{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T15:03:54Z","timestamp":1790694234168,"version":"4.1.0"},"reference-count":61,"publisher":"The Endocrine Society","issue":"3","license":[{"start":{"date-parts":[[2019,11,2]],"date-time":"2019-11-02T00:00:00Z","timestamp":1572652800000},"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\/501100001665","name":"Agence Nationale de la Recherche","doi-asserted-by":"publisher","award":["ANR-2011-JCJC-00701"],"award-info":[{"award-number":["ANR-2011-JCJC-00701"]}],"id":[{"id":"10.13039\/501100001665","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Epig\u00e9n\u00e9tique et Cancer","award":["EPIG201303"],"award-info":[{"award-number":["EPIG201303"]}]},{"name":"Direction G\u00e9n\u00e9rale de l\u2019Offre de Soins","award":["PRT-K 2014"],"award-info":[{"award-number":["PRT-K 2014"]}]},{"name":"Direction G\u00e9n\u00e9rale de l\u2019Offre de Soins","award":["INCa_DGOS_8663"],"award-info":[{"award-number":["INCa_DGOS_8663"]}]},{"DOI":"10.13039\/501100004099","name":"Ligue Nationale contre le Cancer","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100004099","id-type":"DOI","asserted-by":"crossref"}]},{"name":"European Union Seventh Framework Program","award":["FP7\/2007\u20132013"],"award-info":[{"award-number":["FP7\/2007\u20132013"]}]},{"name":"European Union Seventh Framework Program","award":["259735"],"award-info":[{"award-number":["259735"]}]},{"name":"European Union Seventh Framework Program","award":["633983"],"award-info":[{"award-number":["633983"]}]},{"name":"Institut National du Cancer and Direction G\u00e9n\u00e9rale de l\u2019Offre de Soins"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,3,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>Pheochromocytomas and paragangliomas (PPGLs) are neuroendocrine tumors explained by germline or somatic mutations in about 70% of cases. Patients with SDHB mutations are at high risk of developing metastatic disease, yet no reliable tumor biomarkers are available to predict tumor aggressiveness.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>We aimed at identifying long noncoding RNAs (lncRNAs) specific for PPGL molecular groups and metastatic progression.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Design and Methods<\/jats:title>\n                  <jats:p>To analyze the expression of lncRNAs, we used a mining approach of transcriptome data from a well-characterized series of 187 tumor tissues. Clustering consensus analysis was performed to determine a lncRNA-based classification, and informative transcripts were validated in an independent series of 51 PPGLs. The expression of metastasis-related lncRNAs was confirmed by RT-qPCR. Receiver operating characteristic (ROC) curve analysis was used to estimate the predictive accuracy of potential markers.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Main Outcome Measure<\/jats:title>\n                  <jats:p>Univariate\/multivariate and metastasis-free survival (MFS) analyses were carried out for the assessment of risk factors and clinical outcomes.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Four lncRNA-based subtypes strongly correlated with mRNA expression clusters (chi-square P-values from 1.38 \u00d7 10\u201332 to 1.07 \u00d7 10\u201367). We identified one putative lncRNA (GenBank: BC063866) that accurately discriminates metastatic from benign tumors in patients with SDHx mutations (area under the curve 0.95; P = 4.59 \u00d7 10\u201305). Moreover, this transcript appeared as an independent risk factor associated with poor clinical outcome of SDHx carriers (log-rank test P = 2.29 \u00d7 10\u201305).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusion<\/jats:title>\n                  <jats:p>Our findings extend the spectrum of transcriptional dysregulations in PPGL to lncRNAs and provide a novel biomarker that could be useful to identify potentially metastatic tumors in patients carrying SDHx mutations.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgz168","type":"journal-article","created":{"date-parts":[[2019,11,2]],"date-time":"2019-11-02T23:46:06Z","timestamp":1572738366000},"page":"898-907","source":"Crossref","is-referenced-by-count":19,"title":["Transcriptome Analysis of lncRNAs in Pheochromocytomas and Paragangliomas"],"prefix":"10.1210","volume":"105","author":[{"given":"Sylvie","family":"Job","sequence":"first","affiliation":[{"name":"Programme Cartes d\u2019Identit\u00e9 des Tumeurs, Ligue Nationale Contre Le Cancer, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4208-2393","authenticated-orcid":false,"given":"Adrien","family":"Georges","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Universit\u00e9 Paris Descartes, Sorbonne Paris Cit\u00e9, Facult\u00e9 de M\u00e9decine, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nelly","family":"Burnichon","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Universit\u00e9 Paris Descartes, Sorbonne Paris Cit\u00e9, Facult\u00e9 de M\u00e9decine, Paris, France"},{"name":"Genetics department, AP-HP, H\u00f4pital europ\u00e9en Georges Pompidou, Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandre","family":"Buffet","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Universit\u00e9 Paris Descartes, Sorbonne Paris Cit\u00e9, Facult\u00e9 de M\u00e9decine, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laurence","family":"Amar","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Universit\u00e9 Paris Descartes, Sorbonne Paris Cit\u00e9, Facult\u00e9 de M\u00e9decine, Paris, France"},{"name":"Assistance Publique-H\u00f4pitaux de Paris, H\u00f4pital Europ\u00e9en Georges Pompidou, Hypertension unit, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J\u00e9r\u00f4me","family":"Bertherat","sequence":"additional","affiliation":[{"name":"INSERM, U1016, Institut Cochin, Paris, France. 10 CNRS UMR8104, Paris, France"},{"name":"Rare Adrenal Cancer Network COMETE, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5424-2134","authenticated-orcid":false,"given":"Nabila","family":"Bouatia-Naji","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Universit\u00e9 Paris Descartes, Sorbonne Paris Cit\u00e9, Facult\u00e9 de M\u00e9decine, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aur\u00e9lien","family":"de Reyni\u00e8s","sequence":"additional","affiliation":[{"name":"Programme Cartes d\u2019Identit\u00e9 des Tumeurs, Ligue Nationale Contre Le Cancer, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Delphine","family":"Drui","sequence":"additional","affiliation":[{"name":"Service d\u2019Endocrinologie, Diab\u00e9tologie et Maladies M\u00e9taboliques, L\u2019institut du Thorax, Centre Hospitalier Universitaire de Nantes, H\u00f4pital Nord La\u00ebnnec, Nantes, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charlotte","family":"Lussey-Lepoutre","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Sorbonne Universit\u00e9, Piti\u00e9-Salp\u00eatri\u00e8re Hospital, Department of nuclear medicine, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Judith","family":"Favier","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Universit\u00e9 Paris Descartes, Sorbonne Paris Cit\u00e9, Facult\u00e9 de M\u00e9decine, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4816-670X","authenticated-orcid":false,"given":"Anne-Paule","family":"Gimenez-Roqueplo","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Universit\u00e9 Paris Descartes, Sorbonne Paris Cit\u00e9, Facult\u00e9 de M\u00e9decine, Paris, France"},{"name":"Genetics department, AP-HP, H\u00f4pital europ\u00e9en Georges Pompidou, Paris France"},{"name":"Rare Adrenal Cancer Network COMETE, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7232-9871","authenticated-orcid":false,"given":"Luis Jaime","family":"Castro-Vega","sequence":"additional","affiliation":[{"name":"Paris University, PARCC, INSERM, Equipe labellis\u00e9e par la Ligue contre le cancer, Paris, France"},{"name":"Universit\u00e9 Paris Descartes, Sorbonne Paris Cit\u00e9, Facult\u00e9 de M\u00e9decine, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2019,11,2]]},"reference":[{"issue":"9","key":"2021122101513641000_CIT0001","doi-asserted-by":"crossref","first-page":"1080","DOI":"10.1038\/onc.2015.172","article-title":"Rethinking pheochromocytomas and paragangliomas from a genomic perspective","volume":"35","author":"Castro-Vega","year":"2016","journal-title":"Oncogene."},{"issue":"2","key":"2021122101513641000_CIT0002","doi-asserted-by":"crossref","first-page":"181","DOI":"10.1016\/j.ccell.2017.01.001","article-title":"Comprehensive molecular characterization of pheochromocytoma and paraganglioma","volume":"31","author":"Fishbein","year":"2017","journal-title":"Cancer 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analysis defines core genomic alterations in pheochromocytomas and paragangliomas","volume":"6","author":"Castro-Vega","year":"2015","journal-title":"Nat Commun."},{"issue":"5","key":"2021122101513641000_CIT0010","doi-asserted-by":"crossref","DOI":"10.1093\/jnci\/djv053","article-title":"Whole-exome sequencing identifies MDH2 as a new familial paraganglioma gene","volume":"107","author":"Casc\u00f3n","year":"2015","journal-title":"J Natl Cancer Inst"},{"issue":"20","key":"2021122101513641000_CIT0011","doi-asserted-by":"crossref","first-page":"6315","DOI":"10.1158\/1078-0432.CCR-16-2250","article-title":"Targeted exome sequencing of Krebs cycle genes reveals candidate cancer-predisposing mutations in pheochromocytomas and paragangliomas","volume":"23","author":"Remacha","year":"2017","journal-title":"Clin Cancer Res."},{"issue":"12","key":"2021122101513641000_CIT0012","doi-asserted-by":"crossref","first-page":"1652","DOI":"10.1038\/s41436-018-0068-7","article-title":"Role of MDH2 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