Case and Research Letter
Nodular Mucinosis Associated With Light-Chain Monoclonal Gammopathy of Uncertain Significance
Mucinosis nodular asociada a gammapatía monoclonal de cadenas ligeras de significado incierto
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Rodríguez-Jiménez, P. Chicharro, A. Ascensión, D. de Argila, E. Daudén" "autores" => array:5 [ 0 => array:4 [ "nombre" => "P." "apellidos" => "Rodríguez-Jiménez" "email" => array:1 [ 0 => "pedro.rodriguez.jimenez90@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "P." "apellidos" => "Chicharro" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "A." "apellidos" => "Ascensión" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "D." "apellidos" => "de Argila" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 4 => array:3 [ "nombre" => "E." "apellidos" => "Daudén" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicios de Dermatología, Hospital Universitario de La Princesa, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicios de Anatomía Patológica, Hospital Universitario de La Princesa, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Mucinosis nodular asociada a gammapatía monoclonal de cadenas ligeras de significado incierto" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 619 "Ancho" => 1550 "Tamanyo" => 147830 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Brownish erythematous plaques on both elbows.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Nodular mucinosis is a chronic primary idiopathic mucinosis that, according to the 2001 classification proposed by Rongioletti et al.,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> belongs to the group of the localized forms of lichen myxedematosus, which includes acral persistent papular mucinosis, a mild form of papular mucinosis, self-healing papular mucinosis, papular mucinosis of infancy, and the nodular form. Localized mucinoses are characterized by the appearance of small numbers of waxy papules (or plaques or nodules due to confluence), usually on the lower limbs or trunk. These localized forms must be differentiated from the diffuse form or scleromyxedema, which is characterized by a rash of groups of small, firm waxy papules of around 2 to 3<span class="elsevierStyleHsp" style=""></span>mm in diameter, arising predominantly on the upper part of the trunk and on the neck, face, forearms, and hands.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a> The papules show a linear distribution and the perilesional skin is shiny. Muscle, joint, nervous system, gastrointestinal, pulmonary, or otohinolaryngologic manifestations may be present, and paraproteinemia is an almost constant feature. Atypical and intermediate forms also exist; these include patients with scleromyxedema but with no systemic signs or paraproteinemia, patients with localized forms associated with paraproteinemia, and combined or not otherwise specified cases.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We present the case of a 72-year-old woman with no past history of interest. She was seen in our department for the appearance 3 months earlier of 2 brownish erythematous plaques with a slightly scaly surface on both elbows (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The lesions had grown both radially and in thickness and were slightly tender. Biopsy revealed mild orthokeratotic hyperkeratosis with papillomatosis and, in the papillary dermis, abundant interstitial mucin deposits that stained positive with alcian blue. The diagnosis was localized mucinosis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Laboratory tests including complete blood count, biochemistry, and 24-hour urine analysis were normal. Plasma protein electrophoresis showed a peak of 890<span class="elsevierStyleHsp" style=""></span>mg/dL in the gamma region that was shown by immunofixation to be an oligoclonal band in a polyclonal background. Serum free light chains were measured and a kappa chain of 30<span class="elsevierStyleHsp" style=""></span>mg/L was detected, with a kappa-to-lambda ratio of 1.88. The hematology department did not consider it necessary to perform bone marrow study. With these results, we made a diagnosis of light chain monoclonal gammopathy of uncertain significance based on the Myeloma Working Group 2014 criteria.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> Treatment was started with topical clobetasol propionate under occlusive dressings for 1 month, with almost complete resolution of the lesions. At the time of writing, the patient continues on follow-up in the hematology department.</p><p id="par0020" class="elsevierStylePara elsevierViewall">This was therefore an atypical form of lichen myxedematosus; only 4 such cases have been published to date.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">4–7</span></a> The pathogenesis of the association is unknown. In 1 of the cases, the skin lesions disappeared 8 years after their onset and the paraproteinemia some years later, with no systemic clinical repercussions.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> Another case occurred in a 60-year-old may with skin lesions clinically and histologically compatible with papular mucinosis. After 3 months of treatment with oral corticosteroids with no response, the patient presented disorientation and a deterioration in his general state that led to a diagnosis of immunoglobulin M type multiple myeloma. He then received standard therapy with bortezomib and dexamethasone and the skin lesions disappeared after 4 treatment cycles.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">6</span></a> Finally, the interesting case of a 38-year-old woman with lesions of papular mucinosis that presented a Köebner phenomenon—a feature not previously described in the literature—and a monoclonal gammopathy of uncertain significance.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">It should be noted that, as previously mentioned by other authors,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> the terms lichen myxedematosus, scleromyxedema, and papular mucinosis have tended to be used interchangeably in the literature and in daily clinical practice and, despite the 2001 reclassification,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> confusion between the terms persists, particularly regarding the atypical forms such as ours.</p><p id="par0030" class="elsevierStylePara elsevierViewall">In our patient, thanks to the cutaneous manifestations, we will continue to monitor her closely and, if there is progression to light chain multiple myeloma (estimated incidence of 0.3% per year<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a>) or to primary amyloidosis, it should be possible to make an early diagnosis and initiate treatment, which is fundamental to the prognosis of this disease.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of Interest" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Rodríguez-Jiménez P, Chicharro P, Ascensión A, de Argila D, Daudén E. Mucinosis nodular asociada a gammapatía monoclonal de cadenas ligeras de significado incierto. Actas Dermosifiliogr. 2017;108:272–273.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 619 "Ancho" => 1550 "Tamanyo" => 147830 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Brownish erythematous plaques on both elbows.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1638 "Ancho" => 1625 "Tamanyo" => 843710 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Orthokeratotic hyperkeratosis with papillomatosis. Hematoxylin and eosin, original magnification<span class="elsevierStyleHsp" style=""></span>x40. B, Mucin deposits that stain positively with alcian blue. Alcian blue, original magnification<span class="elsevierStyleHsp" style=""></span>x100.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0040" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Updated classification of papular mucinosis, lichen myxedematosus, and scleromyxedema" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "F. Rongioletti" 1 => "A. Rebora" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1067/mjd.2001.111630" "Revista" => array:6 [ "tituloSerie" => "J Am Acad Dermatol." 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 9 | 10 | 19 |
| 2024 Octubre | 88 | 47 | 135 |
| 2024 Septiembre | 87 | 28 | 115 |
| 2024 Agosto | 120 | 75 | 195 |
| 2024 Julio | 103 | 36 | 139 |
| 2024 Junio | 79 | 25 | 104 |
| 2024 Mayo | 81 | 39 | 120 |
| 2024 Abril | 92 | 32 | 124 |
| 2024 Marzo | 89 | 23 | 112 |
| 2024 Febrero | 78 | 36 | 114 |
| 2024 Enero | 47 | 31 | 78 |
| 2023 Diciembre | 63 | 10 | 73 |
| 2023 Noviembre | 78 | 39 | 117 |
| 2023 Octubre | 75 | 24 | 99 |
| 2023 Septiembre | 79 | 29 | 108 |
| 2023 Agosto | 55 | 18 | 73 |
| 2023 Julio | 76 | 31 | 107 |
| 2023 Junio | 88 | 19 | 107 |
| 2023 Mayo | 56 | 31 | 87 |
| 2023 Abril | 76 | 31 | 107 |
| 2023 Marzo | 65 | 28 | 93 |
| 2023 Febrero | 55 | 29 | 84 |
| 2023 Enero | 36 | 22 | 58 |
| 2022 Diciembre | 57 | 37 | 94 |
| 2022 Noviembre | 39 | 29 | 68 |
| 2022 Octubre | 22 | 24 | 46 |
| 2022 Septiembre | 35 | 26 | 61 |
| 2022 Agosto | 34 | 27 | 61 |
| 2022 Julio | 23 | 27 | 50 |
| 2022 Junio | 29 | 23 | 52 |
| 2022 Mayo | 53 | 50 | 103 |
| 2022 Abril | 47 | 34 | 81 |
| 2022 Marzo | 43 | 54 | 97 |
| 2022 Febrero | 41 | 30 | 71 |
| 2022 Enero | 45 | 44 | 89 |
| 2021 Diciembre | 41 | 38 | 79 |
| 2021 Noviembre | 38 | 56 | 94 |
| 2021 Octubre | 48 | 55 | 103 |
| 2021 Septiembre | 33 | 39 | 72 |
| 2021 Agosto | 33 | 42 | 75 |
| 2021 Julio | 28 | 37 | 65 |
| 2021 Junio | 33 | 27 | 60 |
| 2021 Mayo | 45 | 46 | 91 |
| 2021 Abril | 64 | 54 | 118 |
| 2021 Marzo | 71 | 35 | 106 |
| 2021 Febrero | 57 | 27 | 84 |
| 2021 Enero | 38 | 24 | 62 |
| 2020 Diciembre | 38 | 22 | 60 |
| 2020 Noviembre | 31 | 22 | 53 |
| 2020 Octubre | 26 | 14 | 40 |
| 2020 Septiembre | 42 | 19 | 61 |
| 2020 Agosto | 32 | 23 | 55 |
| 2020 Julio | 25 | 19 | 44 |
| 2020 Junio | 21 | 28 | 49 |
| 2020 Mayo | 15 | 12 | 27 |
| 2020 Abril | 14 | 10 | 24 |
| 2020 Marzo | 17 | 14 | 31 |
| 2020 Febrero | 6 | 0 | 6 |
| 2020 Enero | 4 | 0 | 4 |
| 2019 Diciembre | 9 | 0 | 9 |
| 2019 Noviembre | 4 | 0 | 4 |
| 2019 Septiembre | 5 | 0 | 5 |
| 2019 Agosto | 4 | 0 | 4 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Junio | 4 | 0 | 4 |
| 2019 Mayo | 6 | 2 | 8 |
| 2019 Abril | 2 | 4 | 6 |
| 2019 Marzo | 3 | 0 | 3 |
| 2019 Febrero | 2 | 0 | 2 |
| 2018 Diciembre | 5 | 0 | 5 |
| 2018 Noviembre | 1 | 0 | 1 |
| 2018 Septiembre | 4 | 0 | 4 |
| 2018 Febrero | 27 | 2 | 29 |
| 2018 Enero | 27 | 5 | 32 |
| 2017 Diciembre | 71 | 6 | 77 |
| 2017 Noviembre | 21 | 4 | 25 |
| 2017 Octubre | 17 | 3 | 20 |
| 2017 Septiembre | 18 | 7 | 25 |
| 2017 Agosto | 23 | 6 | 29 |
| 2017 Julio | 25 | 6 | 31 |
| 2017 Junio | 34 | 10 | 44 |
| 2017 Mayo | 32 | 15 | 47 |
| 2017 Abril | 161 | 33 | 194 |
| 2017 Marzo | 23 | 25 | 48 |
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