Severe refractory autoimmune hemolytic anemia with five-year complete hematologic response to third course of treatment with rituximab: a case report

Abstract

IntroductionRituximab is an emerging treatment for autoimmune hemolytic anemia. We report the case of a patient with a five-year complete hematologic response to a third course of treatment with rituximab. Cases of response to rituximab re-treatments have been reported, but none to our knowledge that failed multiple prior treatments and achieved as durable a response.Case presentationA 45-year-old Hispanic man presented at age 26 with darkening urine and cold intolerance. His blood tests revealed elevated lactic dehydrogenase and bilirubin, a hemoglobin level of 7.4g/dL, and a positive Coombs test for complement C3 and immunoglobulin G antibody. A diagnosis of autoimmune hemolytic anemia was made. After failing multiple therapies including prednisone, splenectomy, immunoglobulin, cyclosporine, danocrine and azathioprine, our patient was treated with a four-week course of rituximab at a dose of 375mg/m2 weekly, 10 years following initial presentation. He achieved a rapid and complete hematologic response that lasted 25 months. Re-treatment with the same course of rituximab prompted a second response that lasted 18 months. A third re-treatment has achieved an ongoing five-year complete hematologic response.ConclusionsThis is an unusual case of a durable five-year remission of autoimmune hemolytic anemia with rituximab re-treatment following relapse after two prior courses of rituximab and despite the persistence of immunoglobulin G and complement-coated red blood cells. No mechanistic explanations for improved response to rituximab re-treatment in autoimmune hemolytic anemia have been reported in the literature. Future studies of rituximab or other B cell-targeting antibodies in the treatment of autoimmune hemolytic anemia should explore autoantibody immunoglobulin G subclass switching and alterations in complement inhibitory proteins on red blood cell membranes as potential correlates of hematologic response.

Efficacy and Safety of Rituximab Therapy for Lupus Nephritis Among SLE Female Patients; a Retrospective Hospital-Based Study

“…Similar findings were reported by some authors who studied the effects of RTX in refractory autoimmune hemolytic anemia. 22 The albumin creatinine ratio in this study was statistically decreased after 6 months of Rituximab use, leading to reducing of the median urinary protein creatinine ratio from 446 to 190 mg/mmol. Other studies showed similar findings (80).…”

“…Similar findings were reported by some authors who studied the effects of RTX in refractory autoimmune hemolytic anemia. 22 …”

Rituximab: Is it Possible to Link Both Autoimmune Haemolytic Anemia and Aplastic Anemia Regarding the Etiology and Management?

“…In the past few years, rituximab has been recognized as a safe and effective emerging treatment for autoimmune hemolytic anemia (AIHA) [1,2]. Rituximab was also considered as a preferred secondline therapy of warm antibody hemolytic anemia in adults in some major European centers and it was shown that second-line treatment with rituximab led to response rates similar to splenectomy (approximately 70%) [3].…”