基本情報
- 所属
- 自治医科大学 医学部小児科学講座 /附属病院とちぎ子ども医療センター小児科 教授
- 学位
- 博士(医学)(2001年12月 岩手医科大学)
- J-GLOBAL ID
- 200901088615999604
- researchmap会員ID
- 5000060482
研究分野
1経歴
13-
2021年11月 - 現在
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2019年10月 - 現在
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2014年4月 - 2019年9月
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2012年4月 - 2014年3月
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2006年10月 - 2012年3月
学歴
2-
2001年12月
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1985年4月 - 1991年3月
委員歴
7-
2021年8月 - 現在
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2020年8月 - 現在
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2018年5月 - 現在
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2016年10月 - 現在
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2018年7月 - 2024年4月
受賞
3論文
97-
Journal of gastroenterology and hepatology 39(2) 312-318 2023年12月6日BACKGROUND AND AIM: Even with increasing numbers of biologic agents available for management of ulcerative colitis (UC), infliximab (IFX) retains an important place in treatment of pediatric patients with this disease. As few reports have addressed outcomes in pediatric UC patients who had to discontinue IFX, we examined clinical course and prognosis after IFX failure in pediatric UC. METHODS: A prospective cohort study of pertinent cases enrolled in the Japanese Pediatric Inflammatory Bowel Disease Registry between 2012 and 2020 was conducted to determine outcomes for pediatric UC patients who received IFX but required its discontinuation during follow-up (IFX failure). RESULTS: Of the 301 pediatric UC patients in the registry, 75 were treated with IFX; in 36 of these, IFX was discontinued during follow-up. Severity of UC at onset and absence of concomitant immunomodulator therapy were significant risk factors for IFX failure (P = 0.005 and P = 0.02, respectively). The cumulative colectomy rate after IFX failure was 41.3% at 1 year and 47.5% at 2 years. Colectomy was significantly more frequent when IFX was discontinued before June 1, 2018, than when IFX was discontinued later (P = 0.013). This difference likely involves availability of additional biologic agents for treatment of UC beginning in mid-2018 (P = 0.005). CONCLUSION: In pediatric UC patients, approximately 50% underwent colectomy during a 2-year interval following IFX failure. Prognosis after IFX failure appeared to improve with availability of new biologic agents and small-molecule drugs in mid-2018.
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J Anus Rectum Colon 27(7) 284-300 2023年10月 査読有り
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Brain & development 45(9) 517-522 2023年7月8日BACKGROUND: Niemann-Pick disease type C (NPC) is an autosomal recessive inherited and neurodegenerative disorder. Approximately 10% of NPC patients have acute liver failure and sometimes need liver transplantation (LT), and 7% reportedly develop inflammatory bowel disease (IBD). We report the case of a girl with NPC who had a re- accumulation of cholesterol in the transplanted liver and NPC-related IBD. CASE REPORT: The patient underwent living donor liver transplantation (LDLT) due to severe acute liver failure caused by an unknown etiology inherited from her father. At 1 year and 6 months (1Y6M), she developed neurological delay, catalepsy, and vertical supranuclear gaze palsy. The foam cells were found in her skin, and fibroblast Filipin staining was positive; hence, she was diagnosed with NPC. It was identified that her father had NPC heterozygous pathogenic variant. At 2 years, she had anal fissure, skin tag and diarrhea. She was diagnosed with NPC-related IBD, using a gastrointestinal endoscopy. Three years after LT, liver biopsy revealed foam cells and numerous fatty droplets. At 8 years, broken hepatocytes and substantial fibrosis were observed. She died from circulation failure due to hypoalbuminemia at 8Y2M. CONCLUSIONS: In NPC, load of cholesterol metabolism is suggested to persist even after LT. LDLT from NPC heterozygous variant donor was insufficient to metabolize cholesterol overload. In NPC patients, the possibility of cholesterol re-accumulation should be considered when LT is performed. NPC-related IBD should be considered when NPC patients have anorectal lesions or diarrhea.
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Journal of gastroenterology and hepatology 38(7) 1107-1115 2023年7月BACKGROUND: Vedolizumab (VDZ) is a humanized monoclonal antibody that binds to α4β7 integrin expressed in T-lymphocytes and is gut selective. Few studies have evaluated the safety and efficacy of VDZ in pediatric ulcerative colitis (UC) patients, especially from Asia. METHODS: A longitudinal multicenter retrospective study was conducted at 10 Japanese tertiary medical institutions. Patients aged ≤18 years old who received VDZ for UC between January 2019 and July 2021 were enrolled. Information on the clinical characteristics, prior/concomitant treatment, and safety during the observation period was collected. RESULTS: The data obtained from 48 patients (males, n = 30; females, n = 18) were analyzed. The median age at VDZ induction was 14 (range 4-18) years old. VDZ was indicated in 73% of patients as switching from previous biologics due to primary failure, loss of response, and adverse events (AEs) and was the first biologic in 27%. Remission was achieved or maintained at weeks 14, 30, and 54 in 79.2%, 75.0%, and 65.8% of patients, respectively. There were no significant differences between the number of previous biologics exposures and VDZ effectiveness. The hematocrit, serum albumin concentrations, and erythrocyte sedimentation rate (ESR) at baseline differed significantly by VDZ effectiveness. Nine AEs, including infusion reaction, were noted in seven (14.3%) patients. There were no severe AEs related to VDZ administration. CONCLUSIONS: VDZ was safe and effective in children with UC. The hematocrit, albumin, and ESR at VDZ initiation might be predictors for VDZ effectiveness. VDZ may be an important option for pediatric patients and can be used as an alternative to immunomodulators.
MISC
191-
小児科 43(13) 2054-2060 2002年12月 招待有り血便をきたす疾患は多岐にわたるが,乳児期に好発する疾患がある.病歴,便の性状,身体所見を詳細に検討のうえ,必要な検査を行い,緊急性の有無を迅速に判断して鑑別診断を進めることが重要である.頻度的には,感染性腸炎を除けば裂肛やアレルギー性腸炎が多いとされる.著者等が経験したsigmoidoscopyの対象となった症例では,乳児良性直腸出血,アレルギー性直腸炎,乳児遷延性下痢症が多かった.乳児良性直腸出血は,その臨床像,組織学的評価から,アレルギー性直腸炎と連続性の病態と考えられる.又,乳児遷延性下痢症も消化管免疫の破綻を反映している.これらの病態はいずれも乳児期の消化管免疫の特殊性が基礎にあると考えられ,さらなる研究が期待される
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小児科診療 65(7) 1055-1062 2002年7月 招待有り小児潰瘍性大腸炎の診断について,自験例20例の経験と最近の知見から述べた.診断には厚生省研究班の診断基準が小児でも適応される.大腸内視鏡検査は,最も診断的価値があり小児例でも積極的に施行されるべきである.自験例の検討では,血便・粘血便が最も頻度が高く95%にみられた.病型分類では全大腸炎型が65%と成人より多く,重症度では重症15%で成人とほぼ同程度であった.鑑別診断としてはCrohn病が重要である
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Journal of Pediatric Gastroenterology and Nutrition 31(Supple. 2) S61 2000年
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リハビリテーション医学 : 日本リハビリテーション医学会誌 30(11) 843-843 1993年11月18日
書籍等出版物
11所属学協会
14共同研究・競争的資金等の研究課題
24-
厚生労働省 厚生労働科学研究費補助金 難治性疾患政策研究事業 2023年4月 - 2026年3月
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厚生労働省 厚生労働科学研究費補助金 難治性疾患政策研究事業 2023年4月 - 2025年3月
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日本学術振興会 科学研究費助成事業 2022年4月 - 2025年3月
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Pfizer Inc. Pfizer Global Medical Grants 2021年1月 - 2024年12月
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厚生労働省 厚生労働科学研究費補助金 難治性疾患政策研究事業 2020年4月 - 2023年3月