EBV-positive diffuse large B cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 (APDS1): a case report and literature review

Activated phosphoinositide 3-kinase δ syndrome (APDS), an inborn error of immunity associated with gain-of-function mutations in the PIK3CD gene, is characterized by dysregulated PI3Kδ signaling. The clinical spectrum commonly includes recurrent respiratory infections and lymphoproliferative manifes...

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Main Authors: Qiu-yuan Xiang, Min Zuo, Ji-Hao Zhou, Chun Feng
Format: Article
Language:English
Published: Frontiers Media S.A. 2025-05-01
Series:Frontiers in Immunology
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Online Access:https://www.frontiersin.org/articles/10.3389/fimmu.2025.1583405/full
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author Qiu-yuan Xiang
Min Zuo
Ji-Hao Zhou
Chun Feng
author_facet Qiu-yuan Xiang
Min Zuo
Ji-Hao Zhou
Chun Feng
author_sort Qiu-yuan Xiang
collection DOAJ
description Activated phosphoinositide 3-kinase δ syndrome (APDS), an inborn error of immunity associated with gain-of-function mutations in the PIK3CD gene, is characterized by dysregulated PI3Kδ signaling. The clinical spectrum commonly includes recurrent respiratory infections and lymphoproliferative manifestations. We present an adolescent male with APDS1 manifesting recurrent sinopulmonary infections, generalized lymphadenopathy, hepatosplenomegaly, gastrointestinal manifestations, and combined T-cell/B-cell lymphopenia, complicated by Epstein-Barr virus-positive diffuse large B-cell lymphoma (EBV+ DLBCL). Whole-exome sequencing identified a heterozygous PIK3CD variant (c.3061G>A p.Glu1021Lys), supporting the molecular diagnosis of APDS1. This case adds to emerging evidence linking APDS1 with EBV-driven lymphomagenesis, thereby further supporting the critical role of PI3K δ pathway dysregulation in promoting EBV-associated lymphoid malignancies.
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institution DOAJ
issn 1664-3224
language English
publishDate 2025-05-01
publisher Frontiers Media S.A.
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series Frontiers in Immunology
spelling doaj-art-9dc62eeac1aa4e439e1361575579b26c2025-08-20T02:39:19ZengFrontiers Media S.A.Frontiers in Immunology1664-32242025-05-011610.3389/fimmu.2025.15834051583405EBV-positive diffuse large B cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 (APDS1): a case report and literature reviewQiu-yuan Xiang0Min Zuo1Ji-Hao Zhou2Chun Feng3Department of Hematology, The Second Clinical Medical College of Jinan University, Shenzhen, ChinaDepartment of Pathology, Shenzhen People’s Hospital (The Second Clinical Medical College of Jinan University; The First Affiliated Hospital of Southern University of Science and Technology), Shenzhen, ChinaDepartment of Hematology, Shenzhen People’s Hospital (The Second Clinical Medical College of Jinan University; The First Affiliated Hospital of Southern University of Science and Technology), Shenzhen, ChinaDepartment of Hematology, Shenzhen People’s Hospital (The Second Clinical Medical College of Jinan University; The First Affiliated Hospital of Southern University of Science and Technology), Shenzhen, ChinaActivated phosphoinositide 3-kinase δ syndrome (APDS), an inborn error of immunity associated with gain-of-function mutations in the PIK3CD gene, is characterized by dysregulated PI3Kδ signaling. The clinical spectrum commonly includes recurrent respiratory infections and lymphoproliferative manifestations. We present an adolescent male with APDS1 manifesting recurrent sinopulmonary infections, generalized lymphadenopathy, hepatosplenomegaly, gastrointestinal manifestations, and combined T-cell/B-cell lymphopenia, complicated by Epstein-Barr virus-positive diffuse large B-cell lymphoma (EBV+ DLBCL). Whole-exome sequencing identified a heterozygous PIK3CD variant (c.3061G>A p.Glu1021Lys), supporting the molecular diagnosis of APDS1. This case adds to emerging evidence linking APDS1 with EBV-driven lymphomagenesis, thereby further supporting the critical role of PI3K δ pathway dysregulation in promoting EBV-associated lymphoid malignancies.https://www.frontiersin.org/articles/10.3389/fimmu.2025.1583405/fullactivated phosphoinositide 3-kinase δ syndromeEpstein-Barr virusdiffuse large B-cell lymphomaEpstein-Barr virus-positive diffuse large B-cell lymphoma (EBV+ DLBCL)inborn error of immunity
spellingShingle Qiu-yuan Xiang
Min Zuo
Ji-Hao Zhou
Chun Feng
EBV-positive diffuse large B cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 (APDS1): a case report and literature review
Frontiers in Immunology
activated phosphoinositide 3-kinase δ syndrome
Epstein-Barr virus
diffuse large B-cell lymphoma
Epstein-Barr virus-positive diffuse large B-cell lymphoma (EBV+ DLBCL)
inborn error of immunity
title EBV-positive diffuse large B cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 (APDS1): a case report and literature review
title_full EBV-positive diffuse large B cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 (APDS1): a case report and literature review
title_fullStr EBV-positive diffuse large B cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 (APDS1): a case report and literature review
title_full_unstemmed EBV-positive diffuse large B cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 (APDS1): a case report and literature review
title_short EBV-positive diffuse large B cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 (APDS1): a case report and literature review
title_sort ebv positive diffuse large b cell lymphoma secondary to activated phosphoinositide 3 kinase δ syndrome type 1 apds1 a case report and literature review
topic activated phosphoinositide 3-kinase δ syndrome
Epstein-Barr virus
diffuse large B-cell lymphoma
Epstein-Barr virus-positive diffuse large B-cell lymphoma (EBV+ DLBCL)
inborn error of immunity
url https://www.frontiersin.org/articles/10.3389/fimmu.2025.1583405/full
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