OBJECTIVE: To present a case of brucellosis-induced severe autoimmune hemolytic anemia (AIHA) that was refractory to traditional corticosteroid treatment and eventually treated with rituximab apart from antibiotic therapy and to discuss the potential role of rituximab in similar cases of AIHA triggered by an underlying reversible cause. CASE SUMMARY: A 79-year-old woman was diagnosed with severe AIHA (reticulocyte count 21.5%, hemoglobin 6 g/dL). Initial treatment with prednisone in a regional hospital was not efficacious. Brucellosis was diagnosed by serology; the disease was further complicated by hepatic and splenic granulomatous involvement and sacral bone localization. Due to the severity of AIHA as demonstrated by reticulocyte count and hemoglobin levels, the initial unresponsiveness to corticosteroid therapy, the potential of the underlying infectious cause to relapse along with AIHA, and the localization of the pathogen in a focal site (bone involvement) that could act as a constant AIHA trigger, the patient was treated aggressively with rituximab, apart from the typical antimicrobial therapy. DISCUSSION: Brucellosis can induce autoimmunity and mimic primary hematologic diseases. We reviewed reports on the unique forms of Brucella-induced hemolysis available in the literature. Massive hemolysis, though, is rare, and in the case of a pathogen such as brucellosis, one cannot ignore the potential for infection relapse accompanied by hemolysis relapse. Cases refractory to corticosteroids are typically treated with invasive amputative procedures such as splenectomy. However, in cases where an underlying therapeutically reversible cause of infection can be identified, the proven short-term efficacy and safety profile of rituximab can be of significance. CONCLUSIONS: Novel therapeutic approaches with molecular agents such as rituximab may assist in treatment of considerably severe infectious pathogen-induced autoimmune hemolytic anemia that is refractory to first-line therapy.
OBJECTIVE: To present a case of brucellosis-induced severe autoimmune hemolytic anemia (AIHA) that was refractory to traditional corticosteroid treatment and eventually treated with rituximab apart from antibiotic therapy and to discuss the potential role of rituximab in similar cases of AIHA triggered by an underlying reversible cause. CASE SUMMARY: A 79-year-old woman was diagnosed with severe AIHA (reticulocyte count 21.5%, hemoglobin 6 g/dL). Initial treatment with prednisone in a regional hospital was not efficacious. Brucellosis was diagnosed by serology; the disease was further complicated by hepatic and splenic granulomatous involvement and sacral bone localization. Due to the severity of AIHA as demonstrated by reticulocyte count and hemoglobin levels, the initial unresponsiveness to corticosteroid therapy, the potential of the underlying infectious cause to relapse along with AIHA, and the localization of the pathogen in a focal site (bone involvement) that could act as a constant AIHA trigger, the patient was treated aggressively with rituximab, apart from the typical antimicrobial therapy. DISCUSSION: Brucellosis can induce autoimmunity and mimic primary hematologic diseases. We reviewed reports on the unique forms of Brucella-induced hemolysis available in the literature. Massive hemolysis, though, is rare, and in the case of a pathogen such as brucellosis, one cannot ignore the potential for infection relapse accompanied by hemolysis relapse. Cases refractory to corticosteroids are typically treated with invasive amputative procedures such as splenectomy. However, in cases where an underlying therapeutically reversible cause of infection can be identified, the proven short-term efficacy and safety profile of rituximab can be of significance. CONCLUSIONS: Novel therapeutic approaches with molecular agents such as rituximab may assist in treatment of considerably severe infectious pathogen-induced autoimmune hemolytic anemia that is refractory to first-line therapy.
Authors: Jerome S Harms; Mike Khan; Cherisse Hall; Gary A Splitter; E Jane Homan; Robert D Bremel; Judith A Smith Journal: Infect Immun Date: 2018-06-21 Impact factor: 3.441