{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,5,14]],"date-time":"2025-05-14T04:48:01Z","timestamp":1747198081200,"version":"3.40.5"},"reference-count":40,"publisher":"Walter de Gruyter GmbH","issue":"3","license":[{"start":{"date-parts":[[2023,8,25]],"date-time":"2023-08-25T00:00:00Z","timestamp":1692921600000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,10,4]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec id=\"j_almed-2023-0071_abs_001\">\n                  <jats:title>Objectives<\/jats:title>\n                  <jats:p>The chimeras causing the CAH-X syndrome (SCAH-X) result from recombination between <jats:italic>CYP21A2<\/jats:italic>-<jats:italic>TNXB<\/jats:italic> and their respective pseudogenes (<jats:italic>CYP21A1P<\/jats:italic>-<jats:italic>TNXA<\/jats:italic>). The clinical manifestations of this syndrome include congenital adrenal hyperplasia (CAH) and Ehlers\u2013Danlos syndrome (EDS). Since SCAH-X has been recently described, the number of publications available is limited. The objective of this study was to set up a molecular approach and a screening algorithm for detecting CAH-X chimeras, determine their frequency and distribution in the Spanish population, and assess their clinical pattern of occurrence in a group of patients.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_almed-2023-0071_abs_002\">\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>A total of 186 patients were eligible for CAH-X molecular genetic testing. Testing included MLPA, heterodimer detection by capillary gel electrophoresis, and sequencing of exons 40, 41, and 43 of <jats:italic>TNXB<\/jats:italic>. A review was performed of the medical history of 20 patients from three hospitals of reference and the signs and symptoms of EDS they exhibited.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_almed-2023-0071_abs_003\">\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>In total, 78 CAH patients were carriers of CAH-X chimeras (41.9\u202f%). Forty-six patients were carriers of CH1 (24.7\u202f%), 24 of CH2 (12.9\u202f%), and 8 of CH3 (4.3\u202f%), with a heterogeneous geographical distribution. Seven (35\u202f%) of the\u00a020 carriers of a CAH-X chimera who underwent clinical examination experienced clinical manifestations of EDS.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_almed-2023-0071_abs_004\">\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>The impact of SCAH-X in the Spanish population was assessed by genetic testing. In the light of the clinical pattern of occurrence and significant prevalence of\u00a0SCAH-X in the Spanish population, early diagnosis of this entity is essential for an appropriate follow-up of clinical manifestations.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1515\/almed-2023-0071","type":"journal-article","created":{"date-parts":[[2023,8,24]],"date-time":"2023-08-24T08:20:45Z","timestamp":1692865245000},"page":"258-267","source":"Crossref","is-referenced-by-count":0,"title":["Molecular characterization of the new clinical entity associated with congenital adrenal hyperplasia: the CAH-X syndrome in the Spanish population"],"prefix":"10.1515","volume":"4","author":[{"given":"Laura Mart\u00ednez","family":"Figueras","sequence":"first","affiliation":[{"name":"Instituto de Investigaci\u00f3n Sanitaria Gregorio Mara\u00f1\u00f3n , Madrid , Spain"},{"name":"Laboratorio de Diagn\u00f3stico Molecular , Servicio de Bioqu\u00edmica Cl\u00ednica, Hospital General Universitario Gregorio Mara\u00f1\u00f3n , Madrid , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rafael Mu\u00f1oz","family":"Pacheco","sequence":"additional","affiliation":[{"name":"Laboratorio de Diagn\u00f3stico Molecular , Servicio de Bioqu\u00edmica Cl\u00ednica, Hospital General Universitario Gregorio Mara\u00f1\u00f3n , Madrid , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Dolores Garc\u00eda","family":"Gonz\u00e1lez","sequence":"additional","affiliation":[{"name":"Laboratorio de Diagn\u00f3stico Molecular , Servicio de Bioqu\u00edmica Cl\u00ednica, Hospital General Universitario Gregorio Mara\u00f1\u00f3n , Madrid , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6516-643X","authenticated-orcid":false,"given":"Mar\u00eda Arriba","family":"Dom\u00e8nech","sequence":"additional","affiliation":[{"name":"Laboratorio de Diagn\u00f3stico Molecular , Servicio de Bioqu\u00edmica Cl\u00ednica, Hospital General Universitario Gregorio Mara\u00f1\u00f3n , Madrid , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bego\u00f1a Ezquieta","family":"Zubicaray","sequence":"additional","affiliation":[{"name":"Instituto de Investigaci\u00f3n Sanitaria Gregorio Mara\u00f1\u00f3n , Madrid , Spain"},{"name":"Laboratorio de Diagn\u00f3stico Molecular , Servicio de Bioqu\u00edmica Cl\u00ednica, Hospital General Universitario Gregorio Mara\u00f1\u00f3n , Madrid , Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"374","published-online":{"date-parts":[[2023,8,25]]},"reference":[{"key":"2023100415020102014_j_almed-2023-0071_ref_001","doi-asserted-by":"crossref","unstructured":"Merke, DP, Bornstein, SR. 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