Management of severe strongyloidiasis attended at reference centers in Spain

PLoS Negl Trop Dis. 2018 Feb 23;12(2):e0006272. doi: 10.1371/journal.pntd.0006272. eCollection 2018 Feb.

Abstract

Introduction: Strongyloides stercoralis is a globally distributed nematode that causes diverse clinical symptoms in humans. Spain, once considered an endemic country, has experienced a recent increase in imported cases. The introduction of serology helps diagnosis and is currently replacing microbiological techniques in some settings, but its sensitivity is variable and can be low in immunocompromised patients. Diagnosis can only be confirmed by identification of larvae. Often, this "gold standard" can only be achieved in severe cases, such as disseminated S.stercoralis infection, or S.stercoralis hyperinfection syndrome, where parasite load is high. In addition, these clinical presentations are not well-defined. Our aim is to describe severe cases of S.stercoralis, their epidemiological profile, and their clinical details.

Methods: An observational retrospective study of disseminated S.stercoralis infection, or hyperinfection syndrome. Inclusion criteria: aged over 18, with a diagnosis of disseminated S.stercoralis infection, or hyperinfection syndrome, confirmed by visualization of larvae. Patients were identified through revision of clinical records for the period 2000-2015, in collaboration with eight reference centers throughout Spain.

Results: From the period 2000-2015, eighteen cases were identified, 66.7% of which were male, with a median age of 40 (range 21-70). Most of them were foreigners (94.4%), mainly from Latin America (82.3%) or Western Africa (17.6%). Only one autochthonous case was identified, from 2006. Immunosuppressive conditions were present in fourteen (77%) patients, mainly due steroids use and to retroviral coinfections (four HIV, two HTLV). Transplant preceded the clinical presentation in four of them. Other comorbidities were coinfection with HBV, Trypanosoma cruzi, Mycobacterium leprae or Aspergillus spp. All presented with digestive disorders, with 55.6% also presenting malaise. 44.4% of cases had fever, 27.8% skin complaints, and 16.7% respiratory or neurological disorders. One patient presented anemia, and one other nephrotic syndrome. Diagnosis was confirmed by identification of larvae in fresh stool samples (n = 16; 88.9%), concentration techniques (n = 6; 33.3%), larval culture (n = 5; 29.4%), or digestive biopsies (n = 8; 44%). S.stercoralis forms were identified during necropsy in one case. In addition, ten (55%) had a positive serology. All the cases were treated with ivermectin, six (33%) also received albendazole and one case received thiabendazole followed by ivermectin. All needed inpatient management, involving a mean hospitalization stay of 25 days (range 1-164). Two cases received intensive care and eventually died.

Conclusions: Only eighteen cases of disseminated S.stercoralis infection/hyperinfection syndrome were identified from the 15-year period, most of which were considered to have been imported cases. Among those, immunosuppression was frequent, and mortality due to S.stercoralis was lower than previously described.

Publication types

  • Observational Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Albendazole / administration & dosage
  • Albendazole / therapeutic use
  • Animals
  • Antiparasitic Agents / administration & dosage
  • Antiparasitic Agents / therapeutic use
  • Communicable Diseases, Imported / drug therapy
  • Communicable Diseases, Imported / epidemiology
  • Communicable Diseases, Imported / parasitology
  • Communicable Diseases, Imported / therapy*
  • Comorbidity
  • Disease Management*
  • Emigrants and Immigrants
  • Feces / parasitology
  • Female
  • Humans
  • Immunocompromised Host
  • Ivermectin / administration & dosage
  • Ivermectin / therapeutic use
  • Larva / physiology
  • Male
  • Middle Aged
  • Referral and Consultation
  • Retrospective Studies
  • Spain / epidemiology
  • Strongyloides stercoralis / drug effects*
  • Strongyloides stercoralis / isolation & purification
  • Strongyloidiasis / diagnosis
  • Strongyloidiasis / drug therapy
  • Strongyloidiasis / epidemiology*
  • Strongyloidiasis / therapy*
  • Young Adult

Substances

  • Antiparasitic Agents
  • Ivermectin
  • Albendazole

Grants and funding

This study was supported by the 6th National Plan (PN) of Research + Development + Innovation (I+D+I) 2008–2011, ISCIII- General Division Networks and Cooperative Research Centers + FEDER funds + Collaborative Research Network on Tropical Diseases (RICET): RD12/0018/0020 and RD16/0027/0018 by the Sociedad Española de Medicina Tropical y Salud Internacional (SEMTSI). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.