Hemolytic anemias (HAs) encompasses a heterogeneous group of disorders with either congenital or acquired etiologies. We present a complex case of a 27-year-old woman with hemolytic anemia of multifactorial origin, involving both inherited RBC membrane defects and multiple autoimmune comorbidities. Genetic testing identified heterozygous variants in SPTA1 and SBDS, consistent with carrier status for hereditary elliptocytosis and Shwachman–Diamond syndrome. The patient was also diagnosed with Caspr2-positive Isaacs syndrome, systemic lupus erythematosus, seronegative antiphospholipid syndrome, and anti-aquaporin-4 antibody-positive optic neuritis. Despite extensive immunosuppressive and immunotherapic treatment and splenectomy, the clinical course was marked by recurrent hemolytic crises, thrombotic complications, and progressive neurological involvement, ultimately leading to death. Our experience highlights the challenges posed by the diagnosis and management of HAs, underlining the relevance of a multidisciplinary and personalized approach.

An Unusual Case of Multifactorial Hemolytic Anemia: A Complex Interaction Between Genetic and Autoimmune Factors / Biglietto, M., Peluso, G., Luise, C., Tripi, D., Conforti, M.F., Filipponi, V., Bizzoni, L., Trasarti, S.. - In: HEMATO. - ISSN 2673-6357. - (2025). [10.3390/hemato6020015]

An Unusual Case of Multifactorial Hemolytic Anemia: A Complex Interaction Between Genetic and Autoimmune Factors

Mario Biglietto
Primo
;
Cristina Luise;Diletta Tripi;Maria Francesca Conforti;Valeria Filipponi;Luisa Bizzoni;
2025

Abstract

Hemolytic anemias (HAs) encompasses a heterogeneous group of disorders with either congenital or acquired etiologies. We present a complex case of a 27-year-old woman with hemolytic anemia of multifactorial origin, involving both inherited RBC membrane defects and multiple autoimmune comorbidities. Genetic testing identified heterozygous variants in SPTA1 and SBDS, consistent with carrier status for hereditary elliptocytosis and Shwachman–Diamond syndrome. The patient was also diagnosed with Caspr2-positive Isaacs syndrome, systemic lupus erythematosus, seronegative antiphospholipid syndrome, and anti-aquaporin-4 antibody-positive optic neuritis. Despite extensive immunosuppressive and immunotherapic treatment and splenectomy, the clinical course was marked by recurrent hemolytic crises, thrombotic complications, and progressive neurological involvement, ultimately leading to death. Our experience highlights the challenges posed by the diagnosis and management of HAs, underlining the relevance of a multidisciplinary and personalized approach.
2025
diagnosis; anemia; Isaacs syndrome; autoimmunity
01 Pubblicazione su rivista::01i Case report
An Unusual Case of Multifactorial Hemolytic Anemia: A Complex Interaction Between Genetic and Autoimmune Factors / Biglietto, M., Peluso, G., Luise, C., Tripi, D., Conforti, M.F., Filipponi, V., Bizzoni, L., Trasarti, S.. - In: HEMATO. - ISSN 2673-6357. - (2025). [10.3390/hemato6020015]
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11573/1774650
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