{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T01:18:21Z","timestamp":1790817501118,"version":"4.1.0"},"reference-count":67,"publisher":"The Endocrine Society","issue":"2","license":[{"start":{"date-parts":[[2020,1,24]],"date-time":"2020-01-24T00:00:00Z","timestamp":1579824000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports, Science, and Technology of Japan","doi-asserted-by":"crossref","award":["JP25860765"],"award-info":[{"award-number":["JP25860765"]}],"id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001700","name":"Ministry of Education, Culture, Sports, Science, and Technology of Japan","doi-asserted-by":"crossref","award":["JP17K09881"],"award-info":[{"award-number":["JP17K09881"]}],"id":[{"id":"10.13039\/501100001700","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,2,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Growth impairment in mucopolysaccharidoses (MPSs) is an unresolved issue as it is resistant to enzyme replacement therapy (ERT) and growth hormone therapy. C-type natriuretic peptide (CNP) is a promising agent that has growth-promoting effects. Here we investigate the effects of CNP on growth impairment of MPSs using Gusbmps-2J mice, a model for MPS type VII, with combination therapy of CNP and ERT by hydrodynamic gene delivery. Although monotherapies were not sufficient to restore short statures of treated mice, combination therapy resulted in successful restoration. The synergistic effects of CNP and ERT were not only observed in skeletal growth but also in growth plates. ERT reduced cell swelling in the resting zone and increased cell number by accelerating proliferation or inhibiting apoptosis. CNP thickened the proliferative and hypertrophic zones. Regarding changes in the bone, ERT restored bone sclerosis through decreased bone formation and increased bone resorption, and CNP did not adversely affect this process. In addition, improvement of joint deformation by ERT was suggested by analyses of joint spaces and articular cartilage. CNP additively provided restoration of the short stature of MPS VII mice in combination with ERT, which improved abnormalities of growth plates and bone metabolism.<\/jats:p>","DOI":"10.1210\/endocr\/bqaa008","type":"journal-article","created":{"date-parts":[[2020,1,24]],"date-time":"2020-01-24T05:46:43Z","timestamp":1579844803000},"source":"Crossref","is-referenced-by-count":23,"title":["C-Type Natriuretic Peptide Restores Growth Impairment Under Enzyme Replacement in Mice With Mucopolysaccharidosis VII"],"prefix":"10.1210","volume":"161","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9709-4231","authenticated-orcid":false,"given":"Takafumi","family":"Yamashita","sequence":"first","affiliation":[{"name":"Department of Diabetes, Endocrinology and Nutrition, Kyoto University Graduate School of Medicine, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0254-6974","authenticated-orcid":false,"given":"Toshihito","family":"Fujii","sequence":"first","affiliation":[{"name":"Department of Diabetes, Endocrinology and Nutrition, Kyoto University Graduate School of Medicine, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ichiro","family":"Yamauchi","sequence":"first","affiliation":[{"name":"Department of Diabetes, Endocrinology and Nutrition, Kyoto University Graduate School of Medicine, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yohei","family":"Ueda","sequence":"first","affiliation":[{"name":"Department of Diabetes, Endocrinology and Nutrition, Kyoto University Graduate School of Medicine, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keisho","family":"Hirota","sequence":"first","affiliation":[{"name":"Department of Diabetes, Endocrinology and Nutrition, Kyoto University Graduate School of Medicine, Kyoto, Japan"},{"name":"Laboratory of Bioimaging and Cell Signaling, Kyoto University Graduate School of Biostudies, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yugo","family":"Kanai","sequence":"first","affiliation":[{"name":"Department of Diabetes and Endocrinology, Osaka Red Cross Hospital, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akihiro","family":"Yasoda","sequence":"first","affiliation":[{"name":"Department of Diabetes, Endocrinology and Nutrition, Kyoto University Graduate School of Medicine, Kyoto, Japan"},{"name":"Clinical Research Institute, National Hospital Organization Kyoto Medical Center, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8261-2593","authenticated-orcid":false,"given":"Nobuya","family":"Inagaki","sequence":"first","affiliation":[{"name":"Department of Diabetes, Endocrinology and Nutrition, Kyoto University Graduate School of Medicine, Kyoto, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2020,1,24]]},"reference":[{"issue":"1-2","key":"2020022021322371800_CIT0001","doi-asserted-by":"crossref","first-page":"4","DOI":"10.1016\/j.ymgme.2018.01.003","article-title":"Clinical presentation and diagnosis of mucopolysaccharidoses","volume":"125","author":"Stapleton","year":"2018","journal-title":"Mol Genet Metab."},{"issue":"3","key":"2020022021322371800_CIT0002","doi-asserted-by":"crossref","first-page":"685","DOI":"10.1073\/pnas.84.3.685","article-title":"Cloning, sequencing, and expression of cDNA for human beta-glucuronidase","volume":"84","author":"Oshima","year":"1987","journal-title":"Proc Natl Acad Sci U S A."},{"issue":"4","key":"2020022021322371800_CIT0003","doi-asserted-by":"crossref","first-page":"511","DOI":"10.1002\/humu.20828","article-title":"Mutations and polymorphisms in GUSB gene in mucopolysaccharidosis VII (Sly syndrome)","volume":"30","author":"Tomatsu","year":"2009","journal-title":"Hum Mutat."},{"issue":"2","key":"2020022021322371800_CIT0004","doi-asserted-by":"crossref","first-page":"249","DOI":"10.1016\/S0022-3476(73)80162-3","article-title":"Beta glucuronidase deficiency: report of clinical, radiologic, and biochemical features of a new mucopolysaccharidosis","volume":"82","author":"Sly","year":"1973","journal-title":"J Pediatr."},{"issue":"3","key":"2020022021322371800_CIT0005","doi-asserted-by":"crossref","first-page":"182","DOI":"10.1056\/NEJM200101183440304","article-title":"Enzyme-replacement therapy in mucopolysaccharidosis I","volume":"344","author":"Kakkis","year":"2001","journal-title":"N Engl J Med."},{"issue":"5","key":"2020022021322371800_CIT0006","doi-asserted-by":"crossref","first-page":"581","DOI":"10.1016\/j.jpeds.2004.01.046","article-title":"Enzyme replacement therapy for mucopolysaccharidosis I: a randomized, double-blinded, placebo-controlled, multinational study of recombinant human alpha-L-iduronidase (laronidase)","volume":"144","author":"Wraith","year":"2004","journal-title":"J Pediatr."},{"issue":"8","key":"2020022021322371800_CIT0007","doi-asserted-by":"crossref","first-page":"465","DOI":"10.1097\/01.gim.0000232477.37660.fb","article-title":"A phase II\/III clinical study of enzyme replacement therapy with idursulfase in mucopolysaccharidosis II (Hunter syndrome)","volume":"8","author":"Muenzer","year":"2006","journal-title":"Genet Med."},{"issue":"4","key":"2020022021322371800_CIT0008","doi-asserted-by":"crossref","first-page":"533","DOI":"10.1016\/j.jpeds.2005.12.014","article-title":"Enzyme replacement therapy for mucopolysaccharidosis VI: a phase 3, randomized, double-blind, placebo-controlled, multinational study of recombinant human N-acetylgalactosamine 4-sulfatase (recombinant human arylsulfatase B or rhASB) and follow-on, open-label extension 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