Letter to the Editor
Acquired Madelung's Deformity in Rheumatoid Factor-positive Polyarticular Juvenile Idiopathic Arthritis
Deformidad de Madelung adquirida en la artritis idiopática juvenil poliarticular con factor reumatoide positivo
Walter Alberto Sifuentes Giraldo
, María Luz Gámir Gámir
Corresponding author
Unidad de Reumatología Pediátrica, Servicio de Reumatología, Hospital Universitario Ramón y Cajal, Madrid, Spain
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He had no other relevant family or personal history. He was treated with nonsteroidal antiinflammatory drugs, systemic and intra-articular corticosteroids, gold salts and methotrexate but, despite this, the disease remained active with persistent synovitis of both wrists, developing bilateral subluxation at that level and limitation of dorsiflexion and supination. Radiographs of both wrists showed shortening of the medial distal radius, exaggeration of the radial tilt and proximal migration of the bones of the first row of the carpus, adopting a V shape between the radius and ulna, consistent with Madelung's deformity (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The patient also developed “trigger fingers” in both hands and bilateral hallux valgus. At age 24, he began treatment with etanercept, achieving control of disease activity. Currently no synovitis of the wrists is present, but the limited movement persists, though it does not prevent him from carrying out his usual activities.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Madelung's deformity is a rare congenital disorder characterized by asymmetric carpal growth and curvature of the distal radial physis, resulting in a decrease in grip force of the hand. The common mechanism in Madelung's deformity is a premature closure of the epiphyseal growth cartilage in the medial and anterior portion of the distal palmar radius. This results in a shortening of the radius and, therefore, an apparent ulnar overgrowth. It is frequently associated with genetic syndromes such as Leri-Weill dyschondrosteosis and Turner's syndrome.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Alterations similar to those produced in congenital Madelung's deformity have been described in other disorders, including trauma, tumors, infections, endocrine disorders and generalized skeletal dysplasias.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3–5</span></a> The deformity may also occur in patients with the acquired form related to JIA because early skeletal maturation induced by joint inflammation can cause early closure of the carpal physis.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,6</span></a> There have been 2 cases of severe Madelung's deformity in patients with RF negative polyarticular JIA who also had Turner's syndrome,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> but in our case there was no evidence of another associated genetic syndrome.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Among the clinical and radiological characteristics of Madelung's deformity, dorsal and medial curvature of the distal radius, an increase in the inclination of the joint surface of the distal radius, the triangulation of the carpus with proximal migration and volar migration of the semilunar bone, as well as the prominent ulnar head, are common.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> This deformity is typically present bilaterally and manifested before 20 years of age. Patients have a limited pronation and supination of the affected carpus.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a> Severe cases develop progressive osteoarthritis and instability of the distal radioulnar joint, radiocarpal osteoarthritis and may complicate with rupture of the extensor tendons of the fingers.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">There are no specific guidelines for the treatment of this deformity and it is initially conservative, postponing surgical correction until skeletal maturity is achieved. Several techniques have been described for the correction of Madelung's deformity, but no clear evidence exists that supports the specific use of any of them. Common indications for surgery are pain, limitation in mobility and esthetic concerns.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In conclusion, acquired Madelung's deformity may occur as a complication of JIA, worsening the functional capacity even more in these patients, making it important to consider it in the patient's clinical and radiological evaluation.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Sifuentes Giraldo WA, Gámir Gámir ML. Deformidad de Madelung adquirida en la artritis idiopática juvenil poliarticular con factor reumatoide positivo. Reumatol Clin. 2014;10:266–267.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 863 "Ancho" => 1301 "Tamanyo" => 208369 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Anteroposterior X-rays of the left (A) and right carpus (B) showing bilateral Madelung's deformity.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Juvenile idiopathic arthritis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "B. Prakken" 1 => "S. 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Schneider" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s002470050674" "Revista" => array:6 [ "tituloSerie" => "Pediatr Radiol" "fecha" => "1999" "volumen" => "29" "paginaInicial" => "676" "paginaFinal" => "681" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/10460329" "web" => "Medline" ] ] ] ] ] ] ] ] 7 => array:3 [ "identificador" => "bib0040" "etiqueta" => "8" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Madelung's deformity: a spectrum of presentation" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "L.P. Zebala" 1 => "P.R. Manske" 2 => "C.A. Goldfarb" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.jhsa.2007.08.012" "Revista" => array:6 [ "tituloSerie" => "J Hand Surg Am" "fecha" => "2007" "volumen" => "32" "paginaInicial" => "1393" "paginaFinal" => "1401" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/17996774" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/21735743/0000001000000004/v1_201406280041/S2173574314000616/v1_201406280041/en/main.assets" "Apartado" => array:4 [ "identificador" => "8400" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/21735743/0000001000000004/v1_201406280041/S2173574314000616/v1_201406280041/en/main.pdf?idApp=UINPBA00004M&text.app=https://reumatologiaclinica.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173574314000616?idApp=UINPBA00004M" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 6 | 8 | 14 |
| 2024 October | 101 | 34 | 135 |
| 2024 September | 107 | 22 | 129 |
| 2024 August | 137 | 32 | 169 |
| 2024 July | 99 | 34 | 133 |
| 2024 June | 84 | 27 | 111 |
| 2024 May | 86 | 32 | 118 |
| 2024 April | 81 | 20 | 101 |
| 2024 March | 99 | 29 | 128 |
| 2024 February | 62 | 25 | 87 |
| 2024 January | 77 | 32 | 109 |
| 2023 December | 66 | 28 | 94 |
| 2023 November | 92 | 35 | 127 |
| 2023 October | 106 | 34 | 140 |
| 2023 September | 97 | 42 | 139 |
| 2023 August | 93 | 14 | 107 |
| 2023 July | 87 | 33 | 120 |
| 2023 June | 93 | 31 | 124 |
| 2023 May | 105 | 20 | 125 |
| 2023 April | 51 | 15 | 66 |
| 2023 March | 84 | 31 | 115 |
| 2023 February | 72 | 26 | 98 |
| 2023 January | 41 | 23 | 64 |
| 2022 December | 89 | 42 | 131 |
| 2022 November | 50 | 20 | 70 |
| 2022 October | 63 | 27 | 90 |
| 2022 September | 91 | 35 | 126 |
| 2022 August | 75 | 42 | 117 |
| 2022 July | 67 | 42 | 109 |
| 2022 June | 67 | 38 | 105 |
| 2022 May | 69 | 39 | 108 |
| 2022 April | 93 | 54 | 147 |
| 2022 March | 64 | 43 | 107 |
| 2022 February | 60 | 43 | 103 |
| 2022 January | 87 | 71 | 158 |
| 2021 December | 48 | 45 | 93 |
| 2021 November | 71 | 55 | 126 |
| 2021 October | 78 | 62 | 140 |
| 2021 September | 51 | 43 | 94 |
| 2021 August | 74 | 38 | 112 |
| 2021 July | 38 | 31 | 69 |
| 2021 June | 64 | 35 | 99 |
| 2021 May | 65 | 29 | 94 |
| 2021 April | 162 | 93 | 255 |
| 2021 March | 97 | 23 | 120 |
| 2021 February | 85 | 25 | 110 |
| 2021 January | 97 | 18 | 115 |
| 2020 December | 60 | 19 | 79 |
| 2020 November | 69 | 16 | 85 |
| 2020 October | 50 | 13 | 63 |
| 2020 September | 57 | 20 | 77 |
| 2020 August | 36 | 13 | 49 |
| 2020 July | 27 | 9 | 36 |
| 2020 June | 57 | 14 | 71 |
| 2020 May | 43 | 15 | 58 |
| 2020 April | 46 | 18 | 64 |
| 2020 March | 19 | 4 | 23 |
| 2020 February | 2 | 0 | 2 |
| 2018 May | 8 | 1 | 9 |
| 2018 April | 97 | 9 | 106 |
| 2018 March | 90 | 8 | 98 |
| 2018 February | 87 | 9 | 96 |
| 2018 January | 59 | 11 | 70 |
| 2017 December | 61 | 7 | 68 |
| 2017 November | 64 | 9 | 73 |
| 2017 October | 61 | 3 | 64 |
| 2017 September | 51 | 5 | 56 |
| 2017 August | 24 | 11 | 35 |
| 2017 July | 34 | 8 | 42 |
| 2017 June | 50 | 12 | 62 |
| 2017 May | 61 | 14 | 75 |
| 2017 April | 47 | 8 | 55 |
| 2017 March | 23 | 8 | 31 |
| 2017 February | 50 | 6 | 56 |
| 2017 January | 27 | 7 | 34 |
| 2016 December | 71 | 29 | 100 |
| 2016 November | 68 | 4 | 72 |
| 2016 October | 92 | 14 | 106 |
| 2016 September | 145 | 12 | 157 |
| 2016 August | 77 | 12 | 89 |
| 2016 July | 40 | 15 | 55 |
| 2015 December | 2 | 0 | 2 |
| 2015 September | 1 | 0 | 1 |
| 2015 August | 2 | 0 | 2 |
| 2015 July | 39 | 7 | 46 |
| 2015 June | 47 | 15 | 62 |
| 2015 May | 81 | 16 | 97 |
| 2015 April | 111 | 19 | 130 |
| 2015 March | 104 | 11 | 115 |
| 2015 February | 59 | 10 | 69 |
| 2015 January | 94 | 18 | 112 |
| 2014 December | 57 | 13 | 70 |
| 2014 November | 51 | 16 | 67 |
| 2014 October | 72 | 20 | 92 |
| 2014 September | 46 | 14 | 60 |
| 2014 August | 50 | 23 | 73 |
| 2014 July | 46 | 24 | 70 |
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