{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T22:56:42Z","timestamp":1777503402376,"version":"3.51.4"},"reference-count":44,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2021,6,29]],"date-time":"2021-06-29T00:00:00Z","timestamp":1624924800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100006058","name":"St. Baldrick's Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006058","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["J cachexia sarcopenia muscle"],"published-print":{"date-parts":[[2021,8]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Survival from paediatric high\u2010risk neuroblastoma (HR\u2010NBL) has increased, but <jats:italic>cis<\/jats:italic>\u2010retinoic acid (<jats:italic>cis<\/jats:italic>\u2010RA), the cornerstone of HR\u2010NBL therapy, can cause osteoporosis and premature physeal closure and is a potential threat to skeletal structure in HR\u2010NBL survivors. Sarcopenia is associated with increased morbidity in survivors of paediatric malignancies. Low muscle mass may be associated with poor prognosis in HR\u2010NBL patients but has not been studied in these survivors. The study objective was to assess bone density, body composition and muscle strength in HR\u2010NBL survivors compared with controls.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>This prospective cross\u2010sectional study assessed areal bone mineral density (aBMD) of the whole body, lumbar spine, total hip, femoral neck, distal 1\/3 and ultradistal radius and body composition (muscle and fat mass) using dual\u2010energy X\u2010ray absorptiometry (DXA) and lower leg muscle strength using a dynamometer. Measures expressed as sex\u2010specific standard deviation scores (Z\u2010scores) included aBMD (adjusted for height Z\u2010score), bone mineral apparent density (BMAD), leg lean mass (adjusted for leg length), whole\u2010body fat mass index (FMI) and ankle dorsiflexion peak torque adjusted for leg length (strength\u2010Z). Muscle\u2010specific force was assessed as strength relative to leg lean mass. Outcomes were compared between HR\u2010NBL survivors and controls using Student's <jats:italic>t\u2010<\/jats:italic>test or Mann\u2013Whitney <jats:italic>U<\/jats:italic> test. Linear regression models examined correlations between DXA and dynamometer outcomes.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>We enrolled 20 survivors of HR\u2010NBL treated with <jats:italic>cis<\/jats:italic>\u2010RA [13 male; mean age: 12.4\u00a0\u00b1\u00a01.6\u00a0years; median (range) age at therapy initiation: 2.6 (0.3\u20139.1) years] and 20 age\u2010, sex\u2010 and race\u2010matched controls. Height\u2010Z was significantly lower in HR\u2010NBL survivors compared with controls (\u22121.73\u00a0\u00b1\u00a01.38 vs. 0.34\u00a0\u00b1\u00a01.12, <jats:italic>P<\/jats:italic>\u00a0&lt;\u00a00.001). Areal BMD\u2010Z, BMAD\u2010Z, FMI\u2010Z, visceral adipose tissue and subcutaneous adipose tissue were not significantly different in HR\u2010NBL survivors compared with controls. Compared with controls, HR\u2010NBL survivors had lower leg lean mass\u2010Z (\u22121.46\u00a0\u00b1\u00a01.35 vs. \u2212\u00a00.17\u00a0\u00b1\u00a00.84, <jats:italic>P<\/jats:italic>\u00a0&lt;\u00a00.001) and strength\u2010Z (\u22121.13\u00a0\u00b1\u00a00.86 vs. \u2212\u00a00.15\u00a0\u00b1\u00a00.71, <jats:italic>P<\/jats:italic>\u00a0&lt;\u00a00.001). Muscle\u2010specific force was lower in HR\u2010NBL survivors compared with controls (<jats:italic>P<\/jats:italic>\u00a0&lt;\u00a00.05).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Bone mineral density and adiposity are not severely impacted in HR\u2010NBL survivors with growth failure, but significant sarcopenia persists years after treatment. Future studies are needed to determine if sarcopenia improves with muscle\u2010specific interventions in this population of cancer survivors.<\/jats:p><\/jats:sec>","DOI":"10.1002\/jcsm.12734","type":"journal-article","created":{"date-parts":[[2021,6,29]],"date-time":"2021-06-29T15:48:05Z","timestamp":1624981685000},"page":"1024-1033","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":22,"title":["Sarcopenia and preserved bone mineral density in paediatric survivors of high\u2010risk neuroblastoma with growth failure"],"prefix":"10.1002","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4633-3997","authenticated-orcid":false,"given":"Michelle","family":"Guo","sequence":"first","affiliation":[{"name":"Department of Pediatrics, The Children's Hospital of Philadelphia, Perelman School of Medicine University of Pennsylvania  Philadelphia PA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Babette S.","family":"Zemel","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, The Children's Hospital of Philadelphia, Perelman School of Medicine University of Pennsylvania  Philadelphia PA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Colin P.","family":"Hawkes","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, The Children's Hospital of Philadelphia, Perelman School of Medicine University of Pennsylvania  Philadelphia PA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jin","family":"Long","sequence":"additional","affiliation":[{"name":"Center for Artificial Intelligence in Medicine and Imaging Stanford University  Stanford CA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andrea","family":"Kelly","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, The Children's Hospital of Philadelphia, Perelman School of Medicine University of Pennsylvania  Philadelphia PA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mary B.","family":"Leonard","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Lucile Packard Children's Hospital Stanford, Stanford University School of Medicine Stanford University  Stanford CA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Diego","family":"Jaramillo","sequence":"additional","affiliation":[{"name":"Department of Radiology, New York\u2010Presbyterian Morgan Stanley Children's Hospital Columbia University Irving Medical Center  New York NY USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sogol","family":"Mostoufi\u2010Moab","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, The Children's Hospital of Philadelphia, Perelman School of Medicine University of Pennsylvania  Philadelphia PA USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2021,6,29]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2006.05.6986"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199910143411601"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21683"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1038\/bmt.2013.218"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00247-019-04409-1"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0052237"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1200\/JCO.2009.22.3339"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0950-3579(05)80297-3"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00247-007-0604-0"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1006\/faat.1996.0179"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/0012-1606(87)90409-X"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/0012-1606(86)90096-5"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.28236"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.24040"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(19)31138-9"},{"key":"e_1_2_9_17_1","first-page":"11","article-title":"Skeletal muscle: an endocrine organ","volume":"10","author":"Pratesi A","year":"2013","journal-title":"Clin Cases Miner Bone Metab"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/jcsm.12570"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2011.06.041"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.21037\/qims.2020.04.06"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2018.04.012"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.2215\/CJN.01640309"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2009-1913"},{"key":"e_1_2_9_24_1","volume-title":"Assessment of skeletal maturity and prediction of adult height (TW3) method","author":"Tanner J","year":"2001"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/jbmr.2512"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1038\/oby.2011.367"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1002\/jbmr.2375"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1058-2746(96)80026-7"},{"key":"e_1_2_9_29_1","volume-title":"The Physical Activity Questionnaire for Older Children (PAQ\u2010C) and Adolescents (PAQ\u2010A) Manual","author":"Kowalski KC","year":"2004"},{"key":"e_1_2_9_30_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.109.1.45"},{"key":"e_1_2_9_31_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2020.06.051"},{"key":"e_1_2_9_32_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.dib.2020.106131"},{"key":"e_1_2_9_33_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2009-2057"},{"key":"e_1_2_9_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jocd.2014.01.003"},{"key":"e_1_2_9_35_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2011-1111"},{"key":"e_1_2_9_36_1","first-page":"1283","article-title":"Lumbar spine bone mineral apparent density in children: results from the bone mineral density in childhood study","volume":"104","author":"Kindler JM","year":"2019","journal-title":"J Clin Endocrinol Metab"},{"key":"e_1_2_9_37_1","doi-asserted-by":"publisher","DOI":"10.3945\/ajcn.112.053611"},{"key":"e_1_2_9_38_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2014.12.002"},{"key":"e_1_2_9_39_1","doi-asserted-by":"publisher","DOI":"10.1136\/heartjnl-2014-305723"},{"key":"e_1_2_9_40_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jss.2018.10.048"},{"key":"e_1_2_9_41_1","doi-asserted-by":"publisher","DOI":"10.1038\/bmt.2016.334"},{"key":"e_1_2_9_42_1","doi-asserted-by":"publisher","DOI":"10.1038\/bmt.2016.345"},{"key":"e_1_2_9_43_1","doi-asserted-by":"publisher","DOI":"10.1177\/1043454217698093"},{"key":"e_1_2_9_44_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00520-019-05131-2"},{"key":"e_1_2_9_45_1","doi-asserted-by":"publisher","DOI":"10.1002\/jcsm.12501"}],"container-title":["Journal of Cachexia, Sarcopenia and Muscle"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/jcsm.12734","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/jcsm.12734","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/jcsm.12734","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,29]],"date-time":"2023-08-29T08:07:16Z","timestamp":1693296436000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/jcsm.12734"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,6,29]]},"references-count":44,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2021,8]]}},"alternative-id":["10.1002\/jcsm.12734"],"URL":"https:\/\/doi.org\/10.1002\/jcsm.12734","archive":["Portico"],"relation":{},"ISSN":["2190-5991","2190-6009"],"issn-type":[{"value":"2190-5991","type":"print"},{"value":"2190-6009","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,6,29]]},"assertion":[{"value":"2021-01-18","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-05-21","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2021-06-29","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}