Case and research letter
Primary Anetoderma Associated With Primary Sjögren Syndrome and Anticardiolipin Antibodies
Anetodermia primaria asociada a síndrome de Sjögren primario y anticuerpos anticardiolipina
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Yélamos, M.A. Barnadas, C. Díaz, L. Puig" "autores" => array:4 [ 0 => array:4 [ "nombre" => "O." "apellidos" => "Yélamos" "email" => array:1 [ 0 => "oyelamos@santpau.cat" ] "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" => "M.A." "apellidos" => "Barnadas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "C." "apellidos" => "Díaz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "L." "apellidos" => "Puig" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Reumatología, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Anetodermia primaria asociada a síndrome de Sjögren primario y anticuerpos anticardiolipina" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 710 "Ancho" => 951 "Tamanyo" => 194454 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Acute inflammatory interstitial infiltrate with abundant neutrophils in the reticular dermis (hematoxylin-eosin, original magnification ×200).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Anetoderma is a rare acquired chronic skin condition that involves the loss of elastic fibers. There is no specific effective treatment.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> We present the case of a patient with primary anetoderma associated with both primary Sjögren syndrome and anticardiolipin antibodies. Biopsy showed an acute inflammatory infiltrate observed in the reticular dermis. The condition was treated with colchicine and dapsone.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 21-year-old woman with a history of depression and bulimia, for which she was receiving effective duloxetine treatment; she had no recent changes in weight. She was first referred to our hospital's dermatology department in June 2009 for painful 5-mm erythematous papules that had appeared gradually and were progressing to soft, normal-colored patches on the upper arms and upper third of the back; they measured approximately 1<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleSup">2</span> in size (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). She reported no past history of miscarriage, infertility, or vascular thrombosis.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The patient had been referred by the rheumatology department, where she had consulted for dry mouth and dry eyes. Her Schirmer test was positive (3<span class="elsevierStyleHsp" style=""></span>mm in 5 minutes for the right eye and 5 mm in 5 minutes for the left). Sialometry showed low salivary secretion (0.3<span class="elsevierStyleHsp" style=""></span>mL unstimulated and 6.1<span class="elsevierStyleHsp" style=""></span>mL stimulated). Salient analytical findings included the following: anti-nuclear antibody titer, 1/320, with a speckled pattern; anti-Ro/SS-A and anti-La/SS-B antibodies; rheumatoid factor, 56<span class="elsevierStyleHsp" style=""></span>IU/mL (reference range, <<span class="elsevierStyleHsp" style=""></span>20<span class="elsevierStyleHsp" style=""></span>IU/mL); complement factor C4, 86.6<span class="elsevierStyleHsp" style=""></span>μg/mL (reference range, 120–360<span class="elsevierStyleHsp" style=""></span>μg/mL); and immunoglobulin M anticardiolipin antibodies, 6.73<span class="elsevierStyleHsp" style=""></span>MPL/mL (reference range, <<span class="elsevierStyleHsp" style=""></span>4.6<span class="elsevierStyleHsp" style=""></span>MPL/mL). Serologic tests for human immunodeficiency virus, anti-beta-2-glycoprotein <span class="elsevierStyleSmallCaps">i</span> antibodies, and lupus anticoagulant were all negative. These findings had led the rheumatology department to a diagnosis of primary Sjögren syndrome; antiphospholipid syndrome and lupus erythematosus were ruled out.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Biopsy of the skin lesions revealed a perivascular and interstitial inflammatory infiltrate consisting of lymphocytes and histiocytes, with abundant neutrophils and eosinophils, in the reticular dermis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Staining for elastic fibers showed fragmentation and loss of elastic fibers in the papillary and reticular dermis (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). These findings led to a diagnosis of primary anetoderma in a patient with primary Sjögren syndrome.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Treatment was started with colchicine (1.5<span class="elsevierStyleHsp" style=""></span>mg/d) and the lesions stabilized; however, treatment was suspended after 2 months due to gastrointestinal intolerance. New, painful erythematous lesions appeared subsequently, and dapsone treatment (50<span class="elsevierStyleHsp" style=""></span>mg/d) was started. This was well tolerated and the dosage was increased to 100<span class="elsevierStyleHsp" style=""></span>mg/d; symptoms improved, and the patient's condition became stable. In December 2011, treatment was discontinued due to paresthesia of the hands. Electromyography findings were compatible with carpal tunnel syndrome, and dapsone-induced neuropathy was thus ruled out. Since no new lesions had developed, treatment was not resumed and the patient was still stable at the time of writing.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Anetoderma is a benign elastolytic disease that primarily affects middle-aged women. Its clinical presentation consists of well-demarcated skin-colored or erythematous papules or plaques, leaving whole areas of wrinkled skin with variable degrees of atrophy and soft herniated areas.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Histologically, anetoderma is characterized by focal loss of elastic fibers in the reticular dermis and, less frequently, in the papillary dermis. Other common findings include a lymphohistiocytic infiltrate, plasma cells, multinucleated giant cells, and exceptionally, eosinophils and neutrophils.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Primary anetoderma appears on clinically normal skin and may be associated with autoimmune disorders, unlike secondary anetoderma, which is linked to other skin conditions such as acne, chicken pox, cutaneous lymphoma, syphilis, and cutaneous lupus erythematosus.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> The autoimmune abnormality most frequently associated with primary anetoderma is the presence of antiphospholipid antibodies, with or without associated antiphospholipid syndrome.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Other associations, in descending order of frequency, include systemic lupus erythematosus, alopecia areata, vitiligo, hypothyroidism,<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> or analytical abnormalities, such as antinuclear or antithyroid antibodies, or hypocomplementemia.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> Primary anetoderma is in fact viewed by some as a marker of autoimmune disease that requires a comprehensive workup for immune disorders.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3-6</span></a> A recently published case also describes an association of mid-dermal elastolysis, another elastolytic condition, with the presence of autoantibodies, specifically antiphospholipid antibodies.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The association of primary anetoderma with primary Sjögren syndrome is exceptional, with only 2 cases described in the literature,<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,8</span></a> although there is 1 published case of anetoderma secondary to multiple plasmocytomas in a patient with Sjögren syndrome.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Anticardiolipin antibody positivity was not reported in any of these cases.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Several hypotheses are under consideration for the pathogenesis of primary anetoderma. One possibility is that it involves local ischemia caused by microthrombosis, vasculitis, or immune deposits (chiefly immunoglobulin M and complement factor C3) on vessel walls, giving rise to the degeneration of dermal elastic tissue.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a> Another hypothesis is that neutrophils in the infiltrate alter elastin by secreting elastases, although the role antiphospholipid antibodies might play is unknown.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> No effective treatment has been described for primary anetoderma, but reported findings suggest that neutrophil chemotactic inhibitors such as dapsone or colchicine might be of some use. Braun et al.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">11</span></a> described the stabilization of this condition with colchicine whereas Venencie et al.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> found no improvement using dapsone.</p><p id="par0055" class="elsevierStylePara elsevierViewall">We have presented the first case of primary anetoderma associated with both primary Sjögren syndrome and anticardiolipin antibodies. The presence of Sjögren syndrome together with these antibodies, even at low titers (levels ><span class="elsevierStyleHsp" style=""></span>40<span class="elsevierStyleHsp" style=""></span>MPL/mL are considered moderate to high) further supports the link between primary anetoderma and autoimmune disorders. We also highlight the presence of granulocytes in anetoderma in this case, not only because this unusual finding requires staining for elastic fibers to confirm the diagnosis, but also because patients like ours might benefit from drugs such as dapsone or colchicine.</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: Yélamos O, Barnadas MA, Díaz C, Puig L. Anetodermia primaria asociada a síndrome de Sjögren primario y anticuerpos anticardiolipina. Actas Dermosifiliogr. 2014;105:99–101.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 713 "Ancho" => 951 "Tamanyo" => 82141 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Skin-colored papules with a wrinkled surface on the upper third of the back, suggestive of anetoderma.</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" => 710 "Ancho" => 951 "Tamanyo" => 194454 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Acute inflammatory interstitial infiltrate with abundant neutrophils in the reticular dermis (hematoxylin-eosin, original magnification ×200).</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 713 "Ancho" => 951 "Tamanyo" => 274497 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Histopathology showing loss and fragmentation of elastic fibers in the papillary and reticular dermis (Verhoeff-van Gieson stain, original magnification ×100).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:11 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Anetoderma. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 9 | 8 | 17 |
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| 2021 Diciembre | 49 | 35 | 84 |
| 2021 Noviembre | 47 | 50 | 97 |
| 2021 Octubre | 63 | 43 | 106 |
| 2021 Septiembre | 48 | 43 | 91 |
| 2021 Agosto | 54 | 32 | 86 |
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| 2020 Octubre | 26 | 8 | 34 |
| 2020 Septiembre | 28 | 17 | 45 |
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| 2016 Octubre | 86 | 17 | 103 |
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| 2015 Octubre | 12 | 10 | 22 |
| 2015 Septiembre | 2 | 1 | 3 |
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| 2015 Mayo | 59 | 7 | 66 |
| 2015 Abril | 50 | 10 | 60 |
| 2015 Marzo | 49 | 6 | 55 |
| 2015 Febrero | 68 | 7 | 75 |
| 2015 Enero | 33 | 2 | 35 |
| 2014 Diciembre | 42 | 7 | 49 |
| 2014 Noviembre | 30 | 10 | 40 |
| 2014 Octubre | 54 | 8 | 62 |
| 2014 Septiembre | 28 | 5 | 33 |
| 2014 Agosto | 27 | 4 | 31 |
| 2014 Julio | 34 | 8 | 42 |
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| 2014 Mayo | 25 | 9 | 34 |
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| 2014 Marzo | 24 | 8 | 32 |
| 2014 Febrero | 14 | 3 | 17 |
| 2014 Enero | 1 | 0 | 1 |
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