Abstract

Vascular pythiosis is a rare, neglected, life-threatening disease with mortality of 100% in patients with incomplete surgical resection or patients with persistently elevated serum β-d-glucan (BDG). The study was conducted to understand the clinical outcomes of new treatment protocols and potential use of erythrocyte sedimentation rate (ESR) and c-reactive protein (CRP) as alternative monitoring tools, given recent favorable minimum inhibitory concentrations (MICs) of antibacterial agents and prohibitive cost of serum BDG in Thailand. A prospective cohort study of patients with vascular pythiosis was conducted between February 2019 and August 2020. After diagnosis, patients were followed at 0.5, 1, 1.5, 3, and 6 months. Descriptive statistics, Spearman’s correlation coefficient, and general linear model for longitudinal data were used. Amongst the cohort of ten vascular pythiosis patients, four had residual disease after surgery. Among four with residual disease, one developed disseminated disease and died, one developed relapse disease requiring surgery, and two were successfully managed with antimicrobial agents. The spearman’s correlation coefficients between BDG and ESR, and between BDG and CRP in patients without relapse or disseminated disease were 0.65 and 0.60, respectively. Tetracyclines and macrolides had most favorable minimum inhibitory concentrations and synergistic effects were observed in combinations of these two antibiotic classes. Adjunctive use of azithromycin and doxycycline preliminarily improved survival in vascular pythiosis patients with residual disease. Further studies are needed to understand the trends of ESR and CRP in this population.

Highlights

  • IntroductionAn aquatic fungal-like pathogen, causes pythiosis in humans

  • Pythium insidiosum, an aquatic fungal-like pathogen, causes pythiosis in humans.Human pythiosis was first described in a Thai thalassemia patient in 1988 [1]

  • This is a multicenter, prospective cohort study of vascular pythiosis patients who were treated according to the King Chulalongkorn Memorial Hospital (KCMH) research treatment protocols with a combination of radical surgery and antimicrobial agents from February 2019 to August 2020

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Summary

Introduction

An aquatic fungal-like pathogen, causes pythiosis in humans. Human pythiosis was first described in a Thai thalassemia patient in 1988 [1]. The clinical presentations are classified into four groups: vascular, ocular, subcutaneous/cutaneous, and disseminated infection [2]. Risk factors often include occupation in agriculture and/or underlying hemoglobinopathy, including thalassemia, in patients presenting from an endemic region [2,10,13,14]. The proposed mechanisms of infection in patients with hemoglobinopathy are interference of phagocytic activities of macrophages and neutrophils from iron overload, lower interferon gamma (IFN-γ) production in Thalassemia patients, and intrinsic virulence of P. insidiosum by carrying a gene encoding ferrochelatase [15,16,17]

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