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Review
. 2020 Nov;42(8):e745-e749.
doi: 10.1097/MPH.0000000000001599.

Generalized Verrucosis Revealing a Life-Threatening and Unlabeled T-Cell Lymphopenia Associated With Autoimmune Hemolytic Anemia: A Case Report and Review of Literature

Magali Marxgut  1 , Dalila Adjaoud  2 , Nathalie Aladjidi  3 , Corinne Armari  2 , Léa Marxgut  4 , Amandine Rubio  1   4   5   6
Affiliations
Review

Generalized Verrucosis Revealing a Life-Threatening and Unlabeled T-Cell Lymphopenia Associated With Autoimmune Hemolytic Anemia: A Case Report and Review of Literature

Magali Marxgut et al. J Pediatr Hematol Oncol. 2020 Nov.

Abstract

Primary immunodeficiencies are inherited disorders, which may be revealed in the context of autoimmune hemolytic anemia (AIHA). We report the case of a girl presenting with an enterovirus-related AIHA. Despite being in complete remission for her anemia after treatment, the initial CD4/CD8 lymphopenia dramatically worsened with time. Its sole clinical presentation was generalized verrucosis. Cellular quantitative and functional immunodeficiency was evidenced but no known molecular defect was identified despite extensive workup. This unlabeled profound naive T-lymphopenia was cured by bone marrow transplantation. No similar case was ever described in the scientific literature. Patients with AIHA and/or generalized verrucosis should be screened for primary immunodeficiency, before initiating any immunomodulatory treatment.

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References

    1. Mahlaoui N, Jais J-P, Brosselin P, et al. Prevalence of primary immunodeficiencies in France is underestimated. J Allergy Clin Immunol. 2017;140:1731–1733.
    1. Picard C, Al-Herz W, Bousfiha A, et al. Primary immunodeficiency diseases: an update on the classification from the International Union of Immunological Societies Expert Committee for Primary Immunodeficiency 2015. J Clin Immunol. 2015;35:696–726.
    1. Sri JC, Dubina MI, Kao GF, et al. Generalized verrucosis: a review of the associated diseases, evaluation and treatments. J Am Acad Dermatol. 2011;66:292–311.
    1. Aladjidi N, Leverger G, Leblanc T, et al. New insights into childhood autoimmune hemolytic anemia: a French national observational study of 265 children. Haematologica. 2011;96:655–663.
    1. Fischer A, Provot J, Jais J-P, et al. Members of the CEREDIH French PID Study Group. Autoimmune and inflammatory manifestations occur frequently in patients with primary immunodeficiencies. J Allergy Clin Immunol. 2017;140:1388–1393.