論文 - 盛武 浩
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盛武 浩
Haematologica 111 ( 4 ) 1235 - 1245 2026年4月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Ferrata Storti Foundation (Haematologica)
Driver mutations in KMT2A-rearranged (KMT2A-r) have been identified in acute myeloid leukemia (AML); however, age-related differences in their frequency and prognostic factors remain unclear. In this study, we report age-specific mutation profiles and outcomes in pediatric patients with KMT2A-r AML. In 239 cases of KMT2A-r AML, infants (<1 year, n = 59) showed a significantly higher event-free survival (EFS) and overall survival (OS) compared with children (≥1 year, n = 180). Conversely, in 538 cases of non-KMT2A-r AML, infants exhibited a significantly lower EFS and OS than children. KMT2A::MLLT4 was only detected in children with KMT2A-r AML and was associated with a poor prognosis. In KMT2A-r AML, mutations in signaling pathway genes, such as KRAS, were frequently detected in infants and children. However, the frequency of non-signaling pathway mutations was significantly higher in children. Moreover, non-signaling pathway mutations had no significant effect on the prognosis in infants and children, whereas KRAS mutations were associated with poor prognosis in both groups. Multivariate analysis identified older age, a high white blood cell count, KMT2A::MLLT4, and KRAS mutations as independent adverse prognostic factors for both EFS and OS. These age-specific mutation profiles suggest distinct disease mechanisms across age groups and may help refine risk stratification and treatment strategies for pediatric KMT2A-r AML.
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Clinical characteristics and anti-ZSCAN1 antibody titer analysis in a nationwide survey of ROHHAD (-NET) syndrome. 査読あり
Nakamura-Utsunomiya A, Hasegawa K, Ono T, Kanno J, Shima H, Suzuki Y, Tanaka M, Ikegawa K, Amano N, Mogami Y, Yamada Y, Futagawa N, Higuchi Y, Sasaoka D, Nagamatsu F, Mori J, Kawai M, Moritake H, Nishi M, Matsuo M
The Journal of clinical endocrinology and metabolism 2026年3月
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T1 mapping on cardiac magnetic resonance of myocardial calcification after septic shock 査読あり
高橋 雅子, 兒玉 祥彦, 圓﨑 将大, 永澤 俊, 山田 愛, 盛武 浩
Pediatrics international : official journal of the Japan Pediatric Society 68 ( 1 ) e70337 2026年2月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Wiley
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T1 mapping on cardiac magnetic resonance of myocardial calcification after septic shock 査読あり
高橋 雅子, 兒玉 祥彦, 圓﨑 将大, 永澤 俊, 山田 愛, 盛武 浩
Pediatrics international : official journal of the Japan Pediatric Society 68 ( 1 ) e70337 2026年2月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Wiley
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黒木 梨加, 前田 謙一, 木許 恭宏, 盛武 浩
BMJ case reports 19 e270861 2026年1月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：BMJ
We aimed to report a case presenting with abrupt onset of acute right hemiplegia and severe headache, which was diagnosed as primary angiitis of the central nervous system in childhood (cPACNS). MRI revealed an acute cerebral infarction and long-segment concentric vessel wall enhancement in the left middle cerebral artery, with the enhancement resolving within 26 days following prompt steroid therapy, earlier than in previous reports. Laboratory and cerebrospinal fluid findings were unremarkable, except for transient elevations in lactate and pyruvate levels. Other potential causes were excluded. Early immunosuppressive treatment with intravenous methylprednisolone, followed by oral steroids, caused gradual clinical improvement without cognitive decline. This case highlights the significance of contrast-enhanced MRI during the acute phase in patients presenting with cerebral infarction accompanied by headaches. In patients with cPACNS, early therapeutic intervention may cause the prompt resolution of contrast enhancement, reduce long-term sequelae and improve clinical outcomes.
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Kuroki R., Maeda K., Kimoto Y., Moritake H.
BMJ Case Reports 19 ( 1 ) 2026年1月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：BMJ Case Reports
We aimed to report a case presenting with abrupt onset of acute right hemiplegia and severe headache, which was diagnosed as primary angiitis of the central nervous system in childhood (cPACNS). MRI revealed an acute cerebral infarction and long-segment concentric vessel wall enhancement in the left middle cerebral artery, with the enhancement resolving within 26 days following prompt steroid therapy, earlier than in previous reports. Laboratory and cerebrospinal fluid findings were unremarkable, except for transient elevations in lactate and pyruvate levels. Other potential causes were excluded. Early immunosuppressive treatment with intravenous methylprednisolone, followed by oral steroids, caused gradual clinical improvement without cognitive decline. This case highlights the significance of contrast-enhanced MRI during the acute phase in patients presenting with cerebral infarction accompanied by headaches. In patients with cPACNS, early therapeutic intervention may cause the prompt resolution of contrast enhancement, reduce long-term sequelae and improve clinical outcomes.
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青木 良則, 楯 真由美, 土持 皓平, 盛武 浩
Frontiers in microbiology 16 1742512 2026年1月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Frontiers Media SA
Early empirical antibiotic therapy is common in preterm and very low birth weight (VLBW) infants but may disrupt the developing gut microbiome. However, the effects of brief antibiotic courses remain unclear, particularly in the most immature infants. In this prospective multicenter cohort study, we examined gut microbiome trajectories in VLBW infants (many of whom were extremely preterm) receiving no antibiotics, a short course (≤2 days), or prolonged exposure (≥3 days). Serial stool samples were analyzed using 16S rRNA gene sequencing. Microbiome composition and diversity in the short-course group were similar to those in unexposed infants at all timepoints, indicating that brief antibiotic exposure did not disrupt microbial development. In contrast, prolonged exposure was associated with transient dysbiosis characterized by reduced Bifidobacterium abundance and lower alpha diversity, with partial recovery by discharge. These findings suggest that limiting empirical antibiotic therapy to ≤2 days (48 h) when infection is unconfirmed may not disrupt microbiome development even in highly immature preterm VLBW infants, supporting evidence-based antibiotic stewardship in neonatal intensive care.
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Yamashita N., Kodama Y., Irisa H., Ifuku T., Nakatani K., Uchiyama Y., Moritake H., Watanabe N.
Journal of the American Society of Echocardiography 2026年
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Journal of the American Society of Echocardiography
Background: Periarterial echogenicity in the proximal coronary arteries (CAs) increases in the acute phase of Kawasaki disease (KD). However, some studies have questioned the diagnostic value of periarterial echogenicity in differentiating KD from other febrile diseases (non-KD) because of its relatively low specificity. In this study, the authors quantitatively assessed the degree of echogenicity in the proximal and mid segments of both CAs to determine its additional diagnostic value in patients with clinically suspected KD. Methods: A total of 109 consecutive children (median age, 21 months; interquartile range, 11.0-47.8 months) who underwent transthoracic echocardiography for suspected KD (April 2021 to March 2023) were retrospectively examined. Two-dimensional echocardiographic images in the proximal and mid segments of both CAs were digitally stored and transferred to an offline image analysis system. The mean pixel value of the arterial wall was calculated in grayscale ranging from 0 to 255 (corrected for the intracardiac blood pool adjacent to the target site). Results: A total of 109 patients were included, 87 (80%) ultimately diagnosed with KD (including 18 with incomplete KD) and 22 (20%) ultimately diagnosed with non-Kawasaki febrile diseases. Although the KD group generally showed higher CA wall echogenicity than the non-KD febrile group, there was no significant difference in the mean pixel values at the proximal segment (P = .34 for each). The KD group showed significantly higher echogenicity in the mid segments of both CAs than the non-KD febrile group (mid right coronary artery, P = .0049; mid left anterior descending coronary artery, P = .011). Similar results were observed in a small prospective cohort of 31 children examined under rigorously standardized ultrasound settings. Conclusions: CA echogenicity in the mid segments may have potential diagnostic value in the early evaluation of suspected KD, possibly reflecting the characteristic diffuse involvement of the CAs in the acute phase.
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Aoki Y., Tate M., Ochiai K., Tsuchimochi K., Mizuguchi U., Okazaki K., Moritake H.
Frontiers in Microbiology 16 1742512 2026年
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Frontiers in Microbiology
Early empirical antibiotic therapy is common in preterm and very low birth weight (VLBW) infants but may disrupt the developing gut microbiome. However, the effects of brief antibiotic courses remain unclear, particularly in the most immature infants. In this prospective multicenter cohort study, we examined gut microbiome trajectories in VLBW infants (many of whom were extremely preterm) receiving no antibiotics, a short course (≤2 days), or prolonged exposure (≥3 days). Serial stool samples were analyzed using 16S rRNA gene sequencing. Microbiome composition and diversity in the short-course group were similar to those in unexposed infants at all timepoints, indicating that brief antibiotic exposure did not disrupt microbial development. In contrast, prolonged exposure was associated with transient dysbiosis characterized by reduced Bifidobacterium abundance and lower alpha diversity, with partial recovery by discharge. These findings suggest that limiting empirical antibiotic therapy to ≤2 days (48 h) when infection is unconfirmed may not disrupt microbiome development even in highly immature preterm VLBW infants, supporting evidence-based antibiotic stewardship in neonatal intensive care.
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Successful Treatment of Mediastinal Anthracycline Extravasation by Administration of Dexrazoxane 査読あり
永澤 俊, 山田 愛, 盛武 浩
Pediatric blood & cancer e70026 2025年12月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Wiley
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Novel SKIC3 variants in tricho-hepato-enteric syndrome with hemochromatosis 査読あり
Ochiai K., Aoki Y., Yamada N., Aman M., Yamashita A., Yamaguchi M., Nakato D., Takenouchi T., Kosaki K., Kodama Y., Moritake H.
Human Genome Variation 12 ( 1 ) 14 2025年12月
担当区分：最終著者 記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Human Genome Variation
Tricho-hepato-enteric syndrome (THES), a rare autosomal recessive disorder caused by variants in the SKIC3 or SKIC2 gene, is characterized by intractable diarrhea, woolly hair, growth restriction and liver disease. Here we report a neonatal case of THES with neonatal hemochromatosis, in which the novel compound heterozygous SKIC3 variants NM_014639.4:c.815_816del p.(Gly272AlafsTer9) and NM_014639.4:c.2284G>A p.(Gly762Arg) were identified. Further research is needed to elucidate the mechanisms underlying iron metabolism dysregulation in THES.
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Re-evaluating the MYH9 p.I1816V variant in a patient with atypical clinical presentation 査読あり
此元 隆雄, 若松 美仁, 阪口 嘉美, 黒木 純, 田中 悦子, 盛武 浩
Pediatric nephrology 2025年11月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Springer Science and Business Media LLC
MYH9-related disease (MYH9-RD) is an autosomal dominant disorder typically characterized by macrothrombocytopenia, leukocyte inclusion bodies, and variable non-hematologic manifestations such as hearing loss and nephropathy. We herein describe a 16-year-old boy presenting with persistent proteinuria and biopsy-proven membranous nephropathy with focal segmental sclerosis. Genetic testing identified a rare MYH9 variant (p.I1816V), previously reported in association with Epstein syndrome. However, the patient had normal platelet counts, no leukocyte inclusions, and no abnormalities in non-muscle myosin heavy chain IIA (NMMHC-IIA) expression in neutrophils or podocytes. Although globally rare, the p.I1816V variant is more frequent in East Asian populations and is predicted to be benign by multiple in silico tools. This case illustrates the challenges of interpreting rare variants in the absence of supportive clinical findings and highlights the need for cautious evaluation in the era of next-generation sequencing.
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Lymphatic dysplasia evaluated by indocyanine green lymphography in congenital myotonic dystrophy 査読あり
楯 真由美, 青木 良則, 兒玉 祥彦, 中目 和彦, 児玉 由紀, 盛武 浩
Pediatrics International 67 ( 1 ) e70170 2025年11月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：John Wiley and Sons Inc
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Age-specific mutation profiles and their prognostic implications in pediatric KMT2A-rearranged acute myeloid leukemia. 査読あり
Shoji K, Yoshida K, Iyoda S, Ishikawa M, Tanaka M, Nobe M, Saito N, Shino Y, Nannya Y, Yamato G, Tsujimoto S, Shiba N, Hayashi Y, Shiozawa Y, Shiraishi Y, Chiba K, Okada A, Tanaka H, Miyano S, Koga Y, Goto H, Terui K, Ito E, Kiyokawa N, Tomizawa D, Taga T, Moritake H, Tawa A, Takita J, Nishikori M, Adachi S, Ogawa S, Matsuo H
Haematologica 111 ( 4 ) 1235 - 1245 2025年10月
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Interference of Intravenous Acetaminophen with Continuous Glucose Monitoring System. 査読あり
Matsuyama M, Meiri S, Sawada H, Masuya R, Nakame K, Moritake H
JMA journal 8 ( 4 ) 1463 - 1467 2025年10月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：公益社団法人 日本医師会 / 日本医学会
Sensor-augmented pumps (SAPs) and automated insulin delivery (AID) systems are innovative technologies for diabetes management. Accurate continuous glucose monitoring (CGM) is crucial for their safe and effective use; however, certain commonly used drugs can interfere with CGM accuracy. Although acetaminophen is known to cause falsely elevated CGM glucose values, previous CGM studies have primarily focused on its oral administration, with limited data on intravenous use. We report a case of a CGM reaction after the intravenous administration of acetaminophen in a boy with type 1 diabetes using SAP. The patient received repeated doses of intravenous acetaminophen (15 mg/kg for 15 min) for pain relief. After administration, we recorded a rapid increase in his CGM readings without a corresponding increase in blood glucose levels. The CGM glucose peaked at 29.2 ± 1.9 min (mean ± standard deviation) after administration and an estimated discrepancy of 55 to 114 mg/dL compared with capillary blood glucose measurements. Discrepancies between measured blood glucose and CGM readings were significantly greater at lower glucose levels. These falsely elevated CGM readings could potentially trigger an inappropriate autocorrection bolus in AID systems and increase the risk of hypoglycemia. Medical professionals should be fully aware of acetaminophen-induced CGM interference, particularly the potential risks in patients using AID systems.
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盛武 浩
Frontiers in Hematology 4 1668408 2025年9月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Frontiers Media SA
Background: Epigenetic dysregulation plays a central role in pediatric acute myeloid leukemia (AML), yet its clinical relevance remains underexplored. This study primarily aimed to elucidate the clinical effect of H3K27me3 and H3K4me3 status on pediatric acute myeloid leukemia. We evaluated the prognostic impact of H3K27me3 and H3K4me3 histone trimethylation, along with associated gene expression profiles, in pediatric AML. Methods: We retrospectively analyzed 74 children with newly diagnosed non-FAB M3 and non-Down syndrome AML in a prolonged cohort in Japan. Bone marrow immunohistochemistry assessed H3K27me3 and H3K4me3 expression levels. RNA sequencing was successfully performed on sorted leukemic blasts in six representative cases, owing to limited sample availability. Chemoresistance and epigenetic modulation were evaluated in AML cell lines treated with GSK-J4, a histone demethylase inhibitor. Results: High H3K27me3 expression at diagnosis was significantly associated with superior overall and event-free survival over three years (OS HR 8.0; EFS HR 5.0; both p < 0.01). H3K4me3 levels at diagnosis showed no prognostic impact. Among 14 KMT2A-rearranged cases, all six patients with high H3K27me3 achieved a long-term first remission (median follow-up: 10 years), whereas those with low expression had higher relapse rates. Transcriptomic analysis revealed upregulation of HOXA9, and HOXA-cluster genes and downregulation of ABCB1, in low H3K27me3 samples. In vitro, GSK-J4 increased H3K27me3 and suppressed HOXA9 expression in KG-1 cells, enhancing sensitivity to cytarabine. Conclusion: Low H3K27me3 expression defines a poor-risk group in pediatric AML, potentially via HOXA9-driven dysregulation. H3K27me3 may serve as a prognostic biomarker and potential therapeutic target.
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Yamamoto N, Maeda K, Kimoto Y, Moritake H
BMJ case reports 18 ( 8 ) 2025年8月
担当区分：最終著者 記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：BMJ Case Reports
Rasmussen encephalitis (RE) is a progressive disease characterised by unilateral brain atrophy, drug-resistant epilepsy, epilepsia partialis continua, hemiparesis and cognitive decline. Early initiation of immunomodulatory therapy is crucial to slow disease progression. However, early formal diagnosis is challenging as it typically requires hemispheric atrophy or brain biopsy. This case reports on a preschool-aged boy with RE who began immunotherapy before demonstrating clear hemispheric atrophy. Follow-up MRI did not indicate global hemispheric atrophy; however, FreeSurfer-based volumetric analysis revealed a decreased left:right hemispheric volume ratio, suggesting early left hemispheric atrophy. Subsequently, intensive immunotherapy was administered. Over 3 years of treatment, the patient exhibited gradual hemispheric atrophy on MRI, along with mild motor and cognitive impairments. Serial FreeSurfer-based volumetric analysis may contribute to detecting subtle hemispheric volume changes in RE, facilitating prompt diagnosis and early initiation of immunotherapy - potentially limiting disease progression.
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Novel SKIC3 variants in tricho-hepato-enteric syndrome with hemochromatosis 査読あり
青木 良則, 阿萬 紫, 山下 篤, 山口 昌俊, 児玉 由紀, 盛武 浩
Human Genome Variation 12 14 2025年7月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Springer Nature
Tricho-hepato-enteric syndrome (THES), a rare autosomal recessive disorder caused by variants in the SKIC3 or SKIC2 gene, is characterized by intractable diarrhea, woolly hair, growth restriction and liver disease. Here we report a neonatal case of THES with neonatal hemochromatosis, in which the novel compound heterozygous SKIC3 variants NM_014639.4:c.815_816del p.(Gly272AlafsTer9) and NM_014639.4:c.2284G>A p.(Gly762Arg) were identified. Further research is needed to elucidate the mechanisms underlying iron metabolism dysregulation in THES.
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Fauzi YR, Nakahata S, Shimoda K, Matsuura T, Hagiwara S, Inoue K, Moritake H, Morishita K
Biochemical and biophysical research communications 756 151564 2025年2月
記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Biochemical and Biophysical Research Communications
Previously, we developed a complete human IgG TFR1 antibody (JST-TFR09/PPMX-T003) that showed a potentially practical therapeutic effect against adult T-cell leukemia/lymphoma (ATLL) in vitro and in vivo. In the present study, to elucidate the molecular mechanism underlying ATLL cell death induced by anti-TFR1 antibodies, we performed comprehensive gene expression analysis and mass spectrometry on ATLL cells treated with PPMX-T003 antibody. These results suggest that PPMX-T003 antibody treatment of ATLL cell lines induces ferroptosis mediated by ferritin degradation. PPMX-T003 antibody-treated ATLL cell lines showed a decrease in ferritin proteins, an increase in ferrous iron (Fe2+), reactive oxygen species (ROS) generation, and malondialdehyde as induction of lipid peroxidation. Moreover, treatment with a ferroptosis inhibitor (ferroportin-1) inhibited the cell death induced by PPMX-T003 antibody in ATLL cells. Furthermore, NCO4A and LC3-II were induced following antibody treatment, and ferritin degradation was inhibited by lysosomal inhibitors, suggesting that ferritin degradation depends on autolysosomal system activation. Here, we introduce ferroptosis as one of the potential mechanisms of PPMX-T003 antibody, which is promising for future therapeutic antibodies targeting a wide range of leukemia and cancers, including ATLL.
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Aoki Y., Kota Y., Shimada M., Taniguchi T., Yamauchi S., Matsusaka M., Hamasuna K., Watanabe Y., Kodama Y., Moritake H.
Children 12 ( 2 ) 2025年2月
担当区分：責任著者 記述言語：英語 掲載種別：研究論文（学術雑誌） 出版者・発行元：Children
Background: Preterm infants often have unstable vital signs and prolonged hospital stays that can hinder parent–infant bonding, especially under COVID-19 restrictions. This study aimed to evaluate whether listening to songs recorded by parents was effective in stabilizing the condition of premature infants. Methods: This randomized controlled study was conducted at the University of Miyazaki Hospital from October 2022 to March 2024 during the COVID-19 pandemic period. The participants were preterm infants born at less than 33 weeks gestation and their parents, all of whom recorded songs. The recorded songs were played daily to the infants in the intervention group, while the control group received usual care. Primary outcomes included vital signs (respiratory rate, pulse oximetry saturation, heart rate) and activity level. Results: Data for 33 preterm infants (intervention, n = 17 [total 749 sessions]; control, n = 16 [total 721 sessions]) were analyzed for changes in vital signs and activity levels. The intervention reduced infants’ respiratory rates (4.1 [95% CI: 2.5–5.6], p < 0.001) and slightly but statistically significantly increased pulse oximetry saturation (0.6 [95% CI: 0.02–1.2], p < 0.044). Conclusions: Recorded parental songs were found to safely stabilize the respiratory status of preterm infants and may serve as an accessible intervention to support parent–infant attachment, particularly in settings with restricted parental visitation.