Disseminated Strongyloidiasis in a Patient Treated with Rituximab and Ibrutinib, a Case Report

Case Report

Ann Hematol Oncol. 2022; 9(4): 1404.

Disseminated Strongyloidiasis in a Patient Treated with Rituximab and Ibrutinib, a Case Report

Vic S¹*, Belaz S², Decaux O¹, Robert-Gangneux F² and Revest M³

1Service d’hématologie Clinique, CHU de Rennes, Université de Rennes, Rennes, France

2Laboratoire de Parasitologie et Mycologie, CHU de Rennes, Université de Rennes, Rennes, France

3Service des Maladies Infectieuses, CHU de Rennes, Université de Rennes, Rennes, France

*Corresponding author: Samuel VIC, Service d’hématologie Clinique, CHU Rennes, 2 rue Henri Le Guilloux 35033 Rennes Cedex 9, France

Received: August 17, 2022; Accepted: September 16, 2022; Published: September 23, 2022


We report the case of a woman treated by rituximab and ibrutinib for a marginal-zone lymphoma, presenting diarrhea, dyspnea, retiform purpura, hemolytic anemia and hypereosinophilia revealing a disseminated strongyloidiasis. Diagnosis was done by stool parasitic examination, serology was negative. She was treated by ivermectin. All the symptoms disappeared, including hemolytic anemia. To our knowledge, this is the first report of a disseminated strongyloidiasis in a patient treated by ibrutinib. Hemolytic anemia was not immune-mediated, and possibly related to the infection as it regressed under anti-parasitic treatment.

Keywords: Strongyloidiasis; Lymphoma; Rituximab; Ibrutinib; Hemolytic anemia


ANCA: Anti-Neutrophil Cytoplasmic Antibodies; BTK: Bruton Tyrosine Kinase; CCP: Cyclic Citrullinated Peptide; CT: Computed Tomography; CRP: C-Reactive Protein; G6PD: Glucose-6-Phosphate Dehydrogenase; HTLV-1: Human T-Lymphotropic Virus 1; ICU: Intensive Care Unit; IV: Intravenously; LDH: Lactate Dehydrogenase; SC: Subcutaneously; WBC: White Blood Cell


Strongyloides stercoralis is an intestinal helminth that infects humans through contact with soil contaminated by human feces containing strongyloid larvae. The disease is widely distributed in tropical and subtropical regions, and it is estimated that 30 to 100 million people are infected worldwide [1]. Strongyloidiasis is asymptomatic in more than half of cases, where eosinophilia or a positive serologic test may be the only finding [2]. The parasite has a long-term persistence in the host leading to infection that could last for decades. While strongyloidiasis is mainly a chronic clinically inapparent infection in immunocompetent hosts, immunosuppressed patients are at risk of life-threatening hyperinfection syndrome due to uncontrolled rhabiditoid larvae production, and differentiation into strongyloid larvae, which can translocate through the gut. Larvae migration through tissues carries intestinal bacteria, leading to sepsis, and death in 60 to 70% of cases [3]. The main risk factor for strongyloidiasis hyperinfection is systemic corticosteroid use, but other immunosuppressed backgrounds such as infection by the Human T-Lymphotropic Virus 1 (HTLV-1), hematological malignancies, and transplantation are described [4]. Even if some studies suggested to screen and treat every immunosuppressed patient or candidate to immunosuppression if they may have been exposed to S. stercoralis [5], there are no guidelines for the prevention of infection in patients treated for hematologic diseases. It is commonly recommended to treat every patient coming from a high-risk area and scheduled to start a prolonged corticosteroid or another “highly immunosuppressive” therapy [6]. Data regarding other hematologic treatments, especially recent ones such as targeted therapies, are scarce, and there is no consensus on whether systematic screening or prophylactic treatment should be done. In this article, we report a case of disseminated strongyloidiasis in a patient treated by rituximab and ibrutinib for a lymphoma.

Case Description

The patient was a 70-year-old woman with a splenic marginalzone lymphoma diagnosed one year before, on November 2020. She was included in a clinical trial evaluating a combination of rituximab and ibrutinib: rituximab 375mg/m² Intravenously (IV) on day 1 and then 1400mg Subcutaneously (SC) on days 8-15-21 for the first cycle, then 1400mg SC every 28 days for the following cycles; and a daily administration of oral ibrutinib 560mg. She received a total of 5 cycles from February to July 2021. Treatment was efficient with a complete remission, and well tolerated. The last rituximab injection was performed on the 5th of July 2021, and then she continued to take daily ibrutinib at the same dose. She did not take any other immunosuppressive treatment.

She was admitted to the emergency department on the 21st of December 2021 for diarrhea, nausea and vomiting, lasting for 3 weeks. She presented severe asthenia with clinical signs of extracellular dehydration. She had no fever. Laboratory tests showed a normal electrolyte balance and renal function. C-Reactive Protein (CRP) was 9 mg/L. Hemoglobin level was 11.5 g/dL, platelets 308 G/L, White Blood Cell (WBC) count 11.5 G/L with a high neutrophil count at 8.2 G/L, and no eosinophil excess. An abdominal Computed Tomography (CT) scan was performed, showing uncomplicated colitis. Standard stool culture and search for Clostridium difficile toxins were negative. Levofloxacin was prescribed and ibrutinib was stopped, without any efficacy on diarrhea. Nine days after her admission, she developed abdominal retiform purpura (Figure 1) and dyspnea with desaturation requiring oxygen until 10L/min. Thoracic CT scan showed a diffuse infiltrative pneumonitis with ground-glass opacities and moderate bilateral pleural effusion (Figure 2). SARS-CoV-2 test by polymerase chain reaction was negative on nasopharyngeal swab, urinary antigen testing for Streptococcus pneumoniae and Legionella pneumophila were negative. Biological analysis showed an acute anemia with hemoglobin level at 7.6 g/dL. WBC count was 20 G/L with still a neutrophil excess (16 G/L) and normal eosinophil count (0.06 G/L). Levofloxacin was switched to ceftriaxone, she received 2 packed red blood cells and was transferred to the Intensive Care Unit (ICU).