ACVR1-activating mutation causes neuropathic pain and sensory neuron hyperexcitability in humans

Authors: Yu, Xiaobing, Ton, Amy N., Niu, Zejun, Morales, Blanca M., Chen, Jiadong, Braz, Joao, Lai, Michael H., Barruet, Emilie, Liu, Hongju, Cheung, Kin, Ali, Syed, Chan, Tea, Bigay, Katherine, Ho, Jennifer, Nikolli, Ina, Hansberry, Steven, Wentworth, Kelly, Kriegstein, Arnold, Basbaum, Allan, Hsiao, Edward C.

Source: Pain. 164(1):43-58

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100188070473.0x000001&indx=1&recIds=ETOCvdc_100188070473.0x000001

Reduction of New Heterotopic Ossification (HO) in the Open‐Label, Phase 3 MOVE Trial of Palovarotene for Fibrodysplasia Ossificans Progressiva (FOP)

Authors: Pignolo, Robert J., Hsiao, Edward C., Al Mukaddam, Mona, Baujat, Geneviève, Berglund, Staffan K., Brown, Matthew A., Cheung, Angela M., De Cunto, Carmen, Delai, Patricia, Haga, Nobuhiko, Kannu, Peter, Keen, Richard, Le Quan Sang, Kim‐Hanh, Mancilla, Edna E., Marino, Rose, Strahs, Andrew, Kaplan, Frederick S.

Source: Journal of bone and mineral research. 38(3):381-394

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100186842940.0x000001&indx=1&recIds=ETOCvdc_100186842940.0x000001

The natural history of fibrodysplasia ossificans progressiva: A prospective, global 36-month study

Authors: Pignolo, Robert J., Baujat, Geneviève, Brown, Matthew A., De Cunto, Carmen, Hsiao, Edward C., Keen, Richard, Al Mukaddam, Mona, Le Quan Sang, Kim-Hanh, Wilson, Amy, Marino, Rose, Strahs, Andrew, Kaplan, Frederick S.

Source: In Genetics in Medicine December 2022 24(12):2422-2433

Functional Testing of Bone Morphogenetic Protein (BMP) Pathway Variants Identified on Whole‐Exome Sequencing in a Patient with Delayed‐Onset Fibrodysplasia Ossificans Progressiva (FOP) Using ACVR1R206H‐Specific Human Cellular and Zebrafish Models

Authors: Wentworth, Kelly L., Lalonde, Robert L., Groppe, Jay C., Brewer, Niambi, Moody, Tania, Hansberry, Steven, Taylor, Kimberly E., Shore, Eileen M., Kaplan, Frederick S., Pignolo, Robert J., Yelick, Pamela C., Hsiao, Edward C.

Source: Journal of bone and mineral research. 37(11):2058-2076

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100191739993.0x000001&indx=1&recIds=ETOCvdc_100191739993.0x000001

Palovarotene for Fibrodysplasia Ossificans Progressiva (FOP): Results of a Randomized, Placebo‐Controlled, Double‐Blind Phase 2 Trial

Authors: Pignolo, Robert J., Baujat, Geneviève, Hsiao, Edward C., Keen, Richard, Wilson, Amy, Packman, Jeff, Strahs, Andrew L., Grogan, Donna R., Kaplan, Frederick S.

Source: Journal of bone and mineral research. 37(10):1891-1902

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100163317788.0x000001&indx=1&recIds=ETOCvdc_100163317788.0x000001

Identification of Risk Factors in the Development of Heterotopic Ossification After Primary Total Hip Arthroplasty

Authors: Singh, Sukhmani, Morshed, Saam, Motamedi, Daria, Kidane, Joseph, Paul, Alexandra, Hsiao, Edward C, Wentworth, Kelly L

Source: Journal of clinical endocrinology & metabolism. 107(9):e3944-e3952

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100158283196.0x000001&indx=1&recIds=ETOCvdc_100158283196.0x000001

Gene Therapy for Fibrodysplasia Ossificans Progressiva: Feasibility and Obstacles

Authors: Eekhoff, Elisabeth M.W., de Ruiter, Ruben D., Smilde, Bernard J., Schoenmaker, Ton, de Vries, Teun J., Netelenbos, Coen, Hsiao, Edward C., Scott, Christiaan, Haga, Nobuhiko, Grunwald, Zvi, De Cunto, Carmen L., di Rocco, Maja, Delai, Patricia L. R., Diecidue, Robert J., Madhuri, Vrisha, Cho, Tae-Joon, Morhart, Rolf, Friedman, Clive S., Zasloff, Michael, Pals, Gerard, Shim, Jae-Hyuck, Gao, Guangping, Kaplan, Frederick, Pignolo, Robert J., Micha, Dimitra

Source: Human gene therapy. 33(15/16):782-788

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100157883944.0x000001&indx=1&recIds=ETOCvdc_100157883944.0x000001

Palovarotene (Sohonos), a synthetic retinoid for reducing new heterotopic ossification in fibrodysplasia ossificans progressiva: history, present, and future.

Authors: Hsiao, Edward C, Pacifici, Maurizio

Source: JBMR Plus; Jan2025, Vol. 9 Issue 1, p1-11, 11p

Subject Terms: FIBRODYSPLASIA ossificans progressiva, RETINOIDS, RETINOIC acid receptors, CLINICAL trials, VITAMIN A, DYSPLASIA

ACVR1R206H extends inflammatory responses in human induced pluripotent stem cell-derived macrophages

Authors: Matsuo, Koji, Lepinski, Abigail, Chavez, Robert D., Barruet, Emilie, Pereira, Ashley, Moody, Tania A., Ton, Amy N., Sharma, Aditi, Hellman, Judith, Tomoda, Kiichiro, Nakamura, Mary C., Hsiao, Edward C.

Source: In Bone December 2021 153

Off-on-off-on use of imatinib in three children with fibrodysplasia ossificans progressiva

Authors: Kaplan, Frederick S., Teachey, David T., Andolina, Jeffrey R., Siegel, David M., Mancilla, Edna E., Hsiao, Edward C., Al Mukaddam, Mona, Rocke, David M., Pignolo, Robert J.

Source: In Bone September 2021 150

Virtual Interviews at Graduate Medical Education Training Programs: Determining Evidence-Based Best Practices

Authors: Huppert, Laura A., Hsiao, Edward C., Cho, Kerry C., Marquez, Carina, Chaudhry, Rafia I., Frank, James, Goglin, Sarah E., Hsu, Gerald, Kathpalia, Priya, Khanna, Raman

Source: Academic medicine. 96(8):1137-1145

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCRN627779768&indx=1&recIds=ETOCRN627779768

Personalized Approaches to Spine Surgery.

Authors: PATEL, ARATI, DADA, ABRAHAM, SAGGI, SATVIR, YAMADA, HUNTER, AMBATI, VARDHAAN S., GOLDSTEIN, ELIANNA, HSIAO, EDWARD C., MUMMANENI, PRAVEEN V.

Source: International Journal of Spine Surgery; 12/1/2024, Vol. 18 Issue 6, p676-693, 18p

Subject Terms: BONE density, SPINAL implants, PAIN medicine, ARTIFICIAL intelligence, MEDICAL care

Self-reported baseline phenotypes from the International Fibrodysplasia Ossificans Progressiva (FOP) Association Global Registry

Authors: Pignolo, Robert J., Cheung, Kin, Kile, Sammi, Fitzpatrick, Mary Anne, De Cunto, Carmen, Al Mukaddam, Mona, Hsiao, Edward C., Baujat, Genevieve, Delai, Patricia, Eekhoff, Elisabeth M.W., Di Rocco, Maja, Grunwald, Zvi, Haga, Nobuhiko, Keen, Richard, Levi, Benjamin, Morhart, Rolf, Scott, Christiaan, Sherman, Adam, Zhang, Keqin, Kaplan, Fredrick S.

Source: In Bone May 2020 134

Human iPSC‐Derived Proinflammatory Macrophages cause Insulin Resistance in an Isogenic White Adipose Tissue Microphysiological System

Authors: Qi, Lin, Matsuo, Koji, Pereira, Ashley, Lee, Yue Tung, Zhong, Fenmiao, He, Yuchen, Zushin, Peter‐James H., Gröger, Marko, Sharma, Aditi, Willenbring, Holger, Hsiao, Edward C., Stahl, Andreas

Source: Small. 19(34)

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100191102726.0x000001&indx=1&recIds=ETOCvdc_100191102726.0x000001

EML4::ALK fusions in complex lymphatic malformations

Authors: Apsel Winger, Beth, Devine, Walter Patrick, Hsiao, Edward C., Zapala, Matthew, Van Ziffle, Jessica, Gupta, Nalin, Frieden, Ilona J., Shimano, Kristin A.

Source: Pediatric blood & cancer. 70(9)

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100188348346.0x000001&indx=1&recIds=ETOCvdc_100188348346.0x000001

Virtual Interviews at Graduate Medical Education Training Programs: Determining Evidence-Based Best Practices

Authors: Huppert, Laura A., Hsiao, Edward C., Cho, Kerry C., Marquez, Carina, Chaudhry, Rafia I., Frank, James, Goglin, Sarah E., Hsu, Gerald, Kathpalia, Priya, Khanna, Raman, Kompala, Tejaswi, Rao, Madhu N., Bower, Bree A., Trafas, Vanessa, Santhosh, Lekshmi, Schwartz, Brian S., Babik, Jennifer M.

Source: Academic medicine. 96(8):1137-1145

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100169885581.0x000001&indx=1&recIds=ETOCvdc_100169885581.0x000001

Adipocyte inflammation is the primary driver of hepatic insulin resistance in a human iPSC-based microphysiological system.

Authors: Qi, Lin, Groeger, Marko, Sharma, Aditi, Goswami, Ishan, Chen, Erzhen, Zhong, Fenmiao, Ram, Apsara, Healy, Kevin, Hsiao, Edward C., Willenbring, Holger, Stahl, Andreas

Source: Nature Communications; 9/12/2024, Vol. 15 Issue 1, p1-12, 12p

Subject Terms: GLUCAGON-like peptide-1 receptor, MICROPHYSIOLOGICAL systems, TYPE 2 diabetes, PLURIPOTENT stem cells, GLUCAGON-like peptide-1 agonists, FAT cells, ADIPOSE tissues

Long-term use of interleukin-1 inhibitors reduce flare activity in patients with fibrodysplasia ossificans progressiva.

Authors: Haviv, Ruby, Zeitlin, Leonid, Moshe, Veronica, Ziv, Amit, Rabinowicz, Noa, Benedetti, Fabrizio De, Prencipe, Giusi, Matteo, Valentina, Cunto, Carmen Laura De, Hsiao, Edward C, Uziel, Yosef

Source: Rheumatology; Sep2024, Vol. 63 Issue 9, p2597-2604, 8p

Subject Terms: FIBRODYSPLASIA ossificans progressiva, DISEASE exacerbation, RESEARCH funding, HEALTH status indicators, VISUAL analog scale, QUESTIONNAIRES, TREATMENT duration, TREATMENT effectiveness, BLOOD plasma, PAIN, RESEARCH, CASE studies, DISEASE relapse, HEALTH outcome assessment, INTERLEUKIN-1, GLUCOCORTICOIDS, INTERLEUKINS, WELL-being, CHEMICAL inhibitors

Inflammation in Fibrodysplasia Ossificans Progressiva and Other Forms of Heterotopic Ossification

Authors: Matsuo, Koji, Chavez, Robert Dalton, Barruet, Emilie, Hsiao, Edward C.

Source: Current osteoporosis reports. 17(6):387-394

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100128364797.0x000001&indx=1&recIds=ETOCvdc_100128364797.0x000001

Special considerations for clinical trials in fibrodysplasia ossificans progressiva (FOP)

Authors: Hsiao, Edward C., Di Rocco, Maja, Cali, Amanda, Zasloff, Michael, Al Mukaddam, Mona, Pignolo, Robert J., Grunwald, Zvi, Netelenbos, Coen, Keen, Richard, Baujat, Genevieve, Brown, Matthew A., Cho, Tae‐Joon, De Cunto, Carmen, Delai, Patricia, Haga, Nobuhiko, Morhart, Rolf, Scott, Christiaan, Zhang, Keqin, Diecidue, Robert J., Friedman, Clive S., Kaplan, Fredrick S., Eekhoff, Elisabeth M.W.

Source: British journal of clinical pharmacology. 85(6):1199-1207

Availability: http://explore.bl.uk/primo_library/libweb/action/display.do?tabs=detailsTab&gathStatTab=true&ct=display&fn=search&doc=ETOCvdc_100085008533.0x000001&indx=1&recIds=ETOCvdc_100085008533.0x000001