A curious case of delayed hemolytic transfusion reaction with evanescent antibodies in a patient with hereditary hemorrhagic telangiectasia.
Journal
Transfusion
ISSN: 1537-2995
Titre abrégé: Transfusion
Pays: United States
ID NLM: 0417360
Informations de publication
Date de publication:
12 2019
12 2019
Historique:
received:
11
02
2019
revised:
04
09
2019
accepted:
13
09
2019
pubmed:
12
11
2019
medline:
17
6
2020
entrez:
12
11
2019
Statut:
ppublish
Résumé
Delayed hemolytic reactions are potential complications of incompatible transfusions and are usually associated with the identification of a new antibody on serologic studies, following a second immunization event. However, in rare cases, the antibody investigation remains negative even if the clinical presentation would lead one to suspect otherwise. A 44-year-old woman with hereditary hemorrhagic telangiectasia presented to the emergency department with hematuria and low back pain after she had received three units of RBCs 2 weeks earlier. Hematology and biochemistry results were consistent with delayed hemolytic transfusion reaction, but surprisingly, serologic antibody investigations were negative. It was only when her plasma was tested with enzyme (ficin)-treated panel cells that anti-e was finally detected, with a 3+ reaction with all homozygous e+ cells. No reaction was seen with heterozygous e+ cells. Four months later, an anti-K was also detected on standard panels, while the anti-e remained detectable only with ficin-treated panel cells. Three years later, both antibodies had vanished and remained undetectable. The weakness of anti-e reaction, combined with the quick evanescence of both antibodies led to the suspicion of a potential underlying immunodeficiency disorder, which was confirmed by low immunoglobulin levels on two occasions. To our knowledge, this is the first case of immunodeficiency disorder diagnosed after the identification of evanescent antibody reactions. This case also outlines the importance of a good clinical history that should lead to further investigations when a hemolytic transfusion reaction is suspected.
Sections du résumé
BACKGROUND
Delayed hemolytic reactions are potential complications of incompatible transfusions and are usually associated with the identification of a new antibody on serologic studies, following a second immunization event. However, in rare cases, the antibody investigation remains negative even if the clinical presentation would lead one to suspect otherwise.
CASE REPORT
A 44-year-old woman with hereditary hemorrhagic telangiectasia presented to the emergency department with hematuria and low back pain after she had received three units of RBCs 2 weeks earlier. Hematology and biochemistry results were consistent with delayed hemolytic transfusion reaction, but surprisingly, serologic antibody investigations were negative. It was only when her plasma was tested with enzyme (ficin)-treated panel cells that anti-e was finally detected, with a 3+ reaction with all homozygous e+ cells. No reaction was seen with heterozygous e+ cells. Four months later, an anti-K was also detected on standard panels, while the anti-e remained detectable only with ficin-treated panel cells. Three years later, both antibodies had vanished and remained undetectable. The weakness of anti-e reaction, combined with the quick evanescence of both antibodies led to the suspicion of a potential underlying immunodeficiency disorder, which was confirmed by low immunoglobulin levels on two occasions.
CONCLUSION
To our knowledge, this is the first case of immunodeficiency disorder diagnosed after the identification of evanescent antibody reactions. This case also outlines the importance of a good clinical history that should lead to further investigations when a hemolytic transfusion reaction is suspected.
Substances chimiques
Antibodies
0
Immunoglobulins
0
Ficain
EC 3.4.22.3
Types de publication
Case Reports
Langues
eng
Sous-ensembles de citation
IM
Pagination
3570-3574Informations de copyright
© 2019 AABB.
Références
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