Refractory Autoimmune Hemolytic Anemia in a Child Resolved After Benign Ovarian Tumor Resection: A Case Report

Autoimmune hemolytic anemia (AIHA) is a rare immune-mediated disorder in children that can present as primary or secondary to other diseases.Here, we report an unusual case of steroid-refractory warm AIHA in an 11-year-old girl whose condition was ultimately cured after removal of a benign ovarian tumor.Despite receiving multiple lines of therapy-including corticosteroids, rituximab, cyclosporine, sirolimus, and mycophenolate mofetil-the patient experienced recurrent hemolysis and steroid dependence for nearly four years.Abdominopelvic imaging performed to evaluate fever revealed bilateral ovarian cystic lesions, including a left-sided dermoid cyst.Surgical resection of the tumor led to complete and sustained hematologic remission, with normalization of hemoglobin, bilirubin, and reticulocyte counts, allowing discontinuation of all immunosuppressive agents.No recurrence of hemolysis was observed during 18 months of follow-up.This case highlights the potential for benign ovarian tumors to act as a rare secondary cause of AIHA through paraneoplastic or immune cross-reactive mechanisms.Awareness of such associations is crucial when evaluating pediatric patients with refractory or relapsing AIHA, as identification and removal of an occult tumor may achieve definitive resolution of hemolysis and avoid long-term immunosuppression.

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Imsang soa hyeoraek jong-yang/Imsang soa hyeol'aeg jong'yang/Clinical pediatric hematology-oncology
Published
2026-04-30
DOI
https://doi.org/10.15264/cpho.2026.33.1.29
Primary Topic
Blood groups and transfusion
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article
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article

Refractory Autoimmune Hemolytic Anemia in a Child Resolved After Benign Ovarian Tumor Resection: A Case Report

Kyung‐Nam Koh, Hyeonjoon Kim, Kyung Duk Park, Su Hyun Yoon et al.
Imsang soa hyeoraek jong-yang/Imsang soa hyeol'aeg jong'yang/Clinical pediatric hematology-oncology
Blood groups and transfusion
article

Refractory Autoimmune Hemolytic Anemia in a Child Resolved After Benign Ovarian Tumor Resection: A Case Report

Kyung‐Nam Koh, Hyeonjoon Kim, Kyung Duk Park, Su Hyun Yoon, Hyery Kim, Sung Han Kang, Ho Joon Im, Dae Yeon Kim
article en

Abstract

Autoimmune hemolytic anemia (AIHA) is a rare immune-mediated disorder in children that can present as primary or secondary to other diseases.Here, we report an unusual case of steroid-refractory warm AIHA in an 11-year-old girl whose condition was ultimately cured after removal of a benign ovarian tumor.Despite receiving multiple lines of therapy-including corticosteroids, rituximab, cyclosporine, sirolimus, and mycophenolate mofetil-the patient experienced recurrent hemolysis and steroid dependence for nearly four years.Abdominopelvic imaging performed to evaluate fever revealed bilateral ovarian cystic lesions, including a left-sided dermoid cyst.Surgical resection of the tumor led to complete and sustained hematologic remission, with normalization of hemoglobin, bilirubin, and reticulocyte counts, allowing discontinuation of all immunosuppressive agents.No recurrence of hemolysis was observed during 18 months of follow-up.This case highlights the potential for benign ovarian tumors to act as a rare secondary cause of AIHA through paraneoplastic or immune cross-reactive mechanisms.Awareness of such associations is crucial when evaluating pediatric patients with refractory or relapsing AIHA, as identification and removal of an occult tumor may achieve definitive resolution of hemolysis and avoid long-term immunosuppression.

Imsang soa hyeoraek jong-yang/Imsang soa hyeol'aeg jong'yang/Clinical pediatric hematology-oncologyVol. 33(1)
Ulsan College (KR), Asan Medical Center (KR), University of Ulsan (KR), Jeonbuk National University Hospital (KR), Chungbuk National University Hospital (KR)
Good health and well-being
Openalex Percentile: Top 6%
Blood groups and transfusion
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