{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,8]],"date-time":"2026-09-08T02:29:54Z","timestamp":1788834594116,"version":"build-2803163510"},"reference-count":48,"publisher":"MDPI AG","issue":"12","license":[{"start":{"date-parts":[[2022,6,15]],"date-time":"2022-06-15T00:00:00Z","timestamp":1655251200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Erich and Hanna Klessmann Foundation"},{"name":"Open Access Publication Funds of the Ruhr-Universit\u00e4t Bochum"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["IJMS"],"abstract":"<jats:p>Fukutin encoded by FKTN is a ribitol 5-phosphate transferase involved in glycosylation of \u03b1-dystroglycan. It is known that mutations in FKTN affect the glycosylation of \u03b1-dystroglycan, leading to a dystroglycanopathy. Dystroglycanopathies are a group of syndromes with a broad clinical spectrum including dilated cardiomyopathy and muscular dystrophy. In this study, we reported the case of a patient with muscular dystrophy, early onset dilated cardiomyopathy, and elevated creatine kinase levels who was a carrier of the compound heterozygous variants p.Ser299Arg and p.Asn442Ser in FKTN. Our work showed that compound heterozygous mutations in FKTN lead to a loss of fully glycosylated \u03b1-dystroglycan and result in cardiomyopathy and end-stage heart failure at a young age.<\/jats:p>","DOI":"10.3390\/ijms23126685","type":"journal-article","created":{"date-parts":[[2022,6,15]],"date-time":"2022-06-15T22:17:01Z","timestamp":1655331421000},"page":"6685","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Compound Heterozygous FKTN Variants in a Patient with Dilated Cardiomyopathy Led to an Aberrant \u03b1-Dystroglycan Pattern"],"prefix":"10.3390","volume":"23","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6899-7693","authenticated-orcid":false,"given":"Anna","family":"Gaertner","sequence":"first","affiliation":[{"name":"Erich und Hanna Klessmann-Institut f\u00fcr Kardiovaskul\u00e4re Forschung und Entwicklung, Klinik f\u00fcr Thorax- und Kardiovaskularchirurgie, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lidia","family":"Burr","sequence":"additional","affiliation":[{"name":"Erich und Hanna Klessmann-Institut f\u00fcr Kardiovaskul\u00e4re Forschung und Entwicklung, Klinik f\u00fcr Thorax- und Kardiovaskularchirurgie, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Baerbel","family":"Klauke","sequence":"additional","affiliation":[{"name":"Erich und Hanna Klessmann-Institut f\u00fcr Kardiovaskul\u00e4re Forschung und Entwicklung, Klinik f\u00fcr Thorax- und Kardiovaskularchirurgie, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9202-2343","authenticated-orcid":false,"given":"Andreas","family":"Brodehl","sequence":"additional","affiliation":[{"name":"Erich und Hanna Klessmann-Institut f\u00fcr Kardiovaskul\u00e4re Forschung und Entwicklung, Klinik f\u00fcr Thorax- und Kardiovaskularchirurgie, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kai Thorsten","family":"Laser","sequence":"additional","affiliation":[{"name":"Zentrum f\u00fcr Angeborene Herzfehler, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0203-2498","authenticated-orcid":false,"given":"Karin","family":"Klingel","sequence":"additional","affiliation":[{"name":"Kardiopathologie, Institut f\u00fcr Pathologie und Neuropathologie, Universit\u00e4tsklinikum T\u00fcbingen, Liebermeisterstra\u00dfe 8, 72076 T\u00fcbingen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jens","family":"Tiesmeier","sequence":"additional","affiliation":[{"name":"Erich und Hanna Klessmann-Institut f\u00fcr Kardiovaskul\u00e4re Forschung und Entwicklung, Klinik f\u00fcr Thorax- und Kardiovaskularchirurgie, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Uwe","family":"Schulz","sequence":"additional","affiliation":[{"name":"Erich und Hanna Klessmann-Institut f\u00fcr Kardiovaskul\u00e4re Forschung und Entwicklung, Klinik f\u00fcr Thorax- und Kardiovaskularchirurgie, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edzard zu","family":"Knyphausen","sequence":"additional","affiliation":[{"name":"Zentrum f\u00fcr Angeborene Herzfehler, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan","family":"Gummert","sequence":"additional","affiliation":[{"name":"Erich und Hanna Klessmann-Institut f\u00fcr Kardiovaskul\u00e4re Forschung und Entwicklung, Klinik f\u00fcr Thorax- und Kardiovaskularchirurgie, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9722-0007","authenticated-orcid":false,"given":"Hendrik","family":"Milting","sequence":"additional","affiliation":[{"name":"Erich und Hanna Klessmann-Institut f\u00fcr Kardiovaskul\u00e4re Forschung und Entwicklung, Klinik f\u00fcr Thorax- und Kardiovaskularchirurgie, Herz und Diabeteszentrum NRW, Universit\u00e4tsklinikum der Ruhr-Universit\u00e4t Bochum, Georgstr. 11, 32545 Bad Oeynhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2022,6,15]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1093\/jb\/mvu066","article-title":"Glycobiology of alpha-dystroglycan and muscular dystrophy","volume":"157","author":"Endo","year":"2015","journal-title":"J. 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