Case Report: Rotavirus Vaccination and Severe Combined Immunodeficiency in Japan.

Kay Tanita,Tomohiro Morio,Masafumi Yamada,Takahiro Tomoda,Masataka Ishimura,Tetsushi Yoshikawa,Kohsuke Imai,Norimasa Tada,Noriko Mitsuiki,Kento Inoue,Yoshiki Kawamura,Hideki Muramatsu,Akihiro Iguchi,Taro Yoshida,Hirokazu Kanegane,Motohiro Kato,Hiroki Miura,Toshihiro Matsui,Katsuhide Eguchi,Shouichi Ohga

doi:10.3389/fimmu.2022.786375

Abstract

Severe combined immunodeficiency (SCID) is an inborn error of immunity that occurs in approximately 1 in 50,000 births, mainly due to impaired lymphocyte differentiation. Without curative treatment, such as hematopoietic cell transplantation (HCT) or gene therapy, severe infection in the first year of life could make this condition fatal. The results of HCT are poor when patients have active infections, thus requiring early diagnosis before onset of infection. In five cases of SCID diagnosed in Japan, the oral rotavirus vaccine had been administered before diagnosis. In this study, we demonstrated that the rotavirus from their stools was a vaccine-derived strain. In some cases, severe gastroenteritis triggered the diagnosis of SCID. However, newborn screening for SCID is available before the first rotavirus vaccination using assays for the detection of T-cell receptor excision circles (TRECs). Therefore, to improve the prognosis of patients with SCID in Japan, we should establish a screening system of TRECs for newborns throughout Japan.

Highlights

Severe combined immunodeficiency (SCID) is a critical inborn error of immunity (IEI) that causes cellular and humoral immunity failure due to impaired lymphocyte differentiation, resulting in severe infections from infancy
Live vaccination is contraindicated [1], patients with SCID often remain asymptomatic until early infancy and are rarely diagnosed before 2 months of age when oral rotavirus vaccination is initiated
DNA was amplified by polymerase chain reaction (PCR) using primers for human rotavirus A gene 10, which encodes the rotavirus Non-structural protein 4 (NSP4) gene, and sequenced using the Sanger method

Summary

Introduction

Severe combined immunodeficiency (SCID) is a critical inborn error of immunity (IEI) that causes cellular and humoral immunity failure due to impaired lymphocyte differentiation, resulting in severe infections from infancy. Mild cases can resolve spontaneously, some cases can cause fatal dehydration and encephalitis or Rotavirus and SCID encephalopathy, leading to the hospitalization of many patients. Live vaccination is contraindicated [1], patients with SCID often remain asymptomatic until early infancy and are rarely diagnosed before 2 months of age when oral rotavirus vaccination is initiated. Rotarix®, a live, monovalent, attenuated, human rotavirus vaccine (RV1), and RotaTeq®, a live, pentavalent, human–bovine reassortant rotavirus vaccine (RV5), were initiated as arbitrary vaccination in 2011 and 2012, respectively, in Japan. Regular administration of these vaccines was initiated in October 2020. This initiative was taken after realizing that the number of cases vaccinated before the diagnosis of SCID will increase

Methods

Results

Conclusion