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Expert Opinion on Orphan Drugs Volume 5, 2017 - Issue 4
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Editorial

Hard targets for a second skeleton: therapeutic horizons for fibrodysplasia ossificans progressiva (FOP)

Frederick S. KaplanCenter for Research in FOP and Related Disorders, The Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USACorrespondence[email protected]
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,
Robert J. PignoloDivision of Geriatric Medicine & Gerontology, Mayo Clinic College of Medicine, Rochester, MN, USAView further author information
,
Mona M. Al MukaddamDepartment of Medicine, Division of Endocrinology, Diabetes and Metabolism, The Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USAView further author information
&
Eileen M. ShoreCenter for Research in FOP and Related Disorders, The Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USAView further author information
Pages 291-294 | Received 06 Feb 2017, Accepted 06 Mar 2017, Published online: 17 Mar 2017

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Robert J. Pignolo & Frederick S. Kaplan. (2020) Druggable targets, clinical trial design and proposed pharmacological management in fibrodysplasia ossificans progressiva. Expert Opinion on Orphan Drugs 8:4, pages 101-109.
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Articles from other publishers (27)

Garima Nagar, Pooja Mittal, Shradheya R R Gupta, Monika Pahuja, Manisha Sanger, Ruby Mishra, Archana Singh & Indrakant Kumar Singh. (2023) Multi-omics therapeutic perspective on ACVR1 gene: from genetic alterations to potential targeting. Briefings in Functional Genomics 22:2, pages 123-142.
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Robert J. Pignolo, Edward C. Hsiao, Mona Al Mukaddam, Geneviève Baujat, Staffan K. Berglund, Matthew A. Brown, Angela M. Cheung, Carmen De Cunto, Patricia Delai, Nobuhiko Haga, Peter Kannu, Richard Keen, Kim‐Hanh Le Quan Sang, Edna E. Mancilla, Rose Marino, Andrew Strahs & Frederick S. Kaplan. (2023) Reduction of New Heterotopic Ossification ( HO ) in the Open‐Label , Phase 3 MOVE Trial of Palovarotene for Fibrodysplasia Ossificans Progressiva ( FOP ) . Journal of Bone and Mineral Research 38:3, pages 381-394.
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Favour Felix-Ilemhenbhio, George A. E. Pickering, Endre Kiss-Toth & Jeremy Mark Wilkinson. (2022) Pathophysiology and Emerging Molecular Therapeutic Targets in Heterotopic Ossification. International Journal of Molecular Sciences 23:13, pages 6983.
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Haitao Wang, Qiang Zhang, Frederick S. Kaplan & Robert J. Pignolo. (2021) Clearance of Senescent Cells From Injured Muscle Abrogates Heterotopic Ossification in Mouse Models of Fibrodysplasia Ossificans Progressiva. Journal of Bone and Mineral Research 37:1, pages 95-107.
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Sarah E. Warner, Frederick S. Kaplan, Robert J. Pignolo, Stacy E. Smith, Edward C. Hsiao, Carmen De Cunto, Maja Di Rocco, Kathleen Harnett, Donna Grogan & Harry K. Genant. (2021) Whole-body Computed Tomography Versus Dual Energy X‑ray Absorptiometry for Assessing Heterotopic Ossification in Fibrodysplasia Ossificans Progressiva. Calcified Tissue International 109:6, pages 615-625.
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Deborah Ribeiro NascimentoSuzana Lopes Bomfim BalaniucDurval Batista PalharesAdam UnderwoodMarilene Garcia PalharesFabiana AlvesFrancisco Oliveira VieiraElaine Maria Souza-FagundesLiane De Rosso GiulianiPaula Cristhina Niz XavierHelen Lima Del PuertoRobson Augusto Souza SantosAmy MilstedJose Mauro BrumIandara Schettert SilvaAlmir Sousa Martins. (2021) Rare and intractable fibrodysplasia ossificans progressiva shows different PBMC phenotype possibly modulated by ascorbic acid and propranolol treatment. Intractable & Rare Diseases Research 10:3, pages 179-189.
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Lovorka Grgurevic, Rudjer Novak, Stela Hrkac, Grgur Salai & Simeon Grazio. (2021) Post-COVID-19 exacerbation of fibrodysplasia ossificans progressiva with multiple flare-ups and extensive heterotopic ossification in a 45-year-old female patient. Rheumatology International 41:8, pages 1495-1501.
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Diana Martín‐García, O. Will Towler, Meiqi Xu, Osmany Alfonso‐Hernández, Paula R. Oliveira, Marleny Alonso‐Clavo, Eileen M. Shore & Frederick S. Kaplan. (2021) Nonclassic fibrodysplasia ossificans progressiva: A child from Angola with an ACVR1 G328E variant . American Journal of Medical Genetics Part A 185:8, pages 2572-2575.
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Mona Al Mukaddam & Robert J Pignolo. 2005. Encyclopedia of Life Sciences. Encyclopedia of Life Sciences 1 7 .
Frederick S. Kaplan, Mona Al Mukaddam, Alexandra Stanley, O. Will Towler & Eileen M. Shore. (2020) Fibrodysplasia ossificans progressiva (FOP): A disorder of osteochondrogenesis. Bone 140, pages 115539.
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Łukasz Pulik, Bartosz Mierzejewski, Maria A. Ciemerych, Edyta Brzóska & Paweł Łęgosz. (2020) The Survey of Cells Responsible for Heterotopic Ossification Development in Skeletal Muscles—Human and Mouse Models. Cells 9:6, pages 1324.
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Robert J. Pignolo, Kin Cheung, Sammi Kile, Mary Anne Fitzpatrick, Carmen De Cunto, Mona Al Mukaddam, Edward C. Hsiao, Genevieve Baujat, Patricia Delai, Elisabeth M.W. Eekhoff, Maja Di Rocco, Zvi Grunwald, Nobuhiko Haga, Richard Keen, Benjamin Levi, Rolf Morhart, Christiaan Scott, Adam Sherman, Keqin Zhang & Fredrick S. Kaplan. (2020) Self-reported baseline phenotypes from the International Fibrodysplasia Ossificans Progressiva (FOP) Association Global Registry. Bone 134, pages 115274.
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Robert J. Pignolo, Haitao Wang & Frederick S. Kaplan. (2020) Fibrodysplasia Ossificans Progressiva (FOP): A Segmental Progeroid Syndrome. Frontiers in Endocrinology 10.
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Mustafa A. M. Salih. 2020. Clinical Child Neurology. Clinical Child Neurology 1377 1392 .
Lovorka Grgurević, Ruđer Novak, Vladimir Trkulja, Lejla Ferhatović Hamzić, Stela Hrkač, Simeon Grazio & Marija Santini. (2019) Elevated plasma RANTES in fibrodysplasia ossificans progressiva – A novel therapeutic target?. Medical Hypotheses 131, pages 109313.
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Hui Lin, Fuli Shi, Jiayu Gao & Ping Hua. (2019) The role of Activin A in fibrodysplasia ossificans progressiva: a prominent mediator. Bioscience Reports 39:8.
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Fuli Shi, Jiayu Gao, Junrong Zou, Ying Ying & Hui Lin. (2019) Targeting heterotopic ossification by inhibiting activin receptor‑like kinase 2 function (Review). Molecular Medicine Reports.
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Zoran Gucev, Velibor Tasic, Dijana Plaseska‐Karanfilska, Marija Dimishkovska, Nevenka Laban, Zoran Bozinovski, Marko Kostovski, Alek Saveski, Momir Polenakovic, O. Will Towler, Eileen M. Shore & Frederick S. Kaplan. (2019) Severe digital malformations in a rare variant of fibrodysplasia ossificans progressiva. American Journal of Medical Genetics Part A.
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Bin Ren, Volker M. Betz, Christian Thirion, Michael Salomon, Volkmar Jansson, Peter E. Müller & Oliver B. Betz. (2018) Osteoinduction within BMP-2 transduced muscle tissue fragments with and without a fascia layer: implications for bone tissue engineering. Gene Therapy 26:1-2, pages 16-28.
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O. Rohleder, P. Mudry, J. Neradil, H. Noskova, O. Slaby & J. Sterba. (2018) Letter to Editor: F.S. Kaplan, et al., Early clinical observations on the use of imatinibmesylate in FOP: A report of seven cases, Bone (2017). Bone 116, pages 171.
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Volker M. Betz, Bin Ren, Carolin Messmer, Volkmar Jansson, Oliver B. Betz & Peter E. Müller. (2018) Bone morphogenetic protein-2 is a stronger inducer of osteogenesis within muscle tissue than heterodimeric bone morphogenetic protein-2/6 and -2/7: Implications for expedited gene-enhanced bone repair. The Journal of Gene Medicine 20:9, pages e3042.
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John B. Lees-Shepard & David J. Goldhamer. (2018) Stem cells and heterotopic ossification: Lessons from animal models. Bone 109, pages 178-186.
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Haitao Wang, Edward M. Behrens, Robert J. Pignolo & Frederick S. Kaplan. (2018) ECSIT links TLR and BMP signaling in FOP connective tissue progenitor cells. Bone 109, pages 201-209.
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Frederick S. Kaplan, Leonid Zeitlin, Stephen P. Dunn, Shira Benor, David Hagin, Mona Al Mukaddam & Robert J. Pignolo. (2018) Acute and chronic rapamycin use in patients with Fibrodysplasia Ossificans Progressiva: A report of two cases. Bone 109, pages 281-284.
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Frederick S. Kaplan, Mona Al Mukaddam & Robert J. Pignolo. (2018) Acute unilateral hip pain in fibrodysplasia ossificans progressiva (FOP). Bone 109, pages 115-119.
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Frederick S. Kaplan, Jeffrey R. Andolina, Peter C. Adamson, David T. Teachey, Jerry Z. Finklestein, David H. Ebb, Benjamin Whitehead, Benjamin Jacobs, David M. Siegel, Richard Keen, Edward Hsiao & Robert J. Pignolo. (2018) Early clinical observations on the use of imatinib mesylate in FOP: A report of seven cases. Bone 109, pages 276-280.
Crossref
Zijuan Qi, Jing Luan, Xiaoyan Zhou, Yazhou Cui & Jinxiang Han. (2017) Fibrodysplasia ossificans progressiva: Basic understanding and experimental models. Intractable & Rare Diseases Research 6:4, pages 242-248.
Crossref

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