{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T20:40:13Z","timestamp":1790973613595,"version":"4.1.0"},"reference-count":46,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100006364","name":"Institut National Du Cancer","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100006364","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["American J Hematol"],"published-print":{"date-parts":[[2023,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Familial forms of monoclonal gammopathy, defined as multiple myeloma (MM) or Monoclonal Gammopathy of Undetermined Significance (MGUS), are relatively infrequent and most series reported in the literature describe a limited number of families. MM rarely occurs in a familial context. MGUS is observed much more commonly, which can in some cases evolve toward full\u2010blown MM. Although recurrent cytogenetic abnormalities have been described in tumor cells of sporadic cases of MM, the pathogenesis of familial MM remains largely unexplained. In order to identify genetic factors predisposing to familial monoclonal gammopathy, the Intergroupe Francophone du My\u00e9lome identified 318 families with at least two confirmed cases of monoclonal gammopathy. There were 169 families with parent\/child pairs and 164 families with cases in at least two siblings, compatible with an autosomal transmission. These familial cases were compared with sporadic cases who were matched for age at diagnosis, sex and immunoglobulin isotype, with 10 sporadic cases for each familial case. The gender distribution, age and immunoglobulin subtypes of familial cases were unremarkable in comparison to sporadic cases. With a median follow\u2010up of 7.4\u2009years after diagnosis, the percentage of MGUS cases having evolved to MM was 3%. The median overall survival of the 148 familial MM cases was longer than that of matched sporadic cases, with projected values of 7.6 and 16.1\u2009years in patients older and younger than 65\u2009years, respectively. These data suggest that familial cases of monoclonal gammopathy are similar to sporadic cases in terms of clinical presentation and carry a better prognosis.<\/jats:p>","DOI":"10.1002\/ajh.26785","type":"journal-article","created":{"date-parts":[[2023,1,2]],"date-time":"2023-01-02T07:04:22Z","timestamp":1672643062000},"page":"264-271","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Clinical characteristics and outcome of 318 families with familial monoclonal gammopathy: A multicenter Intergroupe Francophone du My\u00e9lome study"],"prefix":"10.1002","volume":"98","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1875-134X","authenticated-orcid":false,"given":"Charles","family":"Dumontet","sequence":"first","affiliation":[{"name":"Hospices Civils de Lyon  Lyon France"},{"name":"CRCL, UMR INSERM 1052\/CNRS 5286\/University of Lyon\u2010France  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6550-2827","authenticated-orcid":false,"given":"Delphine","family":"Demangel","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Perrine","family":"Galia","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lionel","family":"Karlin","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laurent","family":"Roche","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mathieu","family":"Fauvernier","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Camille","family":"Golfier","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9967-706X","authenticated-orcid":false,"given":"Marie\u2010Charlotte","family":"Laude","sequence":"additional","affiliation":[{"name":"Hospices Civils de Lyon  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xavier","family":"Leleu","sequence":"additional","affiliation":[{"name":"Hematology Department CHU Poitiers  Poitiers France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Philippe","family":"Rodon","sequence":"additional","affiliation":[{"name":"Hematology Department CH P\u00e9rigueux  P\u00e9rigueux France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Murielle","family":"Roussel","sequence":"additional","affiliation":[{"name":"IUC\u2010Oncop\u00f4le  Toulouse France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabelle","family":"Aza\u00efs","sequence":"additional","affiliation":[{"name":"Hematology Department CHU Poitiers  Poitiers France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chantal","family":"Doyen","sequence":"additional","affiliation":[{"name":"UCL Mont\u2010Godinne  Louvain Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Borhane","family":"Slama","sequence":"additional","affiliation":[{"name":"Clinical Hematology Department CH Avignon  Avignon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Salomon","family":"Manier","sequence":"additional","affiliation":[{"name":"Hematology Department CHRU  Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olivier","family":"Decaux","sequence":"additional","affiliation":[{"name":"Hematology Department CHU Rennes, Inserm UMR1236  Rennes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maroulio","family":"Pertesi","sequence":"additional","affiliation":[{"name":"Genetic Cancer Susceptibility, International Agency for Research on Cancer  Lyon France"},{"name":"Department of Laboratory Medicine Hematology and Transfusion Medicine  Lund Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marie","family":"Beaumont","sequence":"additional","affiliation":[{"name":"Hematology Department CHU Dunkerque  Dunkerque France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Denis","family":"Caillot","sequence":"additional","affiliation":[{"name":"Clinical Hematology Department H\u00f4pital F. 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