>Bulk density</topic><topic>Child</topic><topic>Clinical</topic><topic>Dual energy X-ray absorptiometry</topic><topic>Duchenne muscular dystrophy</topic><topic>Duchenne's muscular dystrophy</topic><topic>DXA bone mineral density</topic><topic>Dystrophy</topic><topic>Follow-Up Studies</topic><topic>Fractures</topic><topic>Fractures, Bone - chemically induced</topic><topic>Fractures, Bone - epidemiology</topic><topic>Fractures, Bone - etiology</topic><topic>Fragility</topic><topic>Glucocorticoids</topic><topic>Humans</topic><topic>Male</topic><topic>Muscular dystrophy</topic><topic>Muscular Dystrophy, Duchenne - complications</topic><topic>Muscular Dystrophy, Duchenne - drug therapy</topic><topic>osteoporosis</topic><topic>Pregnenediones - adverse effects</topic><topic>Pregnenediones - therapeutic use</topic><topic>Side effects</topic><topic>Spine</topic><topic>Spine (lumbar)</topic><topic>Time measurement</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Panicucci, Chiara</creatorcontrib><creatorcontrib>Casalini, Sara</creatorcontrib><creatorcontrib>Angelelli, Alessia</creatorcontrib><creatorcontrib>Brolatti, Noemi</creatorcontrib><creatorcontrib>Pedemonte, Marina</creatorcontrib><creatorcontrib>Patti, Giuseppa</creatorcontrib><creatorcontrib>Maghnie, Mohamad</creatorcontrib><creatorcontrib>Bruno, Claudio</creatorcontrib><creatorcontrib>Di Iorgi, Natascia</creatorcontrib><collection>Wiley Online Library Open Access</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>Neurosciences Abstracts</collection><collection>Nucleic Acids Abstracts</collection><collection>Physical Education Index</collection><collection>Toxicology Abstracts</collection><collection>Virology and AIDS Abstracts</collection><collection>Environmental Sciences and Pollution Management</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Premium</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Muscle & nerve</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Panicucci, Chiara</au><au>Casalini, Sara</au><au>Angelelli, Alessia</au><au>Brolatti, Noemi</au><au>Pedemonte, Marina</au><au>Patti, Giuseppa</au><au>Maghnie, Mohamad</au><au>Bruno, Claudio</au><au>Di Iorgi, Natascia</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Bone Health Determinants in Ambulant Prepubertal Boys With Duchenne Muscular Dystrophy Treated With Deflazacort: Findings From a 3‐Year Study</atitle><jtitle>Muscle & nerve</jtitle><addtitle>Muscle Nerve</addtitle><date>2025-02</date><risdate>2025</risdate><volume>71</volume><issue>2</issue><spage>191</spage><epage>199</epage><pages>191-199</pages><issn>0148-639X</issn><issn>1097-4598</issn><eissn>1097-4598</eissn><abstract>ABSTRACT
Introduction/Aims
Duchenne muscular dystrophy (DMD) is complicated by bone fragility. This study aimed to elucidate changes in bone mineral density (BMD) and body composition over time and to explore associations with adiposity measures in DMD.
Methods
A three‐year follow‐up analysis was performed of total body (TB) and lumbar spine (LS) dual‐energy x‐ray absorptiometry (DXA) measurements, anthropometric measures, Tanner stage and bone turnover biomarkers assessments, and the incidence of fragility fractures in 26 ambulant prepubertal DMD patients treated with deflazacort (DFZ).
Results
Age at baseline was 7.7 years (interquartile range: 6–9.2). The TB BMD Z‐score declined over time and was negatively related to the TB fat mass percentage and fat mass index (p < 0.05), but not to body mass index (BMI) standard deviation score (SDS). In contrast LS bone mineral apparent density (BMAD) Z‐score remained stable and normal. The cumulative incidence of fragility fractures was 19.2%; DMD boys with fractures displayed a 1.5‐fold higher decline of TB BMD Z‐score/year (p < 0.05) and a worse adiposity profile compared to fracture‐free patients. No difference was found in DFZ dose or duration between the two groups.
Discussion
We observed a high incidence of fragility fractures, and identified fat tissue as a potential detrimental factor for bone health, suggesting a need for monitoring in DMD patients with excessive adiposity. Fat mass measures assessed by DXA could help to identify those at risk, enabling targeted interventions for better bone health. The co‐occurrence of multiple glucocorticoid side effects might characterize patients at higher risk of fractures.
This study identified fat tissue as a potential detrimental factor for bone health, suggesting a need for monitoring in DMD patients with excessive adiposity. The graphical was created with BioRender.com.</abstract><cop>Hoboken, USA</cop><pub>John Wiley & Sons, Inc</pub><pmid>39648958</pmid><doi>10.1002/mus.28309</doi><tpages>9</tpages><orcidid>https://orcid.org/0000-0003-4571-611X</orcidid><oa>free_for_read</oa></addata></record> |
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| subjects | Absorptiometry, Photon Adipose tissue Biomarkers Body composition Body Composition - drug effects Body fat Body mass index Body size Bone composition Bone Density - drug effects Bone Density Conservation Agents - therapeutic use bone fragility Bone mass Bone mineral density Bone turnover Bulk density Child Clinical Dual energy X-ray absorptiometry Duchenne muscular dystrophy Duchenne's muscular dystrophy DXA bone mineral density Dystrophy Follow-Up Studies Fractures Fractures, Bone - chemically induced Fractures, Bone - epidemiology Fractures, Bone - etiology Fragility Glucocorticoids Humans Male Muscular dystrophy Muscular Dystrophy, Duchenne - complications Muscular Dystrophy, Duchenne - drug therapy osteoporosis Pregnenediones - adverse effects Pregnenediones - therapeutic use Side effects Spine Spine (lumbar) Time measurement |
| title | Bone Health Determinants in Ambulant Prepubertal Boys With Duchenne Muscular Dystrophy Treated With Deflazacort: Findings From a 3‐Year Study |
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