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Trop. Med. Infect. Dis. 2018, 3(1), 19; doi:10.3390/tropicalmed3010019

Hemophagocytic Lymphohistiocytosis Associated with Scrub Typhus: Systematic Review and Comparison between Pediatric and Adult Cases

1
Stroke Center, Jichi Medical University 3311-1, Yakushiji, Shimotsuke-city, Tochigi 329-0498, Japan; shigeruf830@jichi.ac.jp
2
Rehabilitation Center, Jichi Medical University 3311-1, Yakushiji, Shimotsuke-city, Tochigi 329-0498, Japan
3
Division of Neurology, Department of Internal Medicine, Shin-Oyama City Hospital 2251-1, Hitotonoya, Oyama-city, Tochigi 323-0827, Japan
4
Division of Neurology, Department of Medicine, Jichi Medical University 3311-1, Yakushiji, Shimotsuke-city, Tochigi 329-0498, Japan
*
Author to whom correspondence should be addressed.
Received: 30 November 2017 / Revised: 31 January 2018 / Accepted: 6 February 2018 / Published: 14 February 2018
(This article belongs to the Special Issue The Past and Present Threat of Rickettsial Diseases)
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Abstract

Background: Scrub typhus is a mite-borne bacterial infection caused by Orientia tsutsugamushi. Hemophagocytic lymphohistiocytosis (HLH) is a potential severe complication. Most reported cases of HLH associated with scrub typhus were single cases or case series with a small sample sizes. Thus, no clear consensus exists on clinical manifestations and differences between pediatric and adult cases of this condition. Methods: a systematic search of English and Japanese articles from PubMed, PubMed Central, and Directory of Open Access Journals databases was performed from 3 December 2016 to 28 December 2017. The primary outcome was mortality in patients with HLH associated with scrub typhus; secondary outcomes were differences in clinical symptoms, laboratory findings, and treatment between pediatric and adult patients with HLH associated with scrub typhus. Results: thirty cases of HLH associated with scrub typhus were identified (age range: 2 months to 75 years; median age: 21.5 years, male:female ratio, 1:1). Eschar was frequently observed in the pediatric group (p = 0.017), whereas acute kidney injury was more prevalent in the adult group (p = 0.010). Two patients died of intracranial hemorrhage complicated with multiple organ failure; overall mortality rate was 6.7%. Conclusions: HLH associated with scrub typhus could be cured with remarkable improvement using single antibiotic therapy in approximately half the cases, with the mortality rate being relatively lower than that of HLH associated with other secondary causes. View Full-Text
Keywords: scrub typhus; Orientia tsutsugamushi; tsutsugamushi disease; hemophagocytosis; hemophagocytic lymphohistiocytosis scrub typhus; Orientia tsutsugamushi; tsutsugamushi disease; hemophagocytosis; hemophagocytic lymphohistiocytosis
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Naoi, T.; Morita, M.; Kawakami, T.; Fujimoto, S. Hemophagocytic Lymphohistiocytosis Associated with Scrub Typhus: Systematic Review and Comparison between Pediatric and Adult Cases. Trop. Med. Infect. Dis. 2018, 3, 19.

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