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Coombs negative autoimmune hemolytic anemia in Crohn's disease
Bong Soo Park1, Sihyung Park1, Kyubok Jin1
1Department of Internal Medicine, Haeundae Paik Hospital, Inje University College of Medicine, Busan, Korea.
Insights
Autoimmune hemolytic anemia (AIHA) is rare in Crohn's disease patients. This case highlights a rare Coombs-negative AIHA diagnosis in a Crohn's patient, identified by elevated red blood cell-bound immunoglobulin G (RBC-IgG) and successfully treated with steroids.
Area of Science:
- Gastroenterology
- Hematology
- Immunology
Background:
- Anemia is a frequent complication of Crohn's disease (CD), with iron deficiency and anemia of chronic disease being most common.
- Autoimmune hemolytic anemia (AIHA), particularly the Coombs-negative variant, is an exceptionally rare form of anemia in CD patients.
Observation:
- A 41-year-old woman with well-controlled Crohn's disease presented with symptoms suggestive of hemolysis, including dark urine, dizziness, and shortness of breath.
- Laboratory findings revealed elevated bilirubin and lactate dehydrogenase (LDH), with undetectable haptoglobin, indicative of hemolysis.
- Direct and indirect Coombs tests were negative, prompting further investigation for Coombs-negative AIHA.
Findings:
- Red blood cell-bound immunoglobulin G (RBC-IgG) levels were significantly elevated (352 IgG molecules/cell vs. cut-off 78.5 IgG molecules/cell), confirming Coombs-negative AIHA.
- The patient's Crohn's disease activity index (CDAI) was below 100, indicating the anemia was not directly related to active disease flares.
Implications:
- This case underscores the importance of considering Coombs-negative AIHA in Crohn's disease patients presenting with unexplained hemolytic anemia, even with controlled disease activity.
- Diagnosis via RBC-IgG measurement is crucial for identifying this rare condition.
- Steroid therapy proved effective in managing this rare presentation of AIHA in a Crohn's disease patient.
Background:
Anemia is a common, important extraintestinal complication of Crohn's disease. The main types of anemia in patients with Crohn's disease are iron deficiency anemia and anemia of chronic disease. Although patients with Crohn's disease may experience various type of anemia, autoimmune hemolytic anemia (AIHA) in patients with Crohn's disease, especially Coombs-negative AIHA, is very rare.
Case Report:
A 41-year-old woman with Crohn's disease presented to our emergency room (ER) with dark urine, dizziness, and shortness of breath. The activity of Crohn's disease had been controlled, with Crohn's disease activity index (CDAI) score below 100 point. On physical examination, the patient had pale conjunctivae and mildly icteric sclerae. Serum bilirubin was raised at 3.1 mg/dL, lactate dehydrogenase (LDH) level was 1418 U/L and the haptoglobin level was <3 mg/dL. Results of direct and the indirect Coombs tests were all negative. We then measured the RBC-IgG to evaluate the possibility of Coombs-negative AIHA. The result revealed that RBC-IgG level was 352 IgG molecules/cell, with the cut-off value at 78.5 IgG molecules/cell.
Conclusions:
We report a case of Coombs-negative AIHA in a patient with Crohn's disease with chronic anemia, diagnosed by red blood cell-bound immunoglobulin G (RBC-IgG) and treated with steroids therapy.
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