Peripheral blood lymphocyte telomere length as a predictor of response to immunosuppressive therapy in childhood aplastic anemia
Hirotoshi Sakaguchi,
Nobuhiro Nishio,
Asahito Hama,
Nozomu Kawashima,
Xinan Wang,
Atsushi Narita,
Sayoko Doisaki,
Yinyan Xu,
Hideki Muramatsu,
Nao Yoshida,
Yoshiyuki Takahashi,
Kazuko Kudo,
Hiroshi Moritake,
Kazuhiro Nakamura,
Ryoji Kobayashi,
Etsuro Ito,
Hiromasa Yabe,
Shouichi Ohga,
Akira Ohara,
Seiji Kojima
Affiliations
Hirotoshi Sakaguchi
Department of Pediatrics, Nagoya University Graduate School of Medicine;Division of Hematology and Oncology, Children’s Medical Center, Japanese Red Cross Nagoya 1st Hospital
Nobuhiro Nishio
Department of Pediatrics, Nagoya University Graduate School of Medicine
Asahito Hama
Department of Pediatrics, Nagoya University Graduate School of Medicine
Nozomu Kawashima
Department of Pediatrics, Nagoya University Graduate School of Medicine
Xinan Wang
Department of Pediatrics, Nagoya University Graduate School of Medicine
Atsushi Narita
Department of Pediatrics, Nagoya University Graduate School of Medicine
Sayoko Doisaki
Department of Pediatrics, Nagoya University Graduate School of Medicine
Yinyan Xu
Department of Pediatrics, Nagoya University Graduate School of Medicine
Hideki Muramatsu
Department of Pediatrics, Nagoya University Graduate School of Medicine
Nao Yoshida
Division of Hematology and Oncology, Children’s Medical Center, Japanese Red Cross Nagoya 1st Hospital
Yoshiyuki Takahashi
Department of Pediatrics, Nagoya University Graduate School of Medicine
Kazuko Kudo
Division of Hematology and Oncology, Shizuoka Children’s Hospital
Hiroshi Moritake
Division of Pediatrics, Department of Reproductive and Developmental Medicine, Faculty of Medicine, University of Miyazaki
Kazuhiro Nakamura
Department of Pediatrics, Hiroshima University Graduate School of Biomedical and Health Sciences
Ryoji Kobayashi
Department of Pediatrics, Sapporo Hokuyu Hospital
Etsuro Ito
Department of Pediatrics, Hirosaki University Graduate School of Medicine
Hiromasa Yabe
Department of Cell Transplantation and Regenerative Medicine, Tokai University School of Medicine, Isehara
Shouichi Ohga
Department of Perinatal and Pediatric Medicine, Graduate School of Medical Sciences, Kyushu University, Fukuoka
Akira Ohara
Department of Pediatrics, Toho University School of Medicine, Tokyo, Japan
Seiji Kojima
Department of Pediatrics, Nagoya University Graduate School of Medicine
Predicting the response to immunosuppressive therapy could provide useful information to help the clinician define treatment strategies for patients with aplastic anemia. In our current study, we evaluated the relationship between telomere length of lymphocytes at diagnosis and the response to immunosuppressive therapy in 64 children with aplastic anemia, using flow fluorescence in situ hybridization. Median age of patients was ten years (range 1.5–16.2 years). Severity of the disease was classified as very severe in 23, severe in 21, and moderate in 20 patients. All patients were enrolled in multicenter studies using antithymocyte globulin and cyclosporine. The response rate to immunosuppressive therapy at six months was 52% (33 of 64). The probability of 5-year failure-free survival and overall survival were 56% (95% confidence interval (CI): 41–69%) and 97% (95%CI: 87–99%), respectively. Median telomere length in responders was −0.4 standard deviation (SD) (−2.7 to +3.0 SD) and −1.5 SD (−4.0 to +1.6 (SD)) in non-responders (P