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We report here on a patient with clinical features of both syndromes, in whom epimutations were found at the BWS and TS imprinted regions, consistent with multilocus imprinting disturbance (MLID). This is the first case report of a patient with clinical features of both conditions who was found to have loss of methylation (LOM) of <jats:italic>KCNQ1OT1<\/jats:italic>: TSS\u2010DMR (ICR2) in the 11p15 imprinted region associated with BWS and LOM of <jats:italic>MEG3<\/jats:italic>: TSS\u2010DMR in the 14q32 imprinted region associated with TS. The report draws attention to the importance of testing for MLID as a cause of atypical clinical presentations of patients with IDs.<\/jats:p>","DOI":"10.1002\/ajmg.a.62717","type":"journal-article","created":{"date-parts":[[2022,3,10]],"date-time":"2022-03-10T06:19:44Z","timestamp":1646893184000},"page":"1896-1903","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["A patient with multilocus imprinting disturbance involving hypomethylation at 11p15 and 14q32, and phenotypic features of <scp>Beckwith\u2010Wiedemann<\/scp> and Temple syndromes"],"prefix":"10.1002","volume":"188","author":[{"given":"Sarah E.","family":"Grosvenor","sequence":"first","affiliation":[{"name":"Human Development and Health, Faculty of Medicine University of Southampton  Tremona Road Southampton UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7560-6320","authenticated-orcid":false,"given":"Justin H.","family":"Davies","sequence":"additional","affiliation":[{"name":"Human Development and Health, Faculty of Medicine University of Southampton  Tremona Road Southampton UK"},{"name":"Department of Paediatric Endocrinology University Hospital Southampton NHS Foundation Trusts  Southampton UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Margaret","family":"Lever","sequence":"additional","affiliation":[{"name":"Wessex Regional Genetics Laboratory Salisbury NHS Foundation Trust  Salisbury UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julie","family":"Sillibourne","sequence":"additional","affiliation":[{"name":"Wessex Regional Genetics Laboratory Salisbury NHS Foundation Trust  Salisbury UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3088-4401","authenticated-orcid":false,"given":"Deborah J. 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