{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,28]],"date-time":"2026-03-28T11:48:09Z","timestamp":1774698489359,"version":"3.50.1"},"reference-count":24,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2023,8,7]],"date-time":"2023-08-07T00:00:00Z","timestamp":1691366400000},"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. Med."],"abstract":"<jats:p>Hemophagocytic lymphohistiocytosis (HLH) is a rare histiocytic disorder characterized by reactive hyperplasia of the mononuclear phagocytic system, which is primarily caused by dysfunction of cytotoxic killer cells and natural killer cells, leading to antigen clearance barriers and the overactivation of the mononuclear phagocytic system due to continuous antigen stimulation. HLH encompasses a group of clinical syndromes marked by the overproduction of inflammatory cytokines. A 68-year-old Chinese man presented with persistent fever, chills, nausea, and vomiting; the patient had no history of any underlying conditions. Laboratory investigations revealed decreased levels of red blood cells, white blood cells, and platelets, along with reduced natural killer cell activity, increased CD25, hyperferritinemia, and the detection of Rickettsia DNA in his blood, meeting the diagnostic criteria of the Histiocyte Society HLH-2004 guidelines. The patient was treated with antibiotics, improving anemia, glucocorticoid therapy, and continuous renal replacement therapy (CRRT), temporarily improving his condition. However, the patient died after 2 years from chronic renal failure caused by septic shock.<\/jats:p>","DOI":"10.3389\/fmed.2023.1209174","type":"journal-article","created":{"date-parts":[[2023,8,7]],"date-time":"2023-08-07T14:35:33Z","timestamp":1691418933000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":4,"title":["Case report: A case of sepsis caused by rickettsial infection-induced hemophagocytic syndrome"],"prefix":"10.3389","volume":"10","author":[{"given":"Yanli","family":"Cao","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peijun","family":"Liu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Qiuling","family":"Song","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jing","family":"Wang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2023,8,7]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"1319","DOI":"10.3390\/pathogens10101319","article-title":"An update on the laboratory diagnosis of rickettsia spp","volume":"10","author":"Stewart","year":"2021","journal-title":"Infection Pathogens."},{"key":"B2","doi-asserted-by":"publisher","first-page":"178","DOI":"10.1016\/j.ijid.2023.03.012","article-title":"Serological cross-reactivity between spotted fever and typhus groups of rickettsia infection in Japan","volume":"130","author":"Aita","year":"2023","journal-title":"Int J Infect Dis."},{"key":"B3","doi-asserted-by":"publisher","first-page":"101515","DOI":"10.1016\/j.berh.2020.101515","article-title":"Hemophagocytic lymphohistiocytosis: an update on pathogenesis, diagnosis, and therapy","volume":"34","author":"Griffin","year":"2020","journal-title":"Best Pract Res Clin Rheumatol."},{"key":"B4","doi-asserted-by":"publisher","first-page":"27","DOI":"10.1146\/annurev-pathol-020117-043625","article-title":"Hemophagocytic lymphohistiocytosis","volume":"13","author":"Al-Samkari","year":"2018","journal-title":"Annu Rev Pathol."},{"key":"B5","doi-asserted-by":"publisher","first-page":"507","DOI":"10.5858\/arpa.2020-0802-RA","article-title":"Hemophagocytic lymphohistiocytosis","volume":"146","author":"Ponnatt","year":"2022","journal-title":"Arch Pathol Lab Med."},{"key":"B6","doi-asserted-by":"publisher","first-page":"124","DOI":"10.1002\/pbc.21039","article-title":"HLH-2004: diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis","volume":"48","author":"Henter","year":"2007","journal-title":"Pediatr Blood Cancer."},{"key":"B7","doi-asserted-by":"publisher","first-page":"411","DOI":"10.1016\/j.blre.2016.05.001","article-title":"Hemophagocytic syndromes (HPSs) including hemophagocytic lymphohistiocytosis (HLH) in adults: a systematic scoping review","volume":"30","author":"Hayden","year":"2016","journal-title":"Blood Rev."},{"key":"B8","doi-asserted-by":"publisher","first-page":"e309","DOI":"10.3928\/19382359-20170717-01","article-title":"An overview of hemophagocytic lymphohistiocytosis","volume":"46","author":"Esteban","year":"2017","journal-title":"Pediatr Ann."},{"key":"B9","doi-asserted-by":"publisher","first-page":"19","DOI":"10.1080\/10428194.2018.1482543","article-title":"Hemophagocytic lymphohistiocytosis in adults","volume":"60","author":"Bhatt","year":"2019","journal-title":"Leuk Lymphoma."},{"key":"B10","doi-asserted-by":"publisher","first-page":"933","DOI":"10.1378\/chest.11-0619","article-title":"Hemophagocytic lymphohistiocytosis: a potentially underrecognized association with systemic inflammatory response syndrome, severe sepsis, and septic shock in adults","volume":"140","author":"Raschke","year":"2011","journal-title":"Chest."},{"key":"B11","doi-asserted-by":"publisher","first-page":"387","DOI":"10.1097\/PCC.0b013e3181a1ae08","article-title":"Secondary hemophagocytic lymphohistiocytosis and severe sepsis\/ systemic inflammatory response syndrome\/multiorgan dysfunction syndrome\/macrophage activation syndrome share common intermediate phenotypes on a spectrum of inflammation","volume":"10","author":"Castillo","year":"2009","journal-title":"Pediatr Crit Care Med."},{"key":"B12","doi-asserted-by":"publisher","first-page":"16045","DOI":"10.1038\/nrdp.2016.45","article-title":"Sepsis and septic shock","volume":"2","author":"Hotchkiss","year":"2016","journal-title":"Nat Rev Dis Primers."},{"key":"B13","doi-asserted-by":"publisher","first-page":"290","DOI":"10.1097\/MCC.0000000000000834","article-title":"Hemodynamic clinical phenotyping in septic shock","volume":"27","author":"Daulasim","year":"2021","journal-title":"Curr Opin Crit Care."},{"key":"B14","doi-asserted-by":"publisher","first-page":"663","DOI":"10.1089\/vbz.2017.2231","article-title":"Molecular identification of rickettsia species in haemaphysalis ticks collected from Southwest China","volume":"18","author":"Zhang","year":"2018","journal-title":"Vector Borne Zoonotic Dis."},{"key":"B15","doi-asserted-by":"publisher","first-page":"343","DOI":"10.4103\/0255-0857.118874","article-title":"First case series of emerging rickettsial neonatal sepsis identified by polymerase chain reaction-based deoxyribonucleic acid sequencing","volume":"31","author":"Aarthi","year":"2013","journal-title":"Indian J Med Microbiol."},{"key":"B16","doi-asserted-by":"publisher","first-page":"bcr2012007155","DOI":"10.1136\/bcr-2012-007155","article-title":"Hitch-hiker taken for a ride: an unusual cause of myocarditis, septic shock and adult respiratory distress syndrome","volume":"2013","author":"Kushawaha","year":"2013","journal-title":"BMJ Case Rep."},{"key":"B17","doi-asserted-by":"publisher","first-page":"111","DOI":"10.1016\/j.jare.2018.05.007","article-title":"Clinical characteristics and effective treatments of scrub typhus-associated hemophagocytic lymphohistiocytosis in children","volume":"15","author":"Jin","year":"2019","journal-title":"J Adv Res."},{"key":"B18","first-page":"1324","article-title":"Secondary hemophagocytic lymphohistiocytosis in zoonoses. A systematic review","volume":"16","author":"Cascio","year":"2012","journal-title":"Eur Rev Med Pharmacol Sci."},{"key":"B19","doi-asserted-by":"publisher","first-page":"536","DOI":"10.3390\/jcm10030536","article-title":"Thrombotic thrombocytopenic purpura: pathophysiology, diagnosis, and management","volume":"10","author":"Sukumar","year":"2021","journal-title":"J Clin Med."},{"key":"B20","doi-asserted-by":"publisher","first-page":"235","DOI":"10.1016\/j.pcl.2018.09.011","article-title":"Hemolytic uremic syndrome","volume":"66","author":"Cody","year":"2019","journal-title":"Pediatr Clin North Am."},{"key":"B21","doi-asserted-by":"publisher","first-page":"e286","DOI":"10.1017\/S0950268819001390","article-title":"diagnosis of spotted fever group rickettsia infections: the Asian perspective","volume":"147","author":"Robinson","year":"2019","journal-title":"Epidemiol. Infect."},{"key":"B22","doi-asserted-by":"publisher","first-page":"165","DOI":"10.3947\/ic.2021.0070","article-title":"Does tigecycline have a place in therapy for rickettsial infection of the central nervous system?","volume":"54","author":"Mastroianni","year":"2022","journal-title":"Infect Chemother."},{"key":"B23","doi-asserted-by":"publisher","first-page":"817","DOI":"10.1111\/ped.13064","article-title":"Hemophagocytic lymphohistiocytosis: pathogenesis, diagnosis, and management","volume":"58","author":"Morimoto","year":"2016","journal-title":"Pediatr. Int."},{"key":"B24","doi-asserted-by":"publisher","first-page":"1118","DOI":"10.1016\/j.amjmed.2014.04.034","article-title":"Reactive hemophagocytic syndrome in adults: a retrospective analysis of 162 patients","volume":"127","author":"Rivi\u00e8re","year":"2014","journal-title":"Am. J. Med"}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2023.1209174\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,7]],"date-time":"2023-08-07T14:35:42Z","timestamp":1691418942000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2023.1209174\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,8,7]]},"references-count":24,"alternative-id":["10.3389\/fmed.2023.1209174"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2023.1209174","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,8,7]]},"article-number":"1209174"}}