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Prognostic factors for survival of herpes simplex virus-associated hemophagocytic lymphohistiocytosis.

Authors
  • Sonoda, Motoshi1
  • Ishimura, Masataka2
  • Eguchi, Katsuhide1
  • Shiraishi, Akira1
  • Kanno, Shunsuke1
  • Kaku, Noriyuki1
  • Inoue, Hirosuke1
  • Motomura, Yoshitomo1
  • Ochiai, Masayuki1
  • Sakai, Yasunari1
  • Nakayama, Manabu3
  • Ohara, Osamu3
  • Ohga, Shouichi1
  • 1 Department of Pediatrics, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka, 812-8582, Japan. , (Japan)
  • 2 Department of Pediatrics, Graduate School of Medical Sciences, Kyushu University, 3-1-1 Maidashi, Higashi-ku, Fukuoka, 812-8582, Japan. [email protected] , (Japan)
  • 3 Department of Technology Development, Kazusa DNA Research Institute, 2-6-7 Kazusa-Kamatari, Kisarazu, 292-0818, Chiba, Japan. , (Japan)
Type
Published Article
Journal
International Journal of Hematology
Publisher
Springer-Verlag
Publication Date
Jan 01, 2020
Volume
111
Issue
1
Pages
131–136
Identifiers
DOI: 10.1007/s12185-019-02738-3
PMID: 31549293
Source
Medline
Keywords
Language
English
License
Unknown

Abstract

Hemophagocytic lymphohistiocytosis (HLH) occurs in neonates with disseminated infection of herpes simplex virus (HSV). Little has been reported on the control of rapid HLH progression. We studied the cytokine profile and genetic basis of two index cases with divergent outcomes after early treatment of type 2 HSV infection. One survivor had fever and elevated serum levels of tumor necrosis factor (TNF)-α, interleukin-6 (IL-6), interferon (IFN)-β, and IFN-γ at diagnosis. The other neonate had no fever or TNF-α production, but significant IL-6 or IFN responses during the treatment course, and died 19 days after birth. Among 16 reported cases of neonatal HSV-HLH including index cases, eight deceased neonates experienced significantly less fever at presentation (p = 0.028), lower platelet counts (p = 0.019), and lower ratios of soluble IL-2 receptor (sIL-2R) to ferritin levels (p = 0.044) than eight survivors. The 100-day overall survival rates were significantly higher in patients with fever (p = 0.004), > 100 × 109/L of platelet counts (p = 0.035) or > 20 of sIL-2R/ferritin ratio at diagnosis (p = 0.004). The first febrile and cytokine responses to HSV infection predict the early outcome of neonatal HSV-HLH.

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