N Engl J Med 2021 01;384(1):42-50
From the Departments of Pediatric Oncology (A.A., T. Kumamoto, M.N., C.O.), Laboratory Medicine (T. Kubo, K. Sunami, H.K.), Diagnostic Pathology (N.M., H.Y.), Breast and Medical Oncology (K. Yonemori, E.N.), Pediatric Surgical Oncology (T.H., N.K.), and Experimental Therapeutics (N. Yamamoto), National Cancer Center Hospital, the Departments of Clinical Genomics (H.I., T. Kubo) and Immune Medicine (K.A.), and the Divisions of Genome Biology (K. Shiraishi, T. Kohno), Cancer Genomics (Y.A., T.S.), and Cancer Immunology (Y.T., H.N.), National Cancer Center Research Institute, the Division of Translational Genomics, Exploratory Oncology Research and Clinical Trial Center (H.I., T. Kubo, T. Kohno), the Children's Cancer Center, National Center for Child Health and Development (T.H.), the Departments of Obstetrics and Gynecology (T. Kuroda, K. Yamada, N. Yanaihara, K. Takahashi, A.O.) and Pathology (T. Kiyokawa), Jikei University School of Medicine, the Departments of Pediatrics (S.H., D.H., A.M.) and Integrated Women's Health (K.O.), St. Luke's International Hospital, and the Department of Pediatrics, Toho University School of Medicine (M.M.), Tokyo, and the Department of Pediatrics, Hokkaido University Graduate School of Medicine, Sapporo (S.H., A.M.) - both in Japan.
Two cases of pediatric lung cancer (in 23-month-old and 6-year-old boys) resulting from mother-to-infant transmission of uterine cervical tumors were incidentally detected during routine next-generation sequencing of paired samples of tumor and normal tissue. Spontaneous regression of some lesions in the first child and slow growth of the tumor mass in the second child suggested the existence of alloimmune responses against the transmitted tumors. Immune checkpoint inhibitor therapy with nivolumab led to a strong regression of all remaining tumors in the first child. (Funded by the Japan Agency for Medical Research and Development and others; TOP-GEAR UMIN Clinical Trials Registry number, UMIN000011141.).