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2023

ANUÁRIO DO HOSPITAL
DONA ESTEFÂNIA

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CASE REPORT: PRIMARY IMUNODEFICIENCIES, MASSIVE EBV+ T-CELL LINFOPROLIFERATION LEADING TO THE DIAGNOSIS OF ICF2 SYNDROME

Gonçalo Luzes Padeira1, Catarina Araújo2, Ana Isabel Cordeiro1, João Freixo3, Catarina Gregório Martins4,5 e João Farela Neves1,4,5

1 - Hospital de Dona Estefânia, Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal
2 - Departamento de Anatomia Patológica, Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal
3 - Centro de Genética Preditiva e Preventiva, Instituto de Biologia Molecular e Celular, Instituto de Investigação e Inovação em Saúde, Porto, Portugal
4 - CEDOC, Chronic Diseases Research Center, NOVA Medical School, Lisbon, Portugal
5 - Comprehensive Health Research Centre (CHRC), NOVA Medical School, Nova University of Lisbon, Lisbon, Portugal

Publicação em versão integral:
- Frontiers in Immunology
- doi: 10.3389/fimmu.2021.654167

Resumo: In immunocompromised patients, EBV may elicit B-cell transformation and proliferation. A 5-year-old microcephalic boy was admitted with fever and non-malignant polymorphic T-cell lymphoproliferative disease associated with EBV. A presumptive diagnosis of primary immunodeficiency with inability to control EBV was made and next-generation sequencing led to the identification of a novel ZBTB24 mutation (ICF2-syndrome). This case shows that susceptibility to EBV seems to be particular of ICF-2 as it has not been described in the other types of ICF. It is mandatory to raise the hypothesis of an underlying PID in case of severe EBV infection.

Palavras Chave: case report, EBV, ICF-2, lymphoproliferation, Primary immune deficiencies