Management of severe strongyloidiasis attended at reference centers in Spain.

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.

A 36-year-old man with vomiting, pain abdomen, significant weight loss, hyponatremia, and hypoglycemia.

Diagnosis of Strongyloides stercoralis hyperinfection can be a challenge. The key to a timely diagnosis is to have a high index of suspicion. We present a rare case of a 36-year-old human immunodeficiency virus negative male patient, who was on multidrug therapy for lepromatous leprosy and was treated for type 2 lepra reactions with steroids in the past. The patient presented with vomiting and pain abdomen, persistent hyponatremia, and terminal hypoglycemia. He had features of malnutrition and had a rapid downhill course following admission. A diagnosis of S. stercoralis hyperinfection with sepsis and multiorgan failure, adrenal hemorrhage, and syndrome of inappropriate antidiuretic hormone secretion was established on a postmortem examination.

Recurrence of strongyloides stercoralis infection in a patient with Hansen's disease: a case report.

In patients with immunosuppressive disorders, S. stercoralis infection may develop into a hyperinfection syndrome which, on rare occasions, may be a life-threatening condition. Therapy of S. stercoralis infection with thiabendazole has been limited, due to its numerous side effects, and has been replaced by albendazole and ivermectin. The present case report describes a case of Strongyloides Hyperinfection Syndrome (SHS) in a patient with Hansen's disease and lack of response to first-line anthelmintic treatment. A 38 year-old man was diagnosed as having borderline lepromatous leprosy. He developed Erythema Nodosum Leprosum and was treated with thalidomide and prednisone. In May 2010 he was diagnosed with S. stercoralis infection and was treated with albendazole. One year later, the stool examination showed continued presence of S. stercoralis larvae. He was treated with ivermectin (6 mg) in a double dose (given 1 month apart) which resulted in larvae excretion clearance. The absence of infection was confirmed three times during a 1 year followup period by stool examination and non-detection of anti-S. stercoralis IgG levels.

[Infection due to Strongyloides stercoralis: epidemiological, clinical, diagnosis findings and outcome in 30 patients]. / Infección por Strongyloides stercoralis: estudio epidemiológico, clínico, diagnóstico y terapéutico en 30 pacientes.

BACKGROUND: Strongyloides stercoralis is a nematode parasite, which is endemic in tropical and subtropical regions. Infection usually remains asymptomatic, but in immunocompromised hosts severe and life-threatening manifestations such as hyperinfection syndrome and disseminated disease might occur. METHODS: We retrospectively analyzed the epidemiological and clinical characteristics, including HIV co-infection, microbiological findings, and outcome in 30 patients with strongyloidiasis, who attended in the Infectious Diseases F. J. Muñiz Hospital in Buenos Aires from January 2004 to December 2008. RESULTS: The study included 20 men and 10 women with an average age of 33 years. HIV co-infection was present in 21 patients (70%) with a median CD4 T cell count of 50 cells/mm³ (range 7-355) (average 56 cells/mm³). Among HIV negative patients the following comorbidities were detected: tuberculosis (n = 3) and chronic alcoholism, leprosy treated with corticosteroids, immunosuppressive treatment for psoriasis, and diabetes mellitus (each in one patient). Two patients did not have any predisposing diseases or immunosuppressive treatments. Seventeen patients presented with diarrhea and were classified as chronic intestinal strongyloidiasis (57%), asymptomatic infection with peripheral eosinophilia was diagnosed in 7 (23%), and 6 patients (20%) developed hyperinfection syndrome. Seventeen patients (57%) presented peripheral eosinophilia. Diagnosis was achieved by direct visualization of larvae in feces by Baermann technique (n = 20), by multiple stool smears examinations (n = 2), by combination of both (n = 1), by visualization of the filariform larvae in duodenal fluid and stool (n = 1), and in fecal and bronchoalveolar lavage specimens (n = 6). Overall mortality in this series was 20% (6/30). There was no significant correlation between age and mortality. A significant inverse correlation between the survival rate and CD4 T-cell count as well as eosinophilia was observed. There was also a significant correlation between HIV co-infection and mortality. Twenty-two patients responded favorably to treatment with ivermectin.

Infección por Strongyloides stercoralis: estudio epidemiológico, clínico, diagnóstico y terapéutico en 30 pacientes / Infection due to Strongyloides stercoralis: epidemiological, clinical, diagnosis findings and outcome in 30 patients

Background: Strongyloides stercoralis is a nematode parasite, which is endemic in tropical and subtropical regions. Infection usually remains asymptomatic, but in immunocompromised hosts severe and life-threatening manifestations such as hyperinfection syndrome and disseminated disease might occur. Methods: We retrospectively analyzed the epidemiological and clinical characteristics, including HIV co-infection, microbiological findings, and outcome in 30 patients with strongyloidiasis, who attended in the Infectious Diseases F. J. Muñiz Hospital in Buenos Aires from January 2004 to December 2008. Results: The study included 20 men and 10 women with an average age of 33 years. HIV co-infection was present in 21 patients (70 percent) with a median CD4 T cell count of 50 cells/mm³ (range 7-355) (average 56 cells/mm³). Among HIV negative patients the following comorbidities were detected: tuberculosis (n = 3) and chronic alcoholism, leprosy treated with corticosteroids, immunosuppressive treatment for psoriasis, and diabetes mellitus (each in one patient). Two patients did not have any predisposing diseases or immunosuppressive treatments. Seventeen patients presented with diarrhea and were classified as chronic intestinal strongyloidiasis (57 percent), asymptomatic infection with peripheral eosinophilia was diagnosed in 7 (23 percent), and 6 patients (20 percent) developed hyperinfection syndrome. Seventeen patients (57 percent) presented peripheral eosinophilia. Diagnosis was achieved by direct visualization of larvae in feces by Baermann technique (n = 20), by multiple stool smears examinations (n = 2), by combination of both (n = 1), by visualization of the filariform larvae in duodenal fluid and stool (n = 1), and in fecal and bronchoalveolar lavage specimens (n = 6). Overall mortality in this series was 20 percent (6/30). There was no significant correlation between age and mortality. A significant inverse correlation between the survival rate and CD4 T-cell count as well as eosinophilia was observed. There was also a significant correlation between HIV co-infection and mortality. Twenty-two patients responded favorably to treatment with ivermectin.

Antecedentes: Strongyloides stercoralis, parásito endémico de áreas tropicales y subtropicales del planeta, en sujetos inmunodeprimidos puede cursar con formas graves y aun mortales como el síndrome de hiperinfestación y la enfermedad diseminada. Métodos: Análisis retrospectivo de las características epidemiológicas, manifestaciones clínicas, co-infección por virus de inmunodeficiencia humana (VIH), hallazgos microbiológicos y evolución de 30 pacientes con estrongiloidiasis, atendidos en el Hospital de Enfermedades Infecciosas F. J. Muñiz de Buenos Aires, entre enero 2004 y diciembre 2008. Resultados: Se incluyeron en la evaluación 20 hombres y 10 mujeres con una mediana de edad de 33 años. Co-infección por VIH hubo en 21 pacientes (70 por ciento); la mediana de linfocitos T CD4+ en ellos al momento del diagnóstico de la parasitosis fue de 50 céls/mm³ (rango 7 a 355), (media de 56 céls/mm³). En los pacientes seronegativos para VIH, se comprobaron las siguientes co-morbilidades: tuberculosis (n: 3) y un caso de cada una de las siguientes afecciones: alcoholismo crónico, diabetes mellitus, reacción lepromatosa bajo corticotera-pia, y psoriasis en tratamiento inmunosupresor. Hubo dos pacientes sin aparentes enfermedades de base. Diecisiete pacientes presentaron enfermedad intestinal crónica con diarrea (57 por ciento), era asintomática y fue sospechada por la eosinofilia periférica (n: 7, 23 por ciento) y se clasificó como síndrome de hiperinfestación (n: 6, 20 por ciento) diagnosticado por la identificación de larvas en la materia fecal y secreciones broncopulmonares. Diecisiete pacientes (57 por ciento) presentaron eosinofilia periférica. El diagnóstico se efectuó por la visualización directa de las larvas en muestras de heces en fresco mediante la técnica de concentración de Baer-man (n: 20); por el examen copro-parasitológico seriado (n: 2) y por ambos métodos (n: 1); en líquido duodenal y materia fecal (n: 1) y por la identificación de larvas en materia fecal y secreciones respiratorias (n: 6). Letalidad global: 20 por ciento (6/30). Los pacientes con eosinofilia tuvieron una menor letalidad que aquellos sin esta respuesta (p < 0,001). No hubo correlación estadística entre la edad y la supervivencia. Sí fue significativa la correlación entre el recuento de CD4 y la letalidad, incluyendo 18 de los 21 pacientes seropositivos para VIH (p: 0,03). Finalmente, la correlación seropositividad para VIH y letalidad también fue significativa. Veintidós pacientes respondieron a la terapia antiparasitaria con ivermectina y evolucionaron favorablemente.

Death caused by strongyloides hyperinfection in a leprosy patient on treatment for a type II leprosy reaction.

Strongyloides stercoralis is present worldwide and can cause hyperinfection in patients on long-term immunosuppressive doses of steroids, as is sometimes the case for patients treated for leprosy reactions. Strongyloides hyperinfection can present with ileus, as is discussed in this case report. Physicians, including surgeons, should be aware of this entity in order to avoid an unnecessary laparotomy. Though patients may survive if diagnosed at an early stage, strongyloides hyperinfection syndrome has a mortality rate of 87% and prevention is therefore of utmost importance.