{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,8]],"date-time":"2026-01-08T22:50:31Z","timestamp":1767912631954,"version":"3.49.0"},"reference-count":12,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,9,27]],"date-time":"2023-09-27T00:00:00Z","timestamp":1695772800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Cardiovasc. Med."],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Lymphocytic-variant hypereosinophilic syndrome (L-HES) is a form of reactive hypereosinophilia, most commonly associated with interleukin-5 over-production by clonal, most commonly CD3<jats:sup>\u2212<\/jats:sup>CD4<jats:sup>+<\/jats:sup>CD2<jats:sup>hi<\/jats:sup>CD5<jats:sup>hi<\/jats:sup>CD45RO<jats:sup>+<\/jats:sup> T-cells. Patients often present with predominant cutaneous and soft-tissue manifestations, while cardiovascular involvement is uncommon.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We reviewed the medical files of two L-HES patients followed in our center who developed serious vascular complications and performed a literature review for similar cases.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Patient 1, a 52-year-old female, presented with an ischemic stroke secondary to left middle cerebral artery dissection after 10 years of indolent L-HES. Blood eosinophilia was controlled with oral corticosteroids (OCS), but OCS-tapering attempts with hydroxyurea and pegylated interferon failed, prompting the introduction of mepolizumab with rapid normalization. Patient 2, a 62-year-old female, had been asymptomatic for 10 years without treatment when a NSTEMI occurred, due to coronary artery occlusion secondary to a large cauliflower-aneurysm of the proximal aorta and aneurysmal dilatation of several coronary arteries, requiring semi-urgent surgical management. Aortic wall staining for eosinophil major basic protein showed eosinophils in the adventitia. Blood eosinophilia was controlled with OCS.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>Patients with apparently clinically benign L-HES may develop arterial complications, consisting in dissection and\/or aneurysm dilatation of medium-to-large vessels with serious consequences. The value of performing regular vascular imaging and monitoring during follow-up has yet to be determined.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fcvm.2023.1256862","type":"journal-article","created":{"date-parts":[[2023,9,28]],"date-time":"2023-09-28T05:37:05Z","timestamp":1695879425000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":2,"title":["Case report: Serious unexpected vascular events in two patients with lymphocytic variant hypereosinophilic syndrome"],"prefix":"10.3389","volume":"10","author":[{"given":"Nathan","family":"Torcida","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Giulia","family":"Casalino","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Antoine","family":"Bondue","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lise","family":"Jodaitis","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Frederic","family":"Vanden Eynden","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Florence","family":"Roufosse","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2023,9,27]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"47","DOI":"10.1111\/all.15544","article-title":"Proposed refined diagnostic criteria and classification of eosinophil disorders and related syndromes","volume":"78","author":"Valent","year":"2023","journal-title":"Allergy"},{"key":"B2","doi-asserted-by":"publisher","first-page":"164","DOI":"10.1136\/heartjnl-2020-317202","article-title":"Hypereosinophilic syndrome: considerations for the cardiologist","volume":"108","author":"Bondue","year":"2022","journal-title":"Heart"},{"key":"B3","doi-asserted-by":"publisher","first-page":"3497","DOI":"10.1016\/j.jaip.2021.04.067","article-title":"Recurrent cardiac arrest due to eosinophilia-related coronary vasospasm successfully treated by benralizumab","volume":"9","author":"Groh","year":"2021","journal-title":"J Allergy Clin Immunol Pract"},{"key":"B4","doi-asserted-by":"publisher","first-page":"1765","DOI":"10.3389\/fimmu.2020.01765","article-title":"Eosinophilia associated with CD3(\u2212)CD4(+) T cells: characterization and outcome of a single-center cohort of 26 patients","volume":"11","author":"Carpentier","year":"2020","journal-title":"Front Immunol"},{"key":"B5","doi-asserted-by":"publisher","first-page":"1228","DOI":"10.1161\/CIRCULATIONAHA.118.036124","article-title":"Coronary arteries and the cell count","volume":"139","author":"Divanji","year":"2019","journal-title":"Circulation"},{"key":"B6","doi-asserted-by":"publisher","first-page":"2278","DOI":"10.1007\/s00415-011-6089-7","article-title":"Dissection of the posterior inferior cerebellar artery in the hypereosinophilic syndrome","volume":"258","author":"van Gaalen","year":"2011","journal-title":"J Neurol"},{"key":"B7","doi-asserted-by":"publisher","first-page":"255","DOI":"10.1097\/MD.0000000000000088","article-title":"The lymphoid variant of hypereosinophilic syndrome: study of 21 patients with CD3\u2212CD4+ aberrant T-cell phenotype","volume":"93","author":"Lef\u00e8vre","year":"2014","journal-title":"Medicine (Baltimore)"},{"key":"B8","doi-asserted-by":"publisher","first-page":"E7","DOI":"10.1136\/heart.84.3.e7","article-title":"Coexistence of giant aneurysm of sinus of valsalva and coronary artery aneurysm associated with idiopathic hypereosinophilic syndrome","volume":"84","author":"Okinaka","year":"2000","journal-title":"Heart"},{"key":"B9","doi-asserted-by":"publisher","first-page":"923","DOI":"10.1016\/0002-9343(82)90853-1","article-title":"Spontaneous coronary artery dissection and eosinophilic inflammation: a cause and effect relationship?","volume":"72","author":"Robinowitz","year":"1982","journal-title":"Am J Med"},{"key":"B10","doi-asserted-by":"publisher","first-page":"179","DOI":"10.1146\/annurev-pathmechdis-012419-032756","article-title":"Contributions of eosinophils to human health and disease","volume":"15","author":"Klion","year":"2020","journal-title":"Annu Rev Pathol Mech Dis"},{"key":"B11","doi-asserted-by":"publisher","first-page":"2969","DOI":"10.1182\/blood-2008-08-175091","article-title":"Molecular profiling of CD3\u2212CD4+ T cells from patients with the lymphocytic variant of hypereosinophilic syndrome reveals targeting of growth control pathways","volume":"114","author":"Ravoet","year":"2009","journal-title":"Blood"},{"key":"B12","doi-asserted-by":"publisher","first-page":"539","DOI":"10.1016\/0002-9343(87)90458-X","article-title":"Peripheral T cell lymphoma presenting as hypereosinophilia with vasculitis. 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