Identification of pathogenesis, and development of therapeutic method for ossification of posterior longitudinal ligament recognized among inherited FGF23-related hypophosphatemic rickets/osteomalacia

• Project/Area Number: 21K09293
• Research Category: Grant-in-Aid for Scientific Research (C)
• Allocation Type: Multi-year Fund
• Section: 一般
• Review Section: Basic Section 56020:Orthopedics-related
• Research Institution: The University of Tokyo
• Principal Investigator: ITO NOBUAKI 東京大学, 医学部附属病院, 特任准教授 (10731862)
• Co-Investigator(Kenkyū-buntansha): 矢野 文子 東京大学, 医学部附属病院, 届出研究員 (80529040)
• Project Period (FY): 2021-04-01 – 2024-03-31
• Project Status: Completed (Fiscal Year 2023)
• Budget Amount: ¥4,160,000 (Direct Cost: ¥3,200,000、Indirect Cost: ¥960,000); Fiscal Year 2023: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000); Fiscal Year 2022: ¥1,690,000 (Direct Cost: ¥1,300,000、Indirect Cost: ¥390,000); Fiscal Year 2021: ¥780,000 (Direct Cost: ¥600,000、Indirect Cost: ¥180,000)
• Keywords: FGF23 / くる病 / 骨軟化症 / 後縦靭帯骨化症 / 異所性骨化症 / 腱付着部症 / 神経障害 / 関節可動域制限 / X連鎖性低リン血症性くる病 / 低リン血症 / インピンジメント症候群 / ピロリン酸 / X染色体連鎖性低リン血症性くる病 / 常染色体潜性低リン血症性くる病 / 変形性関節症

Outline of Research at the Start

FGF23は骨細胞が産生する血清リン濃度調節ホルモンでFGF23過剰分泌により低リン血症性くる病・骨軟化症を発症する遺伝性疾患としてXLH、ARHR1,2がある。これらの疾患では後縦靭帯骨化症等の異所性骨化症を合併し、疼痛や可動域制限、神経障害などでQOLを低下させる。リン感知を担うFGFRの感知閾値低下が、FGF23分泌過剰の発症機序だが、我々はこの異常が同時に異所性骨化を惹起している考えた。OPLL発症モデルマウス(ARHR2マウス)を用いて、FGFRシグナル解析、FGFRシグナル阻害の影響を評価することでFGF23関連疾患におけるOPLL発症機序を解明しその治療、予防薬の開発に繋げる。

Outline of Final Research Achievements

X-linked hypophosphatemic rickets (XLH) is the most frequent form of FGF23-related hypophosphatemia due to loss-of-function varinat in PHEX. XLH also present ectopic ossifications including ossification in the posterior longuitudinal ligament (OPLL) which aggravates patients' QOL. We conducted comprehensive imaging tests among 25 adult XLH patients, which revealed extremely high ectopic ossification rates: OPLL 32% (control adults 2-4%) and osteophyte around hip joints 96% (control adult 15.7%). Then we compared the prevalence of these ectopic ossifications between adult XLH patinets who initiated a treatment befor and after 5 years old among 30 patients. There was no significant difference of the prevalence and severity of ectopic ossifications between these two groups which suggested that the etiology for the development of ectopic ossification in adult XLH patients is independent of serum phosphate levels.

Academic Significance and Societal Importance of the Research Achievements

成人XLH患者において、異所性骨化症による神経障害や関節の可動域障害はQOLを著しく低下させる合併症であるが、その原因が依然として明らかとされていない。今回の検討において、成人XLHにおける異所性骨化症の具体的な頻度が明らかとなり、成人XLHの多くの症例が同病態によって社会活動を制限されていることが明らかとなった。また治療介入時期の違いによって異所性骨化症の程度に差がなかったことから、これまでに開発されてきたFGF23などを標的とした低リン血症の治療では、異所性骨化症の改善にはつながらず、まずは異所性骨化症がなぜ生じるのかを明らかとすることが本病態の治療法の端緒となることを示すことができた。

Research Products

• [Journal Article] Cartilage tissues regulate systemic aging via ectonucleotide pyrophosphatase/phosphodiesterase 1 in mice (2024)
  • Author(s): Arima Takahiro、Taniwaki Takuya、et al.
  • Journal Title: Journal of Biological Chemistry Volume: 300 Issue: 1 Pages: 105512-105512
  • DOI: 10.1016/j.jbc.2023.105512
  • Peer Reviewed
• [Journal Article] The pathophysiology of hypophosphatemia (2024)
  • Author(s): Ito Nobuaki、Hidaka Naoko、Kato Hajime
  • Journal Title: Best Practice & Research Clinical Endocrinology & Metabolism Volume: 38 Issue: 2 Pages: 101851-101851
  • DOI: 10.1016/j.beem.2023.101851
• [Journal Article] Extended curettage for tumour-induced osteomalacia in the bone (2024)
  • Author(s): Okajima Koichi、Kobayashi Hiroshi、Ito Nobuaki、Kato Hajime、Ishibashi Yuki、Zhang Liuzhe、Tsuda Yusuke、Tanaka Sakae
  • Journal Title: Japanese Journal of Clinical Oncology Volume: - Issue: 4 Pages: 463-470
  • DOI: 10.1093/jjco/hyad180
• [Journal Article] FGF-23, Left Ventricular Hypertrophy, and Mortality in Patients With CKD: A Revisit With Mediation Analysis (2024)
  • Author(s): Hidaka Naoko、Inoue Kosuke、Kato Hajime、Hoshino Yoshitomo、Koga Minae、Kinoshita Yuka、Takashi Yuichi、Makita Noriko、Fukumoto Seiji、Nangaku Masaomi、Ito Nobuaki
  • Journal Title: JACC: Advances Volume: 3 Issue: 1 Pages: 100747-100747
  • DOI: 10.1016/j.jacadv.2023.100747
• [Journal Article] Acquired Forms of Fibroblast Growth Factor 23-Related Hypophosphatemic Osteomalacia (2024)
  • Author(s): Ito Nobuaki、Hidaka Naoko、Kato Hajime
  • Journal Title: Endocrinology and Metabolism Volume: - Issue: 2 Pages: 255-261
  • DOI: 10.3803/enm.2023.1908
• [Journal Article] The 2023 Guidelines for the management and treatment of glucocorticoid-induced osteoporosis (2024)
  • Author(s): Tanaka Yoshiya、Soen Satoshi、Hirata Shintaro、Okada Yosuke、Fujiwara Saeko、Tanaka Ikuko、Kitajima Yuriko、Kubota Takuo、Ebina Kosuke、Takashi Yuichi、Inoue Reiko、Yamauchi Mika、Okubo Naoaki、Ueno Masanobu、Ohata Yasuhisa、Ito Nobuaki、Ozono Keiichi、Nakayama Hisanori、Terauchi Masakazu、Tanaka Sakae、Fukumoto Seiji
  • Journal Title: Journal of Bone and Mineral Metabolism Volume: 42 Issue: 2 Pages: 143-154
  • DOI: 10.1007/s00774-024-01502-w
  • Peer Reviewed / Open Access
• [Journal Article] Intact Fibroblast Growth Factor 23 Concentrations in Hypophosphatemic Disorders. (2023)
  • Author(s): Ramos P, Larson B, Ashrafzadeh-Kian S, Ito N, Kato H, Bornhorst JA, Algeciras-Schimnich A.
  • Journal Title: Endocr Pract Volume: 29 Issue: 3 Pages: 193-198
  • DOI: 10.1016/j.eprac.2023.01.003
  • Peer Reviewed / Int'l Joint Research
• [Journal Article] Clinical performance of a new intact FGF23 immunoassay in healthy individuals and patients with chronic hypophosphatemia. (2023)
  • Author(s): Kato H, Miyazaki H, Kimura T, Hoshino Y, Hidaka N, Koga M, Nangaku M, Makita N, Ito N.
  • Journal Title: Bone Rep Volume: 18 Pages: 101659-101659
  • DOI: 10.1016/j.bonr.2023.101659
  • Peer Reviewed / Open Access
• [Journal Article] Patterns of symptoms and insufficiency fractures in patients with tumour-induced osteomalacia (2023)
  • Author(s): Kobayashi Hiroshi、Ito Nobuaki、Nakai Yudai、Katoh Hajime、Okajima Koichi、Zhang Liuzhe、Tsuda Yusuke、Tanaka Sakae
  • Journal Title: The Bone & Joint Journal Volume: 105-B Issue: 5 Pages: 568-574
  • DOI: 10.1302/0301-620x.105b5.bjj-2022-1206.r2
• [Journal Article] Recurrent bilateral adrenal infarction with myelodysplastic/myeloproliferative neoplasm-unclassifiable (MDS/MPN-U): a case report (2023)
  • Author(s): Hoshino Yoshitomo、Manaka Katsunori、Sato Junichiro、Asatsuma Yui、Horikoshi Hirofumi、Takeuchi Maki、Ito Nobuaki、Fujita Megumi、Yasunaga Megumi、Matsuda Kensuke、Honda Akira、Maki Hiroaki、Masamoto Yosuke、Kurokawa Mineo、Nangaku Masaomi、Makita Noriko
  • Journal Title: BMC Endocrine Disorders Volume: 23 Issue: 1
  • DOI: 10.1186/s12902-023-01384-5
• [Journal Article] FGF23-related hypophosphatemia in a patient with small cell lung cancer: a case report and literature review (2023)
  • Author(s): Kato Hajime、Kimura Soichiro、Taguchi Maho、Sunouchi Takashi、Hoshino Yoshitomo、Hidaka Naoko、Edvige Foligno Nadia、Koga Minae、Manaka Katsunori、Tamiya Hiroyuki、Kawakami Masanori、Kage Hidenori、Yasunaga Yoichi、Nangaku Masaomi、Makita Noriko、Ito Nobuaki
  • Journal Title: Endocrine Journal Volume: 70 Issue: 10 Pages: 1005-1013
  • DOI: 10.1507/endocrj.EJ23-0086
  • ISSN: 0918-8959, 1348-4540
• [Journal Article] Genetics of Diffuse Idiopathic Skeletal Hyperostosis and Ossification of the Spinal Ligaments (2023)
  • Author(s): Kato Hajime、Braddock Demetrios T.、Ito Nobuaki
  • Journal Title: Current Osteoporosis Reports Volume: 21 Issue: 5 Pages: 552-566
  • DOI: 10.1007/s11914-023-00814-6
• [Journal Article] A Novel Long Noncoding RNA in Osteocytes Regulates Bone Formation through the Wnt/β-Catenin Signaling Pathway (2023)
  • Author(s): Arai Makoto、Ochi Hiroki、Sunamura Satoko、Ito Nobuaki、Nangaku Masaomi、Takeda Shu、Sato Shingo
  • Journal Title: International Journal of Molecular Sciences Volume: 24 Issue: 17 Pages: 13633-13633
  • DOI: 10.3390/ijms241713633
  • Peer Reviewed / Open Access
• [Journal Article] PAI‐1 antagonist ameliorates hypophosphatemia in the Hyp vitamin D‐resistant rickets model mouse (2023)
  • Author(s): Qian Cheng、Ito Nobuaki、Tsuji Kunikazu、Sato Shingo、Kikuchi Katsushi、Yoshii Toshitaka、Miyata Toshio、Asou Yoshinori
  • Journal Title: FEBS Open Bio Volume: 14 Issue: 2 Pages: 290-299
  • DOI: 10.1002/2211-5463.13745
  • Peer Reviewed / Open Access / Int'l Joint Research
• [Journal Article] The Effect of Asfotase Alfa on Plasma and Urine Pyrophosphate Levels and Pseudofractures in a Patient With Adult‐Onset Hypophosphatasia (2023)
  • Author(s): Hidaka Naoko、Murata Hiroaki、Tachikawa Kanako、Osaki Keiichi、Sekiyama Takashi、Kinoshita Yuka、Kato Hajime、Hoshino Yoshitomo、Kimura Soichiro、Sunouchi Takashi、Watanabe So、Nangaku Masaomi、Makita Noriko、Michigami Toshimi、Ito Nobuaki
  • Journal Title: JBMR Plus Volume: 7 Issue: 12
  • DOI: 10.1002/jbm4.10842
• [Journal Article] Surgical and biochemical outcomes of phosphaturic mesenchymal tumors causing tumor-induced osteomalacia in the head and neck region (2023)
  • Author(s): Tsuda Yusuke、Yasunaga Yoichi、Abe Masanobu、Hoshi Kazuto、Ito Nobuaki、Kondo Kenji、Okajima Koichi、Zhang Liuzhe、Kato Hajime、Hidaka Naoko、Tanaka Sakae、Kobayashi Hiroshi
  • Journal Title: Oral and Maxillofacial Surgery Cases Volume: 9 Issue: 4 Pages: 100335-100335
  • DOI: 10.1016/j.omsc.2023.100335
• [Journal Article] Utility of Multimodality Approach Including Systemic FGF23 Venous Sampling in Localizing Phosphaturic Mesenchymal Tumors. (2022)
  • Author(s): Kato H, Koga M, Kinoshita Y, Hidaka N, Hoshino Y, Takashi Y, Arai M, Kobayashi H, Katsura M, Nakamoto Y, Makise N, Ushiku T, Hoshi K, Nangaku M, Makita N, Fukumoto S, Ito N.
  • Journal Title: J Endocr Soc Volume: 24 Issue: 2
  • DOI: 10.1210/jendso/bvac181
  • Peer Reviewed / Open Access
• [Journal Article] Effect of conventional treatment on dental complications and ectopic ossifications among 30 adults with XLH. (2022)
  • Author(s): Kato H, Okawa R, Ogasawara T, Hoshino Y, Hidaka N, Koga M, Kinoshita Y, Kobayashi H, Taniguchi Y, Fukumoto S, Nangaku M, Makita N, Hoshi K, Nakano K, Ito N.
  • Journal Title: J Clin Endocrinol Metab Volume: dgac732. Issue: 6 Pages: 1405-1414
  • DOI: 10.1210/clinem/dgac732
  • Peer Reviewed / Open Access
• [Journal Article] Incidence of ossification of the spinal ligaments in acromegaly patients. (2022)
  • Author(s): Hoshino Y, Hidaka N, Kato H, Koga M, Taniguchi Y, Kobayashi H, Nangaku M, Makita N, Ito N.
  • Journal Title: Bone Rep Volume: 17 Pages: 101628-101628
  • DOI: 10.1016/j.bonr.2022.101628
  • Peer Reviewed / Open Access
• [Journal Article] The Effect of Burosumab on Intact and C-terminal FGF23 Measurements. (2022)
  • Author(s): Ashrafzadeh-Kian SL, Ito N, Srivastava T, Garg U, Kato H, Algeciras-Schimnich A, Bornhorst JA.
  • Journal Title: Clin Endocrinol (Oxf) Volume: Epub ahead of print. Issue: 2 Pages: 152-157
  • DOI: 10.1111/cen.14832
  • Peer Reviewed / Int'l Joint Research
• [Journal Article] Machine Learning-Based Prediction of Elevated PTH Levels Among the US General Population. (2022)
  • Author(s): Kato H, Hoshino Y, Hidaka N, Ito N, Makita N, Nangaku M, Inoue K.
  • Journal Title: J Clin Endocrinol Metab Volume: 107 Issue: 12 Pages: 3222-3230
  • DOI: 10.1210/clinem/dgac544
  • Peer Reviewed
• [Journal Article] Efficacy of Burosumab in Adults with X-linked Hypophosphatemia (XLH): A Post Hoc Subgroup Analysis of a Randomized Double-Blind Placebo-Controlled Phase 3 Study. (2022)
  • Author(s): Brandi ML, Jan de Beur S, Briot K, Carpenter T, Cheong HI, Cohen-Solal M, Crowley RK, Eastell R, Imanishi Y, Imel EA, Ing SW, Insogna K, Ito N, Javaid K, Kamenicky P, Keen R, Kubota T, Lachmann RH, Perwad F, Pitukcheewanont P, Portale A, Ralston SH, Tanaka H, Weber TJ, Yoo HW, Sun W, Williams A, Nixon A, Takeuchi Y.
  • Journal Title: Calcif Tissue Int Volume: 111 Issue: 4 Pages: 409-418
  • DOI: 10.1007/s00223-022-01006-7
  • Peer Reviewed / Int'l Joint Research
• [Journal Article] Clinical Challenges in Diagnosis, Tumor Localization and Treatment of Tumor-Induced Osteomalacia: Outcome of a Retrospective Surveillance. (2022)
  • Author(s): Hidaka N, Koga M, Kimura S, Hoshino Y, Kato H, Kinoshita Y, Makita N, Nangaku M, Horiguchi K, Furukawa Y, Ohnaka K, Inagaki K, Nakagawa A, Suzuki A, Takeuchi Y, Fukumoto S, Nakatani F, Ito N.
  • Journal Title: J Bone Miner Res Volume: 37 Issue: 8 Pages: 1479-1488
  • DOI: 10.1002/jbmr.4620
  • Peer Reviewed / Open Access
• [Journal Article] A Mild Case of Autosomal Recessive Osteopetrosis Masquerading as the Dominant Form Involving Homozygous Deep Intronic Variations in the CLCN7 Gene. (2022)
  • Author(s): Hofstaetter JG, Atkins GJ, Kato H, Kogawa M, Blouin S, Misof BM, Roschger P, Evdokiou A, Yang D, Solomon LB, Findlay DM, Ito N.
  • Journal Title: Calcif Tissue Int Volume: 111 Issue: 4 Pages: 430-444
  • DOI: 10.1007/s00223-022-00988-8
  • Peer Reviewed / Int'l Joint Research
• [Journal Article] Identification of ENPP1 Haploinsufficiency in Patients With Diffuse Idiopathic Skeletal Hyperostosis and Early‐Onset Osteoporosis (2022)
  • Author(s): Kato Hajime、Ansh Anenya J、Lester Ethan R、Kinoshita Yuka、Hidaka Naoko、Hoshino Yoshitomo、Koga Minae、Taniguchi Yuki、Uchida Taisuke、Yamaguchi Hideki、Niida Yo、Nakazato Masamitsu、Nangaku Masaomi、Makita Noriko、Takamura Toshinari、Saito Taku、Braddock Demetrios T、Ito Nobuaki
  • Journal Title: Journal of Bone and Mineral Research Volume: - Issue: 6 Pages: 1125-1135
  • DOI: 10.1002/jbmr.4550
  • Peer Reviewed / Open Access / Int'l Joint Research
• [Journal Article] Burden of disease of X-linked hypophosphatemia in Japanese and Korean patients: a cross-sectional survey (2022)
  • Author(s): Ito N, Kang HG, Nishida Y, Evins A, Skrinar A, Cheong HI.
  • Journal Title: Endocrine Journal Volume: 69 Issue: 4 Pages: 373-383
  • DOI: 10.1507/endocrj.EJ21-0386
  • NAID: 130008112893
  • ISSN: 0918-8959, 1348-4540
  • Peer Reviewed / Int'l Joint Research
• [Journal Article] Association of Serum Parathyroid Hormone Levels With All-Cause and Cause-Specific Mortality Among U.S. Adults (2022)
  • Author(s): Kato Hajime、Ito Nobuaki、Makita Noriko、Nangaku Masaomi、Leung Angela M.、Inoue Kosuke
  • Journal Title: Endocrine Practice Volume: 28 Issue: 1 Pages: 70-76
  • DOI: 10.1016/j.eprac.2021.09.005
  • Peer Reviewed / Open Access / Int'l Joint Research
• [Journal Article] Radiological evaluation of pseudofracture after the administration of asfotase alfa in an adult with benign prenatal hypophosphatasia: A case report (2022)
  • Author(s): Kato Hajime、Hidaka Naoko、Koga Minae、Kinoshita Yuka、Makita Noriko、Nangaku Masaomi、Ito Nobuaki
  • Journal Title: Bone Reports Volume: 16 Pages: 101163-101163
  • DOI: 10.1016/j.bonr.2021.101163
  • Peer Reviewed / Open Access
• [Journal Article] Incidence of complications in 25 adult patients with X-linked hypophosphatemia. (2021)
  • Author(s): Kato H, Koga M, Kinoshita Y, Taniguchi Y, Kobayashi H, Fukumoto S, Nangaku M, Makita N, Ito N.
  • Journal Title: J Clin Endocrinol Metab. Volume: - Issue: 9 Pages: e3682-e3692
  • DOI: 10.1210/clinem/dgab282
  • Peer Reviewed / Open Access
• [Journal Article] Clinical performance of a novel chemiluminescent enzyme immunoassay for FGF23 (2021)
  • Author(s): Ito Nobuaki、Kubota Takuo、Kitanaka Sachiko、Fujiwara Ikuma、Adachi Masanori、Takeuchi Yasuhiro、Yamagami Hitomi、Kimura Takehide、Shinoda Tatsuya、Minagawa Masanori、Okazaki Ryo、Ozono Keiichi、Seino Yoshiki、Fukumoto Seiji
  • Journal Title: Journal of Bone and Mineral Metabolism Volume: 39 Issue: 6 Pages: 1066-1075
  • DOI: 10.1007/s00774-021-01250-1
  • Peer Reviewed / Int'l Joint Research
• [Journal Article] Performance evaluation of the new chemiluminescent intact FGF23 assay relative to the existing assay system (2021)
  • Author(s): Kato Hajime、Hidaka Naoko、Koga Minae、Ogawa Noriyuki、Takahashi Shichihiro、Miyazaki Hiromi、Nangaku Masaomi、Makita Noriko、Ito Nobuaki
  • Journal Title: Journal of Bone and Mineral Metabolism Volume: 40 Issue: 1 Pages: 101-108
  • DOI: 10.1007/s00774-021-01258-7
  • Peer Reviewed / Open Access
• [Journal Article] Massive calcification around large joints in a patient subsequently diagnosed with adult-onset hypophosphatasia (2021)
  • Author(s): Koga M.、Kinoshita Y.、Kato H.、Kobayashi H.、Shinoda Y.、Nangaku M.、Makita N.、Dahir K. M.、Ito N.
  • Journal Title: Osteoporosis International Volume: 33 Issue: 2 Pages: 505-509
  • DOI: 10.1007/s00198-021-06145-5
  • Peer Reviewed / Open Access / Int'l Joint Research
• [Journal Article] Burosumab treatment in adults with X-linked hypophosphataemia: 96-week patient-reported outcomes and ambulatory function from a randomised phase 3 trial and open-label extension (2021)
  • Author(s): Briot Karine、Portale Anthony A、Brandi Maria Luisa、et al
  • Journal Title: RMD Open Volume: 7 Issue: 3 Pages: e001714-e001714
  • DOI: 10.1136/rmdopen-2021-001714
  • Peer Reviewed / Open Access / Int'l Joint Research
• [Journal Article] Altered Thyroid Function Tests Observed in Hypophosphatasia Patients Treated with Asfotase Alfa (2021)
  • Author(s): Kato Hajime、Hidaka Naoko、Koga Minae、Kinoshita Yuka、Nangaku Masaomi、Makita Noriko、Ito Nobuaki
  • Journal Title: International Journal of Endocrinology Volume: 2021 Pages: 1-5
  • DOI: 10.1155/2021/5492267
  • Peer Reviewed / Open Access
• [Journal Article] Induction of FGF23-related hypophosphatemic osteomalacia by alcohol consumption (2021)
  • Author(s): Hidaka Naoko、Kato Hajime、Koga Minae、Katsura Masaki、Oyama Yuko、Kinoshita Yuka、Fukumoto Seiji、Makita Noriko、Nangaku Masaomi、Ito Nobuaki
  • Journal Title: Bone Reports Volume: 15 Pages: 101144-101144
  • DOI: 10.1016/j.bonr.2021.101144
  • Peer Reviewed / Open Access
• [Presentation] 指定講演 12 成人低ホスファターゼ症 診療の現状 (2022)
  • Author(s): 伊東伸朗
  • Organizer: 第32回臨床内分泌代謝 Update
  • Invited
• [Presentation] Meet the Expert 5 骨軟化症およびその類縁疾患の診断と治療 (2022)
  • Author(s): 伊東伸朗
  • Organizer: 第32回臨床内分泌代謝 Update
• [Presentation] The background of XLH patients with burosumab treatment and its short term effectiveness and safety of:interim analysis of the SUNFLOWER study. (2022)
  • Author(s): 道上敏美
  • Organizer: THE 12TH BIENNIAL SCIENTIFIC MEETING OF THE ASIA PACIFIC PAEDIATRIC ENDOCRINE SOCIETY 2022
  • Int'l Joint Research
• [Presentation] Utility of multimodality approach including systemic FGF23 venous sampling in localizing phosphaturic mesenchymal tumors (2022)
  • Author(s): 加藤創生
  • Organizer: The American Society for Bone and Mineral Research 2022
  • Int'l Joint Research
• [Presentation] Clinical Course and Treatment Outcomes in Tumor-Induced Osteomalacia: A Retrospective Questionnaire Survey (2022)
  • Author(s): 日髙尚子
  • Organizer: The American Society for Bone and Mineral Research 2022
  • Int'l Joint Research
• [Presentation] Assessment of Ossification of the Spinal Ligaments in Acromegaly Patients (2022)
  • Author(s): 星野良朋
  • Organizer: The American Society for Bone and Mineral Research 2022
  • Int'l Joint Research
• [Presentation] モーニングセミナー2 様々な症状を示す成人低ホスファターゼ症 (2022)
  • Author(s): 伊東伸朗
  • Organizer: 第24回日本骨粗鬆症学会
  • Invited
• [Presentation] 教育講演6-2 小児骨疾患の成人期管理 -成人の立場から- 低リン血症性くる病、低ホスファターゼ症、骨形成不全症の成人期の課題とその管理 (2022)
  • Author(s): 伊東伸朗
  • Organizer: 第24回日本骨粗鬆症学会
  • Invited
• [Presentation] 教育講演 様々な症状を示す成人低ホスファターゼ症 (2022)
  • Author(s): 伊東伸朗
  • Organizer: 第22回日本内分泌学会四国支部学術集会
  • Invited
• [Presentation] ランチョンセミナー 成人低ホスファターゼ症の治療とモニタリング (2022)
  • Author(s): 伊東伸朗
  • Organizer: 第22回日本内分泌学会九州支部学術集会
  • Invited
• [Presentation] 成人XLHの歯科合併症に関する検討 (2022)
  • Author(s): 加藤創生
  • Organizer: 第24回日本骨粗鬆症学会
• [Presentation] OPLLにおけるENPP1遺伝子変異の頻度に関する検討 (2022)
  • Author(s): 加藤創生
  • Organizer: 第24回日本骨粗鬆症学会
• [Presentation] 骨形成不全症とその類縁疾患による二次性骨粗鬆症に対するロモソズマブの効果 (2022)
  • Author(s): 加藤創生
  • Organizer: 第24回日本骨粗鬆症学会
• [Presentation] ALPL変異保因者11人の臨床的特徴 (2022)
  • Author(s): 日髙尚子
  • Organizer: 第24回日本骨粗鬆症学会
• [Presentation] Clinical course of 88 tumor-induced osteomalacia patients (2022)
  • Author(s): 日髙尚子
  • Organizer: 第40回日本骨代謝学会学術集会
• [Presentation] Assessment of Ossification of the Spinal Ligaments in 10 Acromegaly Patients (2022)
  • Author(s): 星野良朋
  • Organizer: 第40回日本骨代謝学会学術集会
• [Presentation] 骨代謝疾患Update日本骨代謝学会協同企画 JES、JSBMRを介した本邦での腫瘍性骨軟化症の診療実態に関するアンケート調査 (2022)
  • Author(s): 伊東伸朗
  • Organizer: 第95回 日本内分泌学会学術集会
  • Invited
• [Presentation] 遺伝性FGF23関連低リン血症に合併する傍脊柱靭帯骨化症に関する検討 (2022)
  • Author(s): 加藤創生
  • Organizer: 第95回 日本内分泌学会学術集会
• [Presentation] アスフォターゼアルファによる甲状腺測定系への影響 (2022)
  • Author(s): 加藤創生
  • Organizer: 第95回 日本内分泌学会学術集会
• [Presentation] アルコール誘発性FGF23関連低リン血症性骨軟化症の2症例 (2022)
  • Author(s): 日髙尚子
  • Organizer: 第95回 日本内分泌学会学術集会
• [Presentation] 腫瘍性骨軟化症の全国病態調査 (2022)
  • Author(s): 日髙尚子
  • Organizer: 第95回 日本内分泌学会学術集会
• [Presentation] 19年間ファンコーニ症候群と診断されていたして治療を受けていた腫瘍性骨軟化症の一例 (2022)
  • Author(s): 堀越博文
  • Organizer: 第95回 日本内分泌学会学術集会
• [Presentation] Webセミナー6 骨粗鬆症や脊柱管狭窄症、関節リウマチと鑑別すべき疾患～腫瘍性骨軟化症～ (2022)
  • Author(s): 伊東伸朗
  • Organizer: 第55回 日本整形外科学会骨・軟部腫瘍学術集会
  • Invited
• [Presentation] 日本と韓国におけるXLH患者の主観的疾患評価 (2021)
  • Author(s): 伊東伸朗、西田弥生、他
  • Organizer: 第94回 日本内分泌学会学術集会
• [Presentation] 新規FGF23測定試薬の臨床検体を用いた性能評価 (2021)
  • Author(s): 加藤創生、伊東伸朗、他
  • Organizer: 第94回 日本内分泌学会学術集会
• [Presentation] X染色体連鎖性低リン血症性くる病成人患者における異所性骨化の頻度・重症度に関する検討 (2021)
  • Author(s): 加藤創生、伊東伸朗、他
  • Organizer: 第94回 日本内分泌学会学術集会
• [Presentation] 腫瘍性骨軟化症の原因腫瘍におけるFN1-FGFR1融合遺伝子の解析 (2021)
  • Author(s): 古家美奈絵、伊東伸朗、他
  • Organizer: 第94回 日本内分泌学会学術集会
• [Presentation] 低リン血症性疾患の診断と治療 (2021)
  • Author(s): 伊東伸朗
  • Organizer: 第94回 日本内分泌学会学術集会
  • Invited
• [Presentation] FGF23関連低リン血症性骨軟化症の病態・診断・治療－成人科の立場から－ (2021)
  • Author(s): 伊東伸朗
  • Organizer: 第94回 日本内分泌学会学術集会
  • Invited
• [Presentation] 無症候性原発性副甲状腺機能亢進症の管理 (2021)
  • Author(s): 伊東伸朗
  • Organizer: 第94回 日本内分泌学会学術集会
  • Invited
• [Presentation] Incidence and severity of ectopic ossifications in 25 adult patients with X-linked hypophosphatemia (2021)
  • Author(s): 加藤創生、伊東伸朗、他
  • Organizer: The European Calcified Tissue Society 2021
  • Int'l Joint Research
• [Presentation] Clinically suspected calcium pyrophosphate depositionCPPD disease in a case withof adult-onset hypophosphatemia (2021)
  • Author(s): 日髙尚子、伊東伸朗、他
  • Organizer: The American Society for Bone and Mineral Research 2021
  • Int'l Joint Research
• [Presentation] Two cases with heterozygous ENPP1 mutation presenting different skeletal manifestations (2021)
  • Author(s): 加藤創生、伊東伸朗、他
  • Organizer: The American Society for Bone and Mineral Research 2021
  • Int'l Joint Research
• [Presentation] X 染色体連鎖性低リン血症性くる病成人患者の異所性骨化および骨密度に関する検討 (2021)
  • Author(s): 加藤創生、伊東伸朗、他
  • Organizer: 第39回 日本骨代謝学会学術集会
• [Presentation] びまん性骨増殖性疾患がありにENPP1遺伝子のヘテロ接合型変異を認めた2例 (2021)
  • Author(s): 加藤創生、伊東伸朗、他
  • Organizer: 第39回 日本骨代謝学会学術集会
• [Presentation] 骨形成不全症とその類縁疾患による二次性骨粗鬆症に対するロモソズマブの効果 (2021)
  • Author(s): 加藤創生、伊東伸朗、他
  • Organizer: 第23回日本骨粗鬆症学会
• [Presentation] アルコール多飲・肝障害によるFGF23関連低リン血症性骨軟化症が疑われた一例 (2021)
  • Author(s): 日髙尚子、伊東伸朗、他
  • Organizer: 第23回日本骨粗鬆症学会
• [Presentation] 骨折を伴わず、多関節痛による著しいADL低下を認めた成人型低ホスファターゼ症の一例 (2021)
  • Author(s): 日髙尚子、伊東伸朗、他
  • Organizer: 第23回日本骨粗鬆症学会
• [Presentation] 多彩な症状を呈する低ホスファターゼ症への酵素補充療法の効果 (2021)
  • Author(s): 伊東伸朗
  • Organizer: 第39回 日本骨代謝学会学術集会
  • Invited
• [Presentation] TIO患者のアンケート調査 (2021)
  • Author(s): 伊東伸朗
  • Organizer: 第39回 日本骨代謝学会学術集会
  • Invited
• [Presentation] 骨ミネラル代謝異常疾患患者のトランジション 内科の立場から (2021)
  • Author(s): 伊東伸朗
  • Organizer: 第23回日本骨粗鬆症学会/第39回日本骨代謝学会学術集会
  • Invited
• [Presentation] 続発性骨粗鬆症の鑑別診断 (2021)
  • Author(s): 伊東伸朗
  • Organizer: 第23回日本骨粗鬆症学会
  • Invited
• [Presentation] Two cases with heterozygous ENPP1 mutation presenting different skeletal manifestations (2021)
  • Author(s): 加藤創生、伊東伸朗、他
  • Organizer: the joint Annual Scientific Meeting of the Endocrine Society of Australia (ESA), Society for Reproductive Biology (SRB), and the Australian and New Zealand Bone and Mineral Society (ANZBMS) 2021
  • Int'l Joint Research
• [Presentation] 骨代謝性疾患の診断・管理・医療連携 (2021)
  • Author(s): 伊東伸朗
  • Organizer: 第31回臨床内分泌代謝 Update
  • Invited
• [Remarks] 東京大学大学院医学系研究科 難治性骨疾患治療開発講座
  • URL: https://plaza.umin.ac.jp/bone-mineral-lab/
• [Remarks] 東大病院 内分泌骨ミネラル代謝研究グループ ホームページ
  • URL: https://u-tokyo-bone-mineral-lab.jp/
• [Remarks] 東大病院 内分泌骨ミネラル代謝研究グループ ホームページ
  • URL: https://u-tokyo-bone-mineral-lab.jp/