Clinical Features and Outcomes of Patients With Symptomatic Kaposi Sarcoma Herpesvirus (KSHV)-associated Inflammation: Prospective Characterization of KSHV Inflammatory Cytokine Syndrome (KICS)

Clin Infect Dis. 2016 Mar 15;62(6):730-738. doi: 10.1093/cid/civ996. Epub 2015 Dec 12.

Abstract

Background: Kaposi sarcoma herpesvirus (KSHV) is the cause of Kaposi sarcoma (KS), primary effusion lymphoma (PEL), and a form of Castleman disease (KSHV-MCD). Recently a KSHV-associated inflammatory cytokine syndrome (KICS) distinct from KSHV-MCD was reported.

Methods: We prospectively characterized the clinical, laboratory, virologic and immunologic features of KICS by evaluating symptomatic adults with KSHV using a prespecified definition. These features and overall survival were compared with controls from 2 prospectively characterized human immunodeficiency virus (HIV)-infected cohorts, including 1 with KSHV coinfection.

Results: All 10 KICS subjects were HIV infected males; 5 had HIV viral load (VL) suppressed <50 copies mL (median 72, range <50-74 375); all had KS and 2 also had PEL. All had multiple severe symptoms attributable to KICS: median number of symptoms 8 (6-11), median grade of worst symptom 3 (2-4). These included gastrointestinal disturbance (present in 9); edema (9); respiratory (6); and effusions (5). Laboratory abnormalities included anemia (all); hypoalbuminemia (all) and thrombocytopenia (6). None developed KSHV-MCD; 6 died with median survival from KICS diagnosis 13.6 months. KICS subjects compared with controls had more severe symptoms; lower hemoglobin and albumin; higher C-reactive protein; higher KSHV VL; elevated interleukin (IL)-6 and IL-10; and an increased risk of death (all P < .05). Anemia and hypoalbuminemia at presentation were independently associated with early death.

Conclusions: KICS subjects demonstrated diverse severe symptoms, a high rate of KSHV-associated tumors, high mortality, and a distinct IL-6/IL-10 signature. KICS may be an important unrecognized cause of morbidity and mortality, including symptoms previously ascribed to HIV. Exploration of KSHV-directed therapy is warranted.

Keywords: HIV; Human Herpesvirus 8 (HHV-8); IL-10; IL-6; Kaposi sarcoma herpesvirus (KSHV).

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural

MeSH terms

  • Adult
  • C-Reactive Protein / analysis
  • Coinfection / virology
  • Cytokines / adverse effects*
  • Cytokines / blood
  • Female
  • HIV Infections / drug therapy
  • HIV Infections / epidemiology
  • HIV Infections / immunology
  • HIV Infections / virology
  • Herpesvirus 8, Human / immunology
  • Humans
  • Inflammation / immunology
  • Inflammation / mortality
  • Inflammation / physiopathology*
  • Inflammation / virology*
  • Interleukin-10 / blood
  • Interleukin-6 / blood
  • Male
  • Middle Aged
  • Outcome Assessment, Health Care
  • Patient Outcome Assessment*
  • Prospective Studies
  • Sarcoma, Kaposi / epidemiology
  • Sarcoma, Kaposi / immunology*
  • Sarcoma, Kaposi / mortality
  • Sarcoma, Kaposi / virology*
  • Viral Load
  • Young Adult

Substances

  • Cytokines
  • IL10 protein, human
  • IL6 protein, human
  • Interleukin-6
  • Interleukin-10
  • C-Reactive Protein