A rare case report of autoimmune haemolytic anemia in a female child due to a Donath-Landsteiner antibody

Radonjić, Zorica; Andric, Bogdanka; Šerbić, O.; Mićić, Dragan; Kuzmanović, Miloš; Jovanovic-Srzentic, Snezana; Dinić, R.

doi:10.1016/j.tracli.2020.03.001

Cited by 4 publications

(2 citation statements)

References 15 publications

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“…Paroxysmal cold hemoglobinuria (PCH) is a rare autoimmune hemolytic anemia that is defined by the destruction of red blood cells in the blood vessels due to the activation of the complement system [2,3,[94][95][96][97][98][99][100][101][102][103][104]. This destruction happens when the body is exposed to cold temperatures [3,12,100].…”

Section: Paroxysmal Cold Hemoglobinuria or Donath-landsteiner Test-po...mentioning

confidence: 99%

“…PCH was first documented in 1904 by Julius Donath and Karl Landsteiner, establishing it as one of the earliest recognized kinds of AIHA [3]. The illness may present either as a sudden, non-recurring post-infectious event in children [3,12,21,94,[96][97][98][99][100][101][102] or as recurring occurrences in adults with blood cancers or advanced syphilis [3,20,95,96]. Historically, PCH manifested primarily in patients having syphilis, and exposure to cold resulted in paroxysms of hemoglobinuria.…”

Section: Paroxysmal Cold Hemoglobinuria or Donath-landsteiner Test-po...mentioning

confidence: 99%

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Autoimmune Hemolytic Anemias: Classifications, Pathophysiology, Diagnoses and Management

Loriamini,

Cserti-Gazdewich,

Branch

2024

IJMS

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: Autoimmune hemolytic anemias (AIHAs) are conditions involving the production of antibodies against one’s own red blood cells (RBCs). These can be primary with unknown cause or secondary (by association with diseases or infections). There are several different categories of AIHAs recognized according to their features in the direct antiglobulin test (DAT). (1) Warm-antibody AIHA (wAIHA) exhibits a pan-reactive IgG autoantibody recognizing a portion of band 3 (wherein the DAT may be positive with IgG, C3d or both). Treatment involves glucocorticoids and steroid-sparing agents and may consider IVIG or monoclonal antibodies to CD20, CD38 or C1q. (2) Cold-antibody AIHA due to IgMs range from cold agglutinin syndrome (CAS) to cold agglutin disease (CAD). These are typically specific to the Ii blood group system, with the former (CAS) being polyclonal and the latter (CAD) being a more severe and monoclonal entity. The DAT in either case is positive only with C3d. Foundationally, the patient is kept warm, though treatment for significant complement-related outcomes may, therefore, capitalize on monoclonal options against C1q or C5. (3) Mixed AIHA, also called combined cold and warm AIHA, has a DAT positive for both IgG and C3d, with treatment approaches inclusive of those appropriate for wAIHA and cold AIHA. (4) Paroxysmal cold hemoglobinuria (PCH), also termed Donath–Landsteiner test-positive AIHA, has a DAT positive only for C3d, driven upstream by a biphasic cold-reactive IgG antibody recruiting complement. Although usually self-remitting, management may consider monoclonal antibodies to C1q or C5. (5) Direct antiglobulin test-negative AIHA (DAT-neg AIHA), due to IgG antibody below detection thresholds in the DAT, or by non-detected IgM or IgA antibodies, is managed as wAIHA. (6) Drug-induced immune hemolytic anemia (DIIHA) appears as wAIHA with DAT IgG and/or C3d. Some cases may resolve after ceasing the instigating drug. (7) Passenger lymphocyte syndrome, found after transplantation, is caused by B-cells transferred from an antigen-negative donor whose antibodies react with a recipient who produces antigen-positive RBCs. This comprehensive review will discuss in detail each of these AIHAs and provide information on diagnosis, pathophysiology and treatment modalities.

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Section: Paroxysmal Cold Hemoglobinuria or Donath-landsteiner Test-po...mentioning

confidence: 99%

Section: Paroxysmal Cold Hemoglobinuria or Donath-landsteiner Test-po...mentioning

confidence: 99%

Autoimmune Hemolytic Anemias: Classifications, Pathophysiology, Diagnoses and Management

Loriamini,

Cserti-Gazdewich,

Branch

2024

IJMS

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Acute paroxysmal cold hemoglobinuria upon dengue fever: A case report

Basit,

Anamika,

Ashrafunnisa

et al. 2024

Journal of Acute Disease

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Rationale: Dengue fever is capable of inciting the formation of transient polyclonal antibodies directed at red blood cell antigens, resulting in complement-mediated hemolysis, leading to intravascular hemolysis and hemoglobinuria. Patient’s concern: A 12-year-old male patient who recovered from dengue fever a week ago had red blood cell agglutination, spherocytes, and engulfment of red blood cells (erythrophagocytosis) by monocytes and neutrophils on routine hematological peripheral blood smear. The unexpected blood smear results prompted the lab physicians to investigate autoimmune hemolytic anemia, which revealed a monospecific positive direct antiglobulin test for complement (C3d, C3b) and the presence of Donath-Landsteiner antibody. Diagnosis: Paroxysmal cold hemoglobinuria (PCH), secondary to dengue fever. Interventions: Oxygen supplements, antibiotics, intravenous immunoglobulins, steroid therapy, and packed cell transfusions were administered. Outcomes: The patient’s condition was improved following the therapy. Lessons: Post-dengue PCH is a rare complication that requires a thorough peripheral smear examination for erythrophagocytosis, as advanced hematology analyzers fail to detect such findings.

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Donath-landsteiner hemolytic anemia with erythrophagocytosis and platelet phagocytosis in peripheral smear: Rare findings

Momin¹,

Aluri²,

Bhaskar³

2022

Med J DY Patil Vidyapeeth

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A rare case report of autoimmune haemolytic anemia in a female child due to a Donath-Landsteiner antibody

Cited by 4 publications

References 15 publications

Autoimmune Hemolytic Anemias: Classifications, Pathophysiology, Diagnoses and Management

Autoimmune Hemolytic Anemias: Classifications, Pathophysiology, Diagnoses and Management

Acute paroxysmal cold hemoglobinuria upon dengue fever: A case report

Donath-landsteiner hemolytic anemia with erythrophagocytosis and platelet phagocytosis in peripheral smear: Rare findings

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