Letter to the Editor
Scleromyxedema: Presentation of 2 new cases and review of the literature
Escleromixedema, presentación de 2 nuevos casos y revisión de la literatura
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Fonseca Aizpuru, Carlos Delgado Verges, Esther González García, Luis Barcala Vázquez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Eva M." "apellidos" => "Fonseca Aizpuru" "email" => array:1 [ 0 => "evamfonseca@yahoo.es" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Carlos" "apellidos" => "Delgado Verges" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Esther" "apellidos" => "González García" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Luis" "apellidos" => "Barcala Vázquez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Servicio de Medicina Interna, Hospital Universitario de Cabueñes, Gijón, Asturias, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Hematología, Hospital Universitario de Cabueñes, Gijón, Asturias, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitario de Cabueñes, Gijón, Asturias, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Escleromixedema, presentación de 2 nuevos casos y revisión de la literatura" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Scleromyxedema (SM) is a rare, progressive, chronic disease. Diagnosis is based on 4 criteria: (1) a papular sclerodermoid eruption; (2) microscopic evidence of mucin deposition, fibroblast proliferation and fibrosis; (3) monoclonal gammopathy and (4) absence of thyroid involvement. It may present with severe systemic participation, dermato-neuro syndrome may be fatal. Prognosis and treatment are unclear. At present favourable response has been described with intravenous immunoglobulins (IVIG) and haematopoietic stem cell transplant (HSCT).<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1–3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Our aim was to present 2 new cases and to review the patient series published in the medical literature – Pubmed, with SM diagnosis. A systematic search was made with the following terms: scleromyxedema (MeSH) AND paraproteinemias (MeSH).</p><p id="par0015" class="elsevierStylePara elsevierViewall">Case 1: 37-year-old male, smoker for 10 years of one packet per day. He presented with oedema, erythema and induration in face, arms and hands of 6 months onset. Examination revealed: hard sclerodermiform skin with papular appearance, on the face, microstoma (with dysphagia and loss of 20<span class="elsevierStyleHsp" style=""></span>kg in weight), and involved the pinna, chest, abdomen and arms with sclerodactyly, with a 31 classification score on the Rodnan scale. Analysis was performed with IgG lambda gammopathy. Treatment was prescribed with prednisone and hydroxychloroquine, with no improvement. Skin biopsy confirmed SM.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Case 2: 69-year-old male, with high blood pressure and dyslipidemia. The condition began with a papular eruption in the first finger of his right hand, left hand and face, and treatment was initiated with phototherapy, prednisone and hydroxychloroquine, with no improvement. There was a progression of the skin induration in his face, with microstoma (associated with dysphagia and loss of 7<span class="elsevierStyleHsp" style=""></span>kg in weight), arms and hands with sclerodactyly, chest, abdomen and lower limbs, and classification of 26 on the Rodnan scale. Analysis was performed with IgG lambda gammopathy. A study of systemic involvement was conducted with diagnosis of sensory polyneuropathy in lower limbs and oesophageal hypomotility. Skin biopsy confirmed SM. In both cases treatment was initiated with IVIg, with partial response and he was referred to the haematological department for autologous stem cell transplant (ASCT), with improvement in skin symptoms, reduction of induration on the Rodnan scale and systemic improvement.</p><p id="par0025" class="elsevierStylePara elsevierViewall">SM is a rare fibromucinosis-type skin disorder, described by Montgomery and Underwood in 1953. Classification was reviewed in 2001 by Rongioletti and Rebora. For its diagnosis, the patient has to have met with 4 criteria.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1–3</span></a> It presents within an age range of 28–77 years, with no gender differences. As the disease advances, skin infiltration with sclerosis becomes generalised, leading to significant disability. The extra-cutaneous symptoms include: gastrointestinal dysmotility and peripheral neuropathy. Dermato-neuro syndrome describes the involvement of fever, convulsions and coma, which is an uncommon manifestation, and has a mortality rate of 32%<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> SM is associated with a monoclonal protein and particularly with type IgG lambda. The relationship between skin findings and the paraprotein is unknown.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2–4</span></a> Differential diagnosis has to be made with sclerodermia, and nephrogenic systemic fibrosis, among other diseases.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Treatment of SM is unspecified.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Up until now, a study by Blum et al. is the most extensive series of patients treated with IVIg and with favourable long-term follow-up results. However, response to immunoglobulins was transitory and required maintenance therapy.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> ASCT may be efficacious and efficient.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,3,6,7</span></a> IN 2001 Feasel et al. described the first case of remission of SM after an ASCT.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> In a later study in 2016, Chockalingam and Duvic concluded that ASCT appears to be a safe and effective long-term treatment.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">SM is a chronic disease with a reserved prognosis. There is no specific definitive treatment.<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">8–10</span></a> Randomised, multicentre studies are needed, and larger patient groups.</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: Fonseca Aizpuru EM, Delgado Verges C, González García E, Barcala Vázquez L. Escleromixedema, presentación de 2 nuevos casos y revisión de la literatura. Reumatol Clin. 2020;16:430.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Scleromyxedema: a case series highlighting long-term outcomes of treatment with intravenous immunoglobulin (IVIG)" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "M. Blum" 1 => "F.M. Wigley" 2 => "L.K. 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Bessis" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1016/j.annder.2010.08.011" "Revista" => array:6 [ "tituloSerie" => "Ann Dermatol Venereol" "fecha" => "2010" "volumen" => "137" "paginaInicial" => "782" "paginaFinal" => "788" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/21134580" "web" => "Medline" ] ] ] ] ] ] ] ] 9 => array:3 [ "identificador" => "bib0100" "etiqueta" => "10" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Scleromyxedema: a multicenter study of characteristics, comorbidities, course, and therapy in 30 patients" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "F. Rongioletti" 1 => "G. Merlo" 2 => "E. Cinotti" 3 => "V. Fausti" 4 => "E. Cozzani" 5 => "B. Cribier" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Am Acad Dermatol" "fecha" => "2013" "volumen" => "69" "paginaInicial" => "66" "paginaFinal" => "72" ] ] ] ] ] ] ] ] ] ] "agradecimientos" => array:1 [ 0 => array:4 [ "identificador" => "xack484037" "titulo" => "Acknowledgements" "texto" => "<p id="par0035" class="elsevierStylePara elsevierViewall">Our thanks to Daniel González Fernández, Ana M. Pilar González Rodríguez and Pablo Gonzalvo Rodríguez.</p>" "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/21735743/00000016000005P2/v1_202009270752/S2173574319301224/v1_202009270752/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/21735743/00000016000005P2/v1_202009270752/S2173574319301224/v1_202009270752/en/main.pdf?idApp=UINPBA00004M&text.app=https://reumatologiaclinica.org/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173574319301224?idApp=UINPBA00004M" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 3 | 3 | 6 |
| 2024 October | 48 | 29 | 77 |
| 2024 September | 84 | 18 | 102 |
| 2024 August | 92 | 44 | 136 |
| 2024 July | 85 | 37 | 122 |
| 2024 June | 78 | 38 | 116 |
| 2024 May | 75 | 25 | 100 |
| 2024 April | 63 | 25 | 88 |
| 2024 March | 55 | 23 | 78 |
| 2024 February | 55 | 31 | 86 |
| 2024 January | 45 | 20 | 65 |
| 2023 December | 79 | 33 | 112 |
| 2023 November | 65 | 58 | 123 |
| 2023 October | 55 | 25 | 80 |
| 2023 September | 109 | 44 | 153 |
| 2023 August | 74 | 15 | 89 |
| 2023 July | 63 | 27 | 90 |
| 2023 June | 74 | 24 | 98 |
| 2023 May | 55 | 27 | 82 |
| 2023 April | 48 | 19 | 67 |
| 2023 March | 76 | 28 | 104 |
| 2023 February | 58 | 20 | 78 |
| 2023 January | 50 | 11 | 61 |
| 2022 December | 81 | 34 | 115 |
| 2022 November | 71 | 40 | 111 |
| 2022 October | 78 | 37 | 115 |
| 2022 September | 55 | 28 | 83 |
| 2022 August | 44 | 29 | 73 |
| 2022 July | 46 | 38 | 84 |
| 2022 June | 97 | 35 | 132 |
| 2022 May | 66 | 41 | 107 |
| 2022 April | 78 | 45 | 123 |
| 2022 March | 52 | 59 | 111 |
| 2022 February | 65 | 39 | 104 |
| 2022 January | 66 | 34 | 100 |
| 2021 December | 44 | 45 | 89 |
| 2021 November | 29 | 39 | 68 |
| 2021 October | 34 | 42 | 76 |
| 2021 September | 23 | 40 | 63 |
| 2021 August | 11 | 22 | 33 |
| 2021 July | 17 | 26 | 43 |
| 2021 June | 15 | 25 | 40 |
| 2021 May | 98 | 51 | 149 |
| 2021 April | 55 | 59 | 114 |
| 2021 March | 26 | 21 | 47 |
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