{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T11:51:17Z","timestamp":1753876277787,"version":"3.41.2"},"reference-count":7,"publisher":"Oxford University Press (OUP)","issue":"3","license":[{"start":{"date-parts":[[2021,3,31]],"date-time":"2021-03-31T00:00:00Z","timestamp":1617148800000},"content-version":"vor","delay-in-days":31,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,2,28]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Background\u2003<\/jats:title>\n                  <jats:p>Severe pulmonary arterial hypertension (PAH) is generally treated with multiple PAH-specific vasodilators. If these agents are unsuccessful, additional treatment options are scarce, and the prognosis is poor due to right-sided heart failure. Some of these severe cases are also accompanied by endocrinological side effects. The most common side effect of prostacyclin is thyroid dysfunction, but in very few cases, adrenocorticotropic hormone (ACTH) deficiency may occur.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Case summary\u2003<\/jats:title>\n                  <jats:p>A 35-year-old woman was diagnosed with hereditary PAH 2\u2009years ago. Since her mean pulmonary arterial pressure was high, combination therapy of vasodilators, including prostacyclin, was introduced. Several months later, she was hospitalized with a persistent fever. Laboratory tests showed no findings suggestive of infection. However, hypereosinophilia and decreased secretion of ACTH and cortisol were noted, which led to the diagnosis of ACTH deficiency. A multimodal diagnostic approach, including pituitary magnetic resonance imaging and axillary lymph node biopsy, indicated that the aetiology of the ACTH deficiency was likely autoimmune hypophysitis. She was treated with hydrocortisone supplementation, which significantly relieved her condition.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Discussion\u2003<\/jats:title>\n                  <jats:p>Endocrinological side effects in PAH patients using prostacyclin should be carefully addressed. If right-sided heart failure worsens during the administration of prostacyclin, it is essential to determine whether it is due to progression of pulmonary hypertension or endocrinological side effects. Careful diagnosis and treatment are important for managing the haemodynamics and symptoms of PAH patients given prostacyclin.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/ehjcr\/ytab117","type":"journal-article","created":{"date-parts":[[2021,3,31]],"date-time":"2021-03-31T06:26:52Z","timestamp":1617172012000},"source":"Crossref","is-referenced-by-count":1,"title":["Autoimmune hypophysitis as a cause of adrenocorticotropic hormone deficiency in pulmonary arterial hypertension: a case report"],"prefix":"10.1093","volume":"5","author":[{"given":"Genki","family":"Ichihara","sequence":"first","affiliation":[{"name":"Department of Cardiology, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8034-1005","authenticated-orcid":false,"given":"Masaharu","family":"Kataoka","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan"},{"name":"Second Department of Internal Medicine, University of Occupational and Environmental Health, Kitakyushu, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5576-5789","authenticated-orcid":false,"given":"Yoshinori","family":"Katsumata","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Keiichi","family":"Fukuda","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2021,3,31]]},"reference":[{"key":"2021071002570298800_ytab117-B1","doi-asserted-by":"crossref","first-page":"67","DOI":"10.1093\/eurheartj\/ehv317","article-title":"2015 ESC\/ERS guidelines for the diagnosis and treatment of pulmonary hypertension. The Joint Task Force for the Diagnosis and Treatment of Pulmonary Hypertension of the European Society of Cardiology (ESC) and the European Respiratory Society (ERS): endorsed by: Association for European Paediatric and Congenital Cardiology (AEPC), International Society for Heart and Lung Transplantation (ISHLT)","volume":"37","author":"Gali\u00e8","year":"2016","journal-title":"Eur Heart J"},{"key":"2021071002570298800_ytab117-B2","doi-asserted-by":"crossref","first-page":"598","DOI":"10.1177\/2045893217719250","article-title":"Management of prostacyclin side effects in adult patients with pulmonary arterial hypertension","volume":"7","author":"Kingman","year":"2017","journal-title":"Pulm Circ"},{"key":"2021071002570298800_ytab117-B3","doi-asserted-by":"crossref","first-page":"449","DOI":"10.1016\/j.ijcard.2010.10.115","article-title":"ACTH deficiency and PGI(2) therapy in chronic thromboembolic pulmonary hypertension","volume":"146","author":"Ikari","year":"2011","journal-title":"Int J Cardiol"},{"key":"2021071002570298800_ytab117-B4","doi-asserted-by":"crossref","first-page":"1963","DOI":"10.2169\/internalmedicine.48.2640","article-title":"Secondary adrenal insufficiency and primary antiphospholipid syndrome","volume":"48","author":"Alfadhli","year":"2009","journal-title":"Intern Med"},{"key":"2021071002570298800_ytab117-B5","doi-asserted-by":"crossref","first-page":"941","DOI":"10.1038\/gim.2013.41","article-title":"Alu-mediated nonallelic homologous and nonhomologous recombination in the BMPR2 gene in heritable pulmonary arterial hypertension","volume":"15","author":"Kataoka","year":"2013","journal-title":"Genet Med"},{"key":"2021071002570298800_ytab117-B6","doi-asserted-by":"crossref","first-page":"1766","DOI":"10.3174\/ajnr.A1714","article-title":"A radiologic score to distinguish autoimmune hypophysitis from nonsecreting pituitary adenoma preoperatively","volume":"30","author":"Gutenberg","year":"2009","journal-title":"AJNR Am J Neuroradiol"},{"key":"2021071002570298800_ytab117-B7","doi-asserted-by":"crossref","first-page":"e33518","DOI":"10.1371\/journal.pone.0033518","article-title":"Prostaglandin I2 signaling drives Th17 differentiation and exacerbates experimental autoimmune encephalomyelitis","volume":"7","author":"Zhou","year":"2012","journal-title":"PLoS One"}],"container-title":["European Heart Journal - Case Reports"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/academic.oup.com\/ehjcr\/article-pdf\/5\/3\/ytab117\/36830534\/ytab117.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/academic.oup.com\/ehjcr\/article-pdf\/5\/3\/ytab117\/36830534\/ytab117.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,7,10]],"date-time":"2021-07-10T06:58:32Z","timestamp":1625900312000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/ehjcr\/article\/doi\/10.1093\/ehjcr\/ytab117\/6205404"}},"subtitle":[],"editor":[{"given":"Yuichi","family":"Tamura","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]},{"given":"Erik","family":"Holy","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]},{"given":"Aiko","family":"Ogawa","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]},{"given":"Carlos Minguito","family":"Carazo","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]},{"given":"Deepti","family":"Ranganathan","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]}],"short-title":[],"issued":{"date-parts":[[2021,2,28]]},"references-count":7,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2021,2,28]]}},"URL":"https:\/\/doi.org\/10.1093\/ehjcr\/ytab117","relation":{},"ISSN":["2514-2119"],"issn-type":[{"type":"electronic","value":"2514-2119"}],"subject":[],"published-other":{"date-parts":[[2021,3,1]]},"published":{"date-parts":[[2021,2,28]]},"article-number":"ytab117"}}