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Skeletal malformations and developmental arthropathy in individuals who have fibrodysplasia ossificans progressiva.

Towler OW, Shore EM, Kaplan FS.

Bone. 2020 Jan;130:115116. doi: 10.1016/j.bone.2019.115116. Epub 2019 Oct 23.


Correction to: Natural history of fibrodysplasia ossificans progressiva: cross-sectional analysis of annotated baseline phenotypes.

Pignolo RJ, Baujat G, Brown MA, De Cunto C, Di Rocco M, Hsiao EC, Keen R, Al Mukaddam M, Sang KLQ, Wilson A, White B, Grogan DR, Kaplan FS.

Orphanet J Rare Dis. 2019 May 23;14(1):113. doi: 10.1186/s13023-019-1096-3.


Natural history of fibrodysplasia ossificans progressiva: cross-sectional analysis of annotated baseline phenotypes.

Pignolo RJ, Baujat G, Brown MA, De Cunto C, Di Rocco M, Hsiao EC, Keen R, Al Mukaddam M, Sang KLQ, Wilson A, White B, Grogan DR, Kaplan FS.

Orphanet J Rare Dis. 2019 May 3;14(1):98. doi: 10.1186/s13023-019-1068-7. Erratum in: Orphanet J Rare Dis. 2019 May 23;14(1):113.


Severe digital malformations in a rare variant of fibrodysplasia ossificans progressiva.

Gucev Z, Tasic V, Plaseska-Karanfilska D, Dimishkovska M, Laban N, Bozinovski Z, Kostovski M, Saveski A, Polenakovic M, Towler OW, Shore EM, Kaplan FS.

Am J Med Genet A. 2019 Jul;179(7):1310-1314. doi: 10.1002/ajmg.a.61153. Epub 2019 Apr 22.


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

Hsiao EC, Di Rocco M, Cali A, Zasloff M, Al Mukaddam M, Pignolo RJ, Grunwald Z, Netelenbos C, Keen R, Baujat G, Brown MA, Cho TJ, De Cunto C, Delai P, Haga N, Morhart R, Scott C, Zhang K, Diecidue RJ, Friedman CS, Kaplan FS, Eekhoff EMW.

Br J Clin Pharmacol. 2019 Jun;85(6):1199-1207. doi: 10.1111/bcp.13777. Epub 2018 Nov 6. Review.


Heterotopic Ossification: The Keys to the Kingdom.

Kaplan FS, Pignolo RJ, Shore EM.

Bone. 2018 Apr;109:1-2. doi: 10.1016/j.bone.2018.03.001. No abstract available.


A case report of mesenteric heterotopic ossification: Histopathologic and genetic findings.

Amalfitano M, Fyfe B, Thomas SV, Egan KP, Xu M, Smith AG, Kaplan FS, Shore EM, Pignolo RJ.

Bone. 2018 Apr;109:56-60. doi: 10.1016/j.bone.2018.01.006. Epub 2018 Jan 7.


ECSIT links TLR and BMP signaling in FOP connective tissue progenitor cells.

Wang H, Behrens EM, Pignolo RJ, Kaplan FS.

Bone. 2018 Apr;109:201-209. doi: 10.1016/j.bone.2017.12.024. Epub 2017 Dec 27.


Acute and chronic rapamycin use in patients with Fibrodysplasia Ossificans Progressiva: A report of two cases.

Kaplan FS, Zeitlin L, Dunn SP, Benor S, Hagin D, Al Mukaddam M, Pignolo RJ.

Bone. 2018 Apr;109:281-284. doi: 10.1016/j.bone.2017.12.011. Epub 2017 Dec 11.


Prevalence and risk factors for kidney stones in fibrodysplasia ossificans progressiva.

Gupta RR, Delai PLR, Glaser DL, Rocke DM, Al Mukaddam M, Pignolo RJ, Kaplan FS.

Bone. 2018 Apr;109:120-123. doi: 10.1016/j.bone.2017.12.010. Epub 2017 Dec 11.


Reply to: Macrophages Driving Heterotopic Ossification: Convergence of Genetically-Driven and Trauma-Driven Mechanisms.

Convente MR, Chakkalakal SA, Yang E, Caron RJ, Zhang D, Kambayashi T, Kaplan FS, Shore EM.

J Bone Miner Res. 2018 Feb;33(2):367-368. doi: 10.1002/jbmr.3349. Epub 2017 Dec 27. No abstract available.


Ablation of Gsα signaling in osteoclast progenitor cells adversely affects skeletal bone maintenance.

Ramaswamy G, Fong J, Brewer N, Kim H, Zhang D, Choi Y, Kaplan FS, Shore EM.

Bone. 2018 Apr;109:86-90. doi: 10.1016/j.bone.2017.11.019. Epub 2017 Nov 26.


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

Kaplan FS, Pignolo RJ, Al Mukaddam MM, Shore EM.

Expert Opin Orphan Drugs. 2017;5(4):291-294. doi: 10.1080/21678707.2017.1304211. Epub 2017 Mar 17. No abstract available.


Clinical-pathological correlations in three patients with fibrodysplasia ossificans progressiva.

Wentworth KL, Bigay K, Chan TV, Ho JP, Morales BM, Connor J, Brooks E, Shahriar Salamat M, Sanchez HC, Wool G, Pignolo RJ, Kaplan FS, Hsiao EC.

Bone. 2018 Apr;109:104-110. doi: 10.1016/j.bone.2017.10.009. Epub 2017 Oct 13.


Depletion of Mast Cells and Macrophages Impairs Heterotopic Ossification in an Acvr1R206H Mouse Model of Fibrodysplasia Ossificans Progressiva.

Convente MR, Chakkalakal SA, Yang E, Caron RJ, Zhang D, Kambayashi T, Kaplan FS, Shore EM.

J Bone Miner Res. 2018 Feb;33(2):269-282. doi: 10.1002/jbmr.3304. Epub 2018 Jan 3.


Cartilage-derived retinoic acid-sensitive protein (CD-RAP): A stage-specific biomarker of heterotopic endochondral ossification (HEO) in fibrodysplasia ossificans progressiva (FOP).

Lindborg CM, Brennan TA, Wang H, Kaplan FS, Pignolo RJ.

Bone. 2018 Apr;109:153-157. doi: 10.1016/j.bone.2017.09.016. Epub 2017 Sep 28.


Clinical staging of Fibrodysplasia Ossificans Progressiva (FOP).

Pignolo RJ, Kaplan FS.

Bone. 2018 Apr;109:111-114. doi: 10.1016/j.bone.2017.09.014. Epub 2017 Sep 21.


Mast cell inhibition as a therapeutic approach in fibrodysplasia ossificans progressiva (FOP).

Brennan TA, Lindborg CM, Bergbauer CR, Wang H, Kaplan FS, Pignolo RJ.

Bone. 2018 Apr;109:259-266. doi: 10.1016/j.bone.2017.08.023. Epub 2017 Aug 26.


Imaging assessment of fibrodysplasia ossificans progressiva: Qualitative, quantitative and questionable.

Al Mukaddam M, Rajapakse CS, Pignolo RJ, Kaplan FS, Smith SE.

Bone. 2018 Apr;109:147-152. doi: 10.1016/j.bone.2017.08.011. Epub 2017 Aug 16.


Acute unilateral hip pain in fibrodysplasia ossificans progressiva (FOP).

Kaplan FS, Al Mukaddam M, Pignolo RJ.

Bone. 2018 Apr;109:115-119. doi: 10.1016/j.bone.2017.08.009. Epub 2017 Aug 16.


Early clinical observations on the use of imatinib mesylate in FOP: A report of seven cases.

Kaplan FS, Andolina JR, Adamson PC, Teachey DT, Finklestein JZ, Ebb DH, Whitehead B, Jacobs B, Siegel DM, Keen R, Hsiao E, Pignolo RJ.

Bone. 2018 Apr;109:276-280. doi: 10.1016/j.bone.2017.07.019. Epub 2017 Jul 20.


Vascular ossification: Pathology, mechanisms, and clinical implications.

Fuery MA, Liang L, Kaplan FS, Mohler ER 3rd.

Bone. 2018 Apr;109:28-34. doi: 10.1016/j.bone.2017.07.006. Epub 2017 Jul 5. Review.


Joint-specific risk of impaired function in fibrodysplasia ossificans progressiva (FOP).

Pignolo RJ, Durbin-Johnson BP, Rocke DM, Kaplan FS.

Bone. 2018 Apr;109:124-133. doi: 10.1016/j.bone.2017.06.009. Epub 2017 Jun 13.


International physician survey on management of FOP: a modified Delphi study.

Di Rocco M, Baujat G, Bertamino M, Brown M, De Cunto CL, Delai PLR, Eekhoff EMW, Haga N, Hsiao E, Keen R, Morhart R, Pignolo RJ, Kaplan FS.

Orphanet J Rare Dis. 2017 Jun 12;12(1):110. doi: 10.1186/s13023-017-0659-4.


Longitudinal patient-reported mobility assessment in fibrodysplasia ossificans progressiva (FOP).

Kaplan FS, Al Mukaddam M, Pignolo RJ.

Bone. 2018 Apr;109:158-161. doi: 10.1016/j.bone.2017.06.005. Epub 2017 Jun 6.


The congenital great toe malformation of fibrodysplasia ossificans progressiva? - A close call.

Towler OW, Shore EM, Xu M, Bamford A, Anderson I, Pignolo RJ, Kaplan FS.

Eur J Med Genet. 2017 Jul;60(7):399-402. doi: 10.1016/j.ejmg.2017.04.013. Epub 2017 May 1.


A cumulative analogue joint involvement scale (CAJIS) for fibrodysplasia ossificans progressiva (FOP).

Kaplan FS, Al Mukaddam M, Pignolo RJ.

Bone. 2017 Aug;101:123-128. doi: 10.1016/j.bone.2017.04.015. Epub 2017 Apr 29.


Gsα Controls Cortical Bone Quality by Regulating Osteoclast Differentiation via cAMP/PKA and β-Catenin Pathways.

Ramaswamy G, Kim H, Zhang D, Lounev V, Wu JY, Choi Y, Kaplan FS, Pignolo RJ, Shore EM.

Sci Rep. 2017 Mar 24;7:45140. doi: 10.1038/srep45140.


Analog Method for Radiographic Assessment of Heterotopic Bone in Fibrodysplasia Ossificans Progressiva.

Rajapakse CS, Lindborg C, Wang H, Newman BT, Kobe EA, Chang G, Shore EM, Kaplan FS, Pignolo RJ.

Acad Radiol. 2017 Mar;24(3):321-327. doi: 10.1016/j.acra.2016.10.010. Epub 2016 Dec 15.


ACVR1-Fc suppresses BMP signaling and chondro-osseous differentiation in an in vitro model of Fibrodysplasia ossificans progressiva.

Pang J, Zuo Y, Chen Y, Song L, Zhu Q, Yu J, Shan C, Cai Z, Hao J, Kaplan FS, Shore EM, Zhang K.

Bone. 2016 Nov;92:29-36. doi: 10.1016/j.bone.2016.07.023. Epub 2016 Aug 2.


Cellular Hypoxia Promotes Heterotopic Ossification by Amplifying BMP Signaling.

Wang H, Lindborg C, Lounev V, Kim JH, McCarrick-Walmsley R, Xu M, Mangiavini L, Groppe JC, Shore EM, Schipani E, Kaplan FS, Pignolo RJ.

J Bone Miner Res. 2016 Sep;31(9):1652-65. doi: 10.1002/jbmr.2848. Epub 2016 Apr 20.


The Natural History of Flare-Ups in Fibrodysplasia Ossificans Progressiva (FOP): A Comprehensive Global Assessment.

Pignolo RJ, Bedford-Gay C, Liljesthröm M, Durbin-Johnson BP, Shore EM, Rocke DM, Kaplan FS.

J Bone Miner Res. 2016 Mar;31(3):650-6. doi: 10.1002/jbmr.2728. Epub 2015 Nov 14.


Palovarotene Inhibits Heterotopic Ossification and Maintains Limb Mobility and Growth in Mice With the Human ACVR1(R206H) Fibrodysplasia Ossificans Progressiva (FOP) Mutation.

Chakkalakal SA, Uchibe K, Convente MR, Zhang D, Economides AN, Kaplan FS, Pacifici M, Iwamoto M, Shore EM.

J Bone Miner Res. 2016 Sep;31(9):1666-75. doi: 10.1002/jbmr.2820. Epub 2016 Mar 12.


Granting immunity to FOP and catching heterotopic ossification in the Act.

Kaplan FS, Pignolo RJ, Shore EM.

Semin Cell Dev Biol. 2016 Jan;49:30-6. doi: 10.1016/j.semcdb.2015.12.013. Epub 2015 Dec 17. Review.


Multi-system involvement in a severe variant of fibrodysplasia ossificans progressiva (ACVR1 c.772G>A; R258G): A report of two patients.

Kaplan FS, Kobori JA, Orellana C, Calvo I, Rosello M, Martinez F, Lopez B, Xu M, Pignolo RJ, Shore EM, Groppe JC.

Am J Med Genet A. 2015 Oct;167A(10):2265-71. doi: 10.1002/ajmg.a.37205. Epub 2015 Jun 11.


The immunological contribution to heterotopic ossification disorders.

Convente MR, Wang H, Pignolo RJ, Kaplan FS, Shore EM.

Curr Osteoporos Rep. 2015 Apr;13(2):116-24. doi: 10.1007/s11914-015-0258-z. Review.


Progressive osseous heteroplasia: diagnosis, treatment, and prognosis.

Pignolo RJ, Ramaswamy G, Fong JT, Shore EM, Kaplan FS.

Appl Clin Genet. 2015 Jan 30;8:37-48. doi: 10.2147/TACG.S51064. eCollection 2015. Review.


Clinical Utility Gene Card for: Fibrodysplasia ossificans progressiva.

Bravenboer N, Micha D, Triffit JT, Bullock AN, Ravazollo R, Bocciardi R, di Rocco M, Netelenbos JC, Ten Dijke P, Sánchez-Duffhues G, Kaplan FS, Shore EM, Pignolo RJ, Seemann P, Ventura F, Beaujat G, Eekhoff EM, Pals G.

Eur J Hum Genet. 2015 Oct;23(10). doi: 10.1038/ejhg.2014.274. Epub 2015 Jan 21. No abstract available.


ACVR1 p.Q207E causes classic fibrodysplasia ossificans progressiva and is functionally distinct from the engineered constitutively active ACVR1 p.Q207D variant.

Haupt J, Deichsel A, Stange K, Ast C, Bocciardi R, Ravazzolo R, Di Rocco M, Ferrari P, Landi A, Kaplan FS, Shore EM, Reissner C, Seemann P.

Hum Mol Genet. 2014 Oct 15;23(20):5364-77. doi: 10.1093/hmg/ddu255. Epub 2014 May 22.


From mysteries to medicines: drug development for fibrodysplasia ossificans progressive.

Kaplan FS, Pignolo RJ, Shore EM.

Expert Opin Orphan Drugs. 2013 Aug;1(8):637-649.


Alk2 regulates early chondrogenic fate in fibrodysplasia ossificans progressiva heterotopic endochondral ossification.

Culbert AL, Chakkalakal SA, Theosmy EG, Brennan TA, Kaplan FS, Shore EM.

Stem Cells. 2014 May;32(5):1289-300. doi: 10.1002/stem.1633.


General anesthesia for dental procedures in patients with fibrodysplasia ossificans progressiva: a review of 42 cases in 30 patients.

Kilmartin E, Grunwald Z, Kaplan FS, Nussbaum BL.

Anesth Analg. 2014 Feb;118(2):298-301. doi: 10.1213/ANE.0000000000000021.


Activation of Hedgehog signaling by loss of GNAS causes heterotopic ossification.

Regard JB, Malhotra D, Gvozdenovic-Jeremic J, Josey M, Chen M, Weinstein LS, Lu J, Shore EM, Kaplan FS, Yang Y.

Nat Med. 2013 Nov;19(11):1505-12. doi: 10.1038/nm.3314. Epub 2013 Sep 29.


The phenotype and genotype of fibrodysplasia ossificans progressiva in China: a report of 72 cases.

Zhang W, Zhang K, Song L, Pang J, Ma H, Shore EM, Kaplan FS, Wang P.

Bone. 2013 Dec;57(2):386-91. doi: 10.1016/j.bone.2013.09.002. Epub 2013 Sep 17.


Somitic disruption of GNAS in chick embryos mimics progressive osseous heteroplasia.

Cairns DM, Pignolo RJ, Uchimura T, Brennan TA, Lindborg CM, Xu M, Kaplan FS, Shore EM, Zeng L.

J Clin Invest. 2013 Aug;123(8):3624-33. doi: 10.1172/JCI69746. Epub 2013 Jul 25. Erratum in: J Clin Invest. 2013 Nov;123(11):4981.


Fibrodysplasia ossificans progressiva: diagnosis, management, and therapeutic horizons.

Pignolo RJ, Shore EM, Kaplan FS.

Pediatr Endocrinol Rev. 2013 Jun;10 Suppl 2:437-48. Review.


The skeleton in the closet.

Kaplan FS.

Gene. 2013 Oct 1;528(1):7-11. doi: 10.1016/j.gene.2013.06.022. Epub 2013 Jun 25.


Severe soft tissue ossification in a southern right whale Eubalaena australis.

La Sala LF, Pozzi LM, McAloose D, Kaplan FS, Shore EM, Kompanje EJ, Sidor IF, Musmeci L, Uhart MM.

Dis Aquat Organ. 2012 Dec 27;102(2):149-56. doi: 10.3354/dao02538.


Fibrodysplasia ossificans progressiva: mechanisms and models of skeletal metamorphosis.

Kaplan FS, Chakkalakal SA, Shore EM.

Dis Model Mech. 2012 Nov;5(6):756-62. doi: 10.1242/dmm.010280. Review.

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