Cytomegalovirus Laryngitis in Primary Combined Immunodeficiency Diseases
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LETTER TO EDITOR
Cytomegalovirus Laryngitis in Primary Combined Immunodeficiency Diseases Maiko Inoue 1 & Takeshi Isoda 1 & Motoi Yamashita 1 & Takahiro Tomoda 1 & Kento Inoue 1 & Tsubasa Okano 1 & Teppei Ohkawa 1 & Akifumi Endo 2 & Noriko Mitsuiki 1 & Takahiro Kamiya 2 & Masakatsu Yanagimachi 1 & Kouhei Yamamoto 3 & Yuichiro Inaba 4 & Toru Sasaki 5 & Masatoshi Takagi 1 & Hirokazu Kanegane 6 & Kohsuke Imai 7 & Tomohiro Morio 1 Received: 28 April 2020 / Accepted: 21 September 2020 # Springer Science+Business Media, LLC, part of Springer Nature 2020
Abbreviations AIDS Acquired immune deficiency syndrome Akt Protein kinase B APDS Activated phosphatidylinositol-3-kinase-delta syndrome BMT Bone marrow transplantation CID Combined immunodeficiency disease CMCD Chronic mucocutaneous candidiasis disease CMV Cytomegalovirus EBV Epstein–Barr virus FOXO1 Forkhead box protein O1 FTT Failure to thrive GCV Ganciclovir GOF Gain of function HIV Human immunodeficiency virus HPV Human papillomavirus LN Lymph node mTOR Mammalian target of rapamycin PFA Foscarnet PID Primary immunodeficiency PSL Prednisolone PIK3R1 Phosphoinositide 3-kinase regulatory subunit 1 SCID Severe combined immunodeficiency SCT Stem cell transplantation sjKREC Signal joint kappa-deleting recombination excision circles sjTREC Signal joint T cell receptor excision circles SOT Solid organ transplantation Electronic supplementary material The online version of this article (https://doi.org/10.1007/s10875-020-00873-9) contains supplementary material, which is available to authorized users. * Takeshi Isoda [email protected] Extended author information available on the last page of the article
STAT1 URCBT VGCV WAS
Signal transducer and activator of transcription 1 Unrelated cord blood transplantation Valganciclovir Wiskott–Aldrich syndrome
To the Editor: Cytomegalovirus (CMV) causes invasive diseases through congenital and neonatal infections in fetuses and neonates, as well as pneumonitis, hemorrhagic retinal necrosis, hepatitis, gastroenteritis, and encephalitis in immunocompromised individuals [1–3]. Reduced CD4+ T cell count is a risk factor for CMV disease in HIV-infected patients [4]. CMV-specific CD8+ T cell clones reconstitute cellular immunity against CMV after stem cell transplantation (SCT) [5]. Maternal humoral immunity contributes to prevention of congenital CMV infection [6]. Patients with cytotoxic T cell defects and reduced immunoglobulin production, including those with severe combined immunodeficiency (SCID), are susceptible to gamma-herpes virus infection [7, 8]. CMV laryngitis has been sporadically reported in immunocompromised patients after solid organ transplantation (SOT) and/or hematopoietic SCT and in those with acquired immunodeficiency diseases (Table S1). The reports suggested that CMV may infect the larynx and cause persistent inflammation in patients with combined immunodeficiency diseases (CIDs). We retrospectively collected patients who were diagnosed with possible-toproven CMV laryngitis between January 2000 and December 2019
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