Papers - TAKAHASHI Masako
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Hirono K, Hata Y, Ichimata S, Nishida N, Imamura T, Asano Y, Kuramoto Y, Tsuboi K, Takarada S, Okabe M, Nakaoka H, Ibuki K, Ozawa S, Muneuchi J, Yasuda K, Urayama K, Oka H, Miyamoto T, Baba K, Kato A, Saiki H, Kuwabara N, Harada M, Baba S, Morikawa M, Iwasaki H, Hirata Y, Ito Y, Sakaguchi H, Urata S, Toda K, Kittaka E, Okada S, Hasebe Y, Hoshino S, Fujii T, Mitsushita N, Nii M, Ogino K, Fujino M, Yoshida Y, Fukuda Y, Iwashima S, Takigiku K, Sakata Y, Inuzuka R, Maeda J, Hayabuchi Y, Fujioka T, Namiki H, Fujita S, Nishida K, Kuraoka A, Kan N, Kido S, Watanabe K, Ichida F
Scientific reports 14 ( 1 ) 30469 2024.12
Language：English Publishing type：Research paper (scientific journal) Publisher：Scientific Reports
Dilated cardiomyopathy (DCM) is a progressive myocardial disorder characterized by impaired cardiac contraction and ventricular dilation. However, some patients with DCM improve when experiencing left ventricular reverse remodeling (LVRR). Currently, the detailed association between genotypes and clinical outcomes, including LVRR, particularly among children, remains uncertain. Pediatric patients with DCM from multiple Japanese institutions recorded between 2014 and 2023 were enrolled. We identified their DCM-related genes and explored the association between gene variants and clinical outcomes, including LVRR. We included 123 pediatric patients (62 males; median age: 8 [1–51] months) and found 50 pathogenic variants in 45 (35.0%) of them. The most identified gene was MYH7 (14.0%), followed by RYR2 (12.0%) and TPM1 (8.0%). LVRR was achieved in 47.5% of these patients. The left ventricular ejection fraction remained unchanged (31.4% to 39.8%, P = 0.1913) in patients with sarcomere gene variants and in those with non-sarcomere gene variants (33.4% to 47.8%, P = 0.0522) but significantly increased in those without gene variants (33.6% to 54.1%, P < 0.0001). LVRR was not uniform across functional gene groups. Hence, an individualized gene-guided prediction approach may be adopted for children with DCM.
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Isolated Blind-Ended Major Aortic Pulmonary Collateral Artery With an Aneurysm in an Infant With Trisomy 21. Reviewed
Yonaga R, Kodama Y, Takamura K, Harada M, Moritake H
Cureus 16 ( 10 ) e72078 2024.10
Language：English Publishing type：Research paper (scientific journal)
DOI： 10.7759/cureus.72078
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Determination of Genotype and Phenotypes in Pediatric Patients With Biventricular Noncompaction Reviewed
Hirono K., Hata Y., Imamura T., Tsuboi K., Takarada S., Okabe M., Nakaoka H., Ibuki K., Ozawa S., Ichimata S., Nishida N., Iwasaki H., Urata S., Okada S., Hiratsuji T., Sakaguchi H., Takigiku K., Nakazawa M., Nishihara E., Harada M., Matsuo O., Yasuda K., Yoshida Y., Namiki H., Yasuda K., Ifuku T., Urayama K., Oka H., Ogino K., Kato A., Kan N., Seki S., Seki M., Odanaka Y., Iwashima S., Yoshida S., Miyata T., Miyamoto T., Watanabe K., Kuwabara N., Inuzuka R., Takahashi Y., Sakazaki H., Muneuchi J., Kogaki S., Numano F., Kido S., Nii M., Hoshino S., Ishida H., Maeda J., Hayabuchi Y., Otsubo Y., Ikeda K., Tsukano S., Watanabe M., Momoi N., Fujii T., Fujioka T., Fujino M., Uchiyama H., Baba S., Horigome H., Honda T., Suzuki K., Ichida F.
Journal of the American Heart Association 13 ( 21 ) 2024
Language：English Publishing type：Research paper (scientific journal) Publisher：Journal of the American Heart Association
BACKGROUND: Left ventricular noncompaction (LVNC) is a hereditary type of cardiomyopathy characterized by prominent tra-beculations. Detailed characteristics of biventricular noncompaction (BiVNC) remain unknown. This study aimed to elucidate the clinical characteristics and genetic landscape of BiVNC.METHODS AND RESULTS: We recruited children with left ventricular noncompaction from Japanese multi-institutional centers from 2013 to 2021. Left ventricular noncompaction was classified as BiVNC, congenital heart disease, arrhythmia, dilated car-diomyopathy, or normal function. In these patients, cardiomyopathy-associated genes were screened. A total of 234 patients (127 male; mean age, 4 months [range, 0–6.6 years]) were enrolled in this study, of whom 25 had BiVNC; 55, normal function; 84, dilated cardiomyopathy; 38, congenital heart disease; and 32, arrhythmia. BiVNC was diagnosed during the perinatal period in 10 patients, in whom the prevalence was higher than that in other patients. A total of 14 patients in the group with BiVNC had congenital heart disease, but not necessarily right heart lesions. Left ventricular dyskinesis was frequently observed in the lateral wall (24%) and apex (28%). Eleven pathogenic variants were found in 11 patients with BiVNC (44.0%). The group with BiVNC had a higher ratio of mitochondrial and developmental gene variants than the other groups. Among all groups, the group with BiVNC had the worst survival rate (P=0.0009). CONCLUSIONS: Pediatric patients with BiVNC had a high rate of ventricular dyskinesis and poor outcome. A comprehensive and careful screening for disease-causing genes and phenotype may help identify specific patients with left ventricular noncom-paction and mortality-related cardiac phenotypes.
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Ebihara Shusei, Kodama Yoshihiko, Takamura Kazunari, Harada Masako, Moritake Hiroshi
Journal of Pediatric Cardiology and Cardiac Surgery advpub ( 0 ) 82 - 86 2024
Language：English Publishing type：Research paper (scientific journal) Publisher：特定非営利活動法人 日本小児循環器学会
Symptoms of coronavirus infectious disease 2019 (COVID-19) are usually mild in young patients. Some children, however, present with a significant degree of complications which may be associated with an excessive reaction by the immune system. Herein, we report an analysis of cytokine and chemokine in an 11-year-old girl diagnosed with left ventricular dysfunction and cardiac tamponade complicated with COVID-19. She recovered without complications after intravenous immunoglobulin, dexamethasone, remdesivir, and surgical pericardial drainage. Cytokine concentrations had markedly increased in the pericardial fluid specimen; especially for interleukin-6 being disproportionate to its serum concentration. Cytokine storm originating from the pericardial cavity was considered an underlying mechanism of her condition.
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宮崎県における免疫グロブリン療法不応川崎病に対する 血漿交換療法の現状 Reviewed
山本 絵里子
宮崎医会誌 47 ( 1 ) 36 - 40 2023.3
Language：Japanese Publishing type：Research paper (scientific journal)
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BMPR2 variant may be related to pulmonary hypertension after lung irradiation Reviewed
Harada M., Yamada A., Nagasawa S., Yamashita N., Kinoshita M., Yoshiura K.i., Moritake H.
Pediatrics International 65 ( 1 ) e15652 2023.1
Authorship：Lead author Language：English Publishing type：Research paper (scientific journal) Publisher：Pediatrics International
DOI： 10.1111/ped.15652
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Chida-Nagai A., Masaki N., Maeda K., Sasaki K., Sato H., Muneuchi J., Ochiai Y., Murayama H., Tahara M., Shiono A., Shinozuka A., Kono F., Machida D., Toyooka S., Sugimoto S., Nakamura K., Akagi S., Kondo M., Kasahara S., Kotani Y., Koizumi J., Oda K., Harada M., Nakajima D., Murata A., Nagata H., Yatsunami K., Kobayashi T., Matsunaga Y., Inoue T., Yamagishi H., Nakagawa N., Ohtani K., Yamamoto M., Ito Y., Hokosaki T., Kuwahara Y., Masutani S., Nomura K., Wada T., Sawada H., Abiko M., Takahashi T., Ishikawa Y., Okada S., Naitoh A., Toda T., Ando T., Masuzawa A., Hoshino S., Kawada M., Nomura Y., Ueno K., Ohashi N., Tachibana T., Cao Y., Ueda H., Yanagi S., Koide M., Mitsushita N., Higashi K., Minosaki Y., Hayashi T., Okamoto T., Kuraishi K., Ehara E., Ishida H., Horigome H., Murakami T., Takei K., Ishii T., Harada G., Hirata Y., Maeda J., Tatebe S., Ota C., Hayabuchi Y., Sakazaki H., Sasaki T., Hirono K., Suzuki S., Yasuda M., Takeda A., Sawai M., Miyaji K., Kitagawa A., Nakai Y., Kakimoto N., Agematsu K., Manabe A., Saiki Y.
Frontiers in Cardiovascular Medicine 10 1212882 2023
Language：English Publishing type：Research paper (scientific journal) Publisher：Frontiers in Cardiovascular Medicine
Aims: Limited data exist on risk factors for the long-term outcome of pulmonary arterial hypertension (PAH) associated with congenital heart disease (CHD-PAH). We focused on the index of pulmonary vascular disease (IPVD), an assessment system for pulmonary artery pathology specimens. The IPVD classifies pulmonary vascular lesions into four categories based on severity: (1) no intimal thickening, (2) cellular thickening of the intima, (3) fibrous thickening of the intima, and (4) destruction of the tunica media, with the overall grade expressed as an additive mean of these scores. This study aimed to investigate the relationship between IPVD and the long-term outcome of CHD-PAH. Methods: This retrospective study examined lung pathology images of 764 patients with CHD-PAH aged <20 years whose lung specimens were submitted to the Japanese Research Institute of Pulmonary Vasculature for pulmonary pathological review between 2001 and 2020. Clinical information was collected retrospectively by each attending physician. The primary endpoint was cardiovascular death. Results: The 5-year, 10-year, 15-year, and 20-year cardiovascular death-free survival rates for all patients were 92.0%, 90.4%, 87.3%, and 86.1%, respectively. The group with an IPVD of ≥2.0 had significantly poorer survival than the group with an IPVD <2.0 (P =.037). The Cox proportional hazards model adjusted for the presence of congenital anomaly syndromes associated with pulmonary hypertension, and age at lung biopsy showed similar results (hazard ratio 4.46; 95% confidence interval: 1.45–13.73; P =.009). Conclusions: The IPVD scoring system is useful for predicting the long-term outcome of CHD-PAH. For patients with an IPVD of ≥2.0, treatment strategies, including choosing palliative procedures such as pulmonary artery banding to restrict pulmonary blood flow and postponement of intracardiac repair, should be more carefully considered.
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Left atrial appendage aneurysm enlarged in the neonatal period. Reviewed
Yamashita N, Harada M, Moritake H
Cardiology in the young 33 ( 8 ) 1 - 3 2022.12
Language：English Publishing type：Research paper (scientific journal) Publisher：Cardiology in the Young
We describe a newborn with a congenital left atrial appendage aneurysm. The aneurysm size did not change prenatally. However, it rapidly enlarged after birth. MRI was useful for assessing the aneurysm extent and exact size, and for diagnosis. Respiratory distress and feeding difficulties appeared, and surgery was performed. These symptoms disappeared post-operatively. The patient is alive without complications or recurrence.
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市中感染型メチシリン耐性黄色ブドウ球菌菌血症を併発した細菌性気管炎. Reviewed
入佐浩史
日本小児救急医学会雑誌. 22 ( 1 ) 20 - 24 2022.5
Language：Japanese Publishing type：Research paper (scientific journal)
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妊娠中のポリフェノール含有飲食物の常習的接種が原因と考えられた胎児動脈管早期収縮 Reviewed
楯 真由美
宮崎県医師会雑誌 46 ( 1 ) 38 - 41 2021.3
Authorship：Corresponding author Publishing type：Research paper (scientific journal)