Images in Clinical Rheumatology
Fibrodysplasia Ossificans Progressiva: Case Report of a Terminal Patient
Fibrodisplasia osificante progresiva: comunicación de un caso terminal
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"Fibrodysplasia Ossificans Progressiva: Case Report of a Terminal Patient" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "303" "paginaFinal" => "304" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "María del Mar Muñoz Gómez, Marta Novella Navarro, Fernando Ruiz Santiago, Enrique Raya Álvarez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "María del Mar" "apellidos" => "Muñoz Gómez" "email" => array:1 [ 0 => "mari936@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor1" ] ] ] 1 => array:3 [ "nombre" => "Marta" "apellidos" => "Novella Navarro" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Fernando" "apellidos" => "Ruiz Santiago" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Enrique" "apellidos" => "Raya Álvarez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Departamento de Reumatología, Hospital Universitario San Cecilio, Granada, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Sección de Radiología Musculoesquelética, Hospital Universitario Virgen de las Nieves, Granada, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor1" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Fibrodisplasia osificante progresiva: comunicación de un caso terminal" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1191 "Ancho" => 950 "Tamanyo" => 83441 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Radiograph of left knee showing ankylosis secondary to bone bridges.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The patient was a 29-year-old woman, with a history of type 1A diabetes and primary amenorrhea secondary to cervical agenesis. At the age of 10 years, she was examined because she had developed a tumor in the cervical region, with no previous injury; she underwent a radiological study and bone biopsy that was compatible with myositis ossificans. Over the years, large ectopic ossifications developed in her shoulders, hips, knees, ankles and paravertebral muscles, which produced a marked secondary thoracolumbar scoliosis and a considerably generalized functional limitation. A plain radiology study of the affected areas showed the existence of exuberant bone bridges at different stages of maturation, that provoked the locking and ankylosing of a number of joints, including a knee (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), the spine (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) and a shoulder (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The patient was treated with bisphosphonates and calcium supplements with vitamin D for 5 years, while she was also being followed by the rehabilitation service; however, none of these measures was capable of arresting the course of the disease, which led to the physical disability of the patient.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Fibrodysplasia ossificans progressiva is a rare disease of unknown origin that is characterized by a progressive heterotopic ossification of the muscles and other structures in which connective tissue is abundant.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> In recent years, there has been an advance in our knowledge of the etiological and pathogenic mechanisms of this disorder, which implicates the receptor of bone morphogenetic protein (BMP), the so-called activin A type I receptor/activin-like kinase 2 (AVCR1/ALK2).<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,3</span></a> Some authors have suggested that blocking the activity of the AVCR1/ALK2 receptor through the development of therapeutic agents that behave like signal transduction inhibitors that encode these proteins may be useful.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> In this respect, we know that dorsomorphin, a small molecule produced by the zebrafish, inhibits BMP type I receptors (including ALK2—implicated in the etiology and pathogenesis of the disease—and other activin-like kinases like ALK3 and ALK6)<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a>; however, given the lack of specificity of this molecule, further studies are needed in this line of research, as well as the development of <span class="elsevierStyleItalic">in vivo</span> models to determine the potential efficacy of this molecule in the prevention of the flares of ossification that occur in this serious disease.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical Disclosures</span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Protection of human and animal subjects</span><p id="par0015" class="elsevierStylePara elsevierViewall">The authors declare that no experiments were performed on humans or animals for this study.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Confidentiality of data</span><p id="par0020" class="elsevierStylePara elsevierViewall">The authors declare that they have followed the protocols of their work center on the publication of patient data.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Right to privacy and informed consent</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors have obtained the written informed consent of the patients or subjects mentioned in the article. The corresponding author is in possession of this document.</p></span></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Conflicts of Interest</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors declare they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:3 [ "identificador" => "sec0005" "titulo" => "Ethical Disclosures" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0010" "titulo" => "Protection of human and animal subjects" ] 1 => array:2 [ "identificador" => "sec0015" "titulo" => "Confidentiality of data" ] 2 => array:2 [ "identificador" => "sec0020" "titulo" => "Right to privacy and informed consent" ] ] ] 1 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflicts of Interest" ] 2 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2016-03-05" "fechaAceptado" => "2016-04-22" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Muñoz Gómez MM, Novella Navarro M, Ruiz Santiago F, Raya Álvarez E. Fibrodisplasia osificante progresiva: comunicación de un caso terminal. Reumatol Clin. 2017;13:303–304.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1191 "Ancho" => 950 "Tamanyo" => 83441 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Radiograph of left knee showing ankylosis secondary to bone bridges.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1193 "Ancho" => 950 "Tamanyo" => 162452 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Radiograph of abdomen showing paravertebral bone bridges that are also seen in right hip.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1041 "Ancho" => 950 "Tamanyo" => 133608 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Radiograph of shoulder showing a well-defined plaque of mature bone between the chest wall, humerus and scapula.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Genetic aspects of fibrodysplasia ossificans progressiva" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "J.M. 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Sanchez-Duffhues" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.reuma.2014.05.001" "Revista" => array:6 [ "tituloSerie" => "Reumatol Clin" "fecha" => "2014" "volumen" => "10" "paginaInicial" => "389" "paginaFinal" => "395" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/25107346" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Fibrodysplasia ossificans progressiva: diagnosis, management, and therapeutic horizons" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "R.J. Pignolo" 1 => "E.M. Shore" 2 => "F.S. Kaplan" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:7 [ "tituloSerie" => "Pediatr Endocrinol Rev" "fecha" => "2013" "volumen" => "10" "numero" => "Suppl. 2" "paginaInicial" => "437" "paginaFinal" => "448" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/23858627" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Dorsomorphin inhibits BMP signals required for embryogenesis and iron metabolism" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "P.B. Yu" 1 => "C.C. Hong" 2 => "C. Sachidanandan" 3 => "J.L. Babitt" 4 => "D.Y. Deng" 5 => "S.A. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 12 | 14 | 26 |
| 2024 October | 116 | 32 | 148 |
| 2024 September | 87 | 16 | 103 |
| 2024 August | 82 | 37 | 119 |
| 2024 July | 55 | 36 | 91 |
| 2024 June | 80 | 24 | 104 |
| 2024 May | 99 | 38 | 137 |
| 2024 April | 81 | 21 | 102 |
| 2024 March | 93 | 26 | 119 |
| 2024 February | 78 | 18 | 96 |
| 2024 January | 68 | 23 | 91 |
| 2023 December | 71 | 25 | 96 |
| 2023 November | 89 | 17 | 106 |
| 2023 October | 75 | 22 | 97 |
| 2023 September | 93 | 35 | 128 |
| 2023 August | 64 | 13 | 77 |
| 2023 July | 71 | 22 | 93 |
| 2023 June | 62 | 22 | 84 |
| 2023 May | 81 | 24 | 105 |
| 2023 April | 55 | 6 | 61 |
| 2023 March | 110 | 28 | 138 |
| 2023 February | 61 | 25 | 86 |
| 2023 January | 53 | 17 | 70 |
| 2022 December | 89 | 32 | 121 |
| 2022 November | 115 | 25 | 140 |
| 2022 October | 91 | 34 | 125 |
| 2022 September | 62 | 43 | 105 |
| 2022 August | 67 | 40 | 107 |
| 2022 July | 50 | 33 | 83 |
| 2022 June | 61 | 32 | 93 |
| 2022 May | 80 | 39 | 119 |
| 2022 April | 72 | 39 | 111 |
| 2022 March | 77 | 52 | 129 |
| 2022 February | 105 | 28 | 133 |
| 2022 January | 96 | 40 | 136 |
| 2021 December | 43 | 35 | 78 |
| 2021 November | 74 | 41 | 115 |
| 2021 October | 71 | 57 | 128 |
| 2021 September | 57 | 42 | 99 |
| 2021 August | 74 | 31 | 105 |
| 2021 July | 51 | 37 | 88 |
| 2021 June | 57 | 41 | 98 |
| 2021 May | 76 | 43 | 119 |
| 2021 April | 163 | 79 | 242 |
| 2021 March | 99 | 24 | 123 |
| 2021 February | 38 | 14 | 52 |
| 2021 January | 44 | 14 | 58 |
| 2020 December | 47 | 22 | 69 |
| 2020 November | 48 | 21 | 69 |
| 2020 October | 36 | 11 | 47 |
| 2020 September | 65 | 42 | 107 |
| 2020 August | 19 | 20 | 39 |
| 2020 July | 22 | 13 | 35 |
| 2020 June | 23 | 13 | 36 |
| 2020 May | 30 | 9 | 39 |
| 2020 April | 38 | 15 | 53 |
| 2020 March | 12 | 4 | 16 |
| 2020 February | 1 | 0 | 1 |
| 2018 December | 2 | 0 | 2 |
| 2018 May | 3 | 0 | 3 |
| 2018 April | 78 | 6 | 84 |
| 2018 March | 80 | 10 | 90 |
| 2018 February | 51 | 7 | 58 |
| 2018 January | 58 | 7 | 65 |
| 2017 December | 41 | 15 | 56 |
| 2017 November | 58 | 15 | 73 |
| 2017 October | 57 | 34 | 91 |
| 2017 September | 58 | 27 | 85 |
| 2017 August | 31 | 17 | 48 |
| 2017 July | 2 | 9 | 11 |
| 2017 June | 5 | 11 | 16 |
| 2017 May | 10 | 17 | 27 |
| 2017 April | 0 | 1 | 1 |
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