Case and Research Letters
Fox-Fordyce Disease With an Atypical Clinical Presentation
Enfermedad de Fox-Fordyce con presentación clínica atípica
I. Ballester
, Á. López-Ávila, S. Ortiz
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Servicio de Dermatología, Hospital Universitario Sta. María del Rosell de Cartagena, Murcia, Spain
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Ballester, Á. López-Ávila, S. Ortiz" "autores" => array:3 [ 0 => array:4 [ "nombre" => "I." "apellidos" => "Ballester" "email" => array:1 [ 0 => "ibalnor@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Á." "apellidos" => "López-Ávila" ] 2 => array:2 [ "nombre" => "S." "apellidos" => "Ortiz" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital Universitario Sta. María del Rosell de Cartagena, Murcia, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Enfermedad de Fox-Fordyce con presentación clínica atípica" ] ] "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" => 1991 "Ancho" => 1665 "Tamanyo" => 1014894 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Dilatation and hyperkeratotic plugging of the follicular infundibulum surrounded by an abundance of xanthomized macrophages (hematoxylin-eosin, original magnification ×50).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Fox-Fordyce disease (FFD), or apocrine miliaria, is a rare inflammatory disease of the apocrine glands characterized by the presence of pruritic follicular papules in apocrine-rich areas of the skin. We present a case of histologically confirmed FFD with extensive involvement of 2 unusual sites, the chest and abdomen.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 39-year-old woman was referred for evaluation of an asymptomatic rash that had appeared 2 years earlier. There was no significant personal or family medical history. The rash had first appeared in the presternal area and had then spread over the abdomen to the pubic region. The rash consisted of multiple minute nonconfluent red-to-yellow papules on the breasts and especially the lower chest and abdomen, as far as the pubic region (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The axillae, areolae, and genitals were uninvolved.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Histologic examination of a papule from the lower presternal region revealed dilatation and hyperkeratotic plugging of the follicular infundibulum, which was surrounded by abundant foamy histiocytes (xanthomized macrophages) (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>); a mild-to-moderate periadnexal chronic lymphocytic inflammatory response; and clusters of apocrine cells, with a tendency to cystic dilatation, between the reticular dermis and hypodermis (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">There were no relevant findings in the blood panel and the basic hormone panel, which included testosterone, dehydroepiandrosterone sulfate, progesterone, estradiol, luteinizing hormone, and follicle-stimulating hormone.</p><p id="par0025" class="elsevierStylePara elsevierViewall">No improvement was noted after successive attempts at topical treatment with tretinoin, corticosteroids, and clindamycin.</p><p id="par0030" class="elsevierStylePara elsevierViewall">FFD was first described by Fox and Fordyce in 1902.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> This rare skin condition is most common in women aged 13 to 35 years. The etiology and pathogenesis of FFD are unknown, although genetic, endocrine, environmental, and metabolic factors have been proposed.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Plugging of the apocrine duct at its point of entry in the follicular infundibulum appears to be the chief factor involved in the development of FFD. Plugging is thought to cause secretions to accumulate and then extravasate into the infundibulum and the peri-infundibular dermis, leading to a secondary inflammatory response that largely consists of macrophage recruitment.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Clinical presentation usually includes multiple white-to-yellow follicular papules symmetrically located in characteristic regions with an abundance of apocrine glands.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Lesions are frequently pruritic and worsen in the summer months and at times of emotional stress. Involved areas show little body hair growth and no sweating.</p><p id="par0040" class="elsevierStylePara elsevierViewall">FFD chiefly manifests in areas rich in apocrine glands: the axillae, mammary areolae, periumbilical region, mons pubis, labia minora, scrotum, foreskin, perianal region, free margins of the eyelids, and external ears. Apocrine glands are sparsely present in all other skin regions, especially the trunk and head,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> but FFD has only occasionally been reported in sites other than those listed. Unusual reported locations include the presternal and periumbilical regions and internal surface of the thighs.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,7</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The defining histopathologic features of FFD vary widely. Descriptions include dilatation and hyperkeratosis of the follicular infundibulum, spongiosis and dyskeratosis of the infundibular epithelium, vacuolar degeneration of the dermoepidermal junction, presence of periadnexal inflammatory lymphocytic infiltrate, parakeratosis of the infundibular epithelium in the form of cornoid lamellae, dilatation of the apocrine glands, and perifollicular xanthomatosis. The last is considered among the most characteristic findings.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,8,9</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Generally speaking, treatment of FFD is less than satisfactory, and the condition is typically chronic.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Treatment approaches used with varying results include topical and intralesional corticosteroids, topical or systemic retinoids, topical antibiotics (such as clindamycin), oral contraceptives, and topical pimecrolimus.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Highly refractory cases have sometimes led to surgical treatment, including electrocoagulation, excision, and liposuction.</p><p id="par0055" class="elsevierStylePara elsevierViewall">In conclusion, we believe that FFD should be considered in the differential diagnosis of any progressive rash of red-to-yellow papules on the chest or abdomen. Clinically similar, easily confused conditions more frequently described at these sites include syringomas, steatocystoma multiplex, or lichen nitidus, and distinguishing them from FFD requires the correlation of both clinical and histologic findings.</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: Ballester I, López-Ávila Á, Ortiz S. Enfermedad de Fox-Fordyce con presentación clínica atípica. Actas Dermosifiliogr. 2013;104:832–834.</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" => 1432 "Ancho" => 2175 "Tamanyo" => 351123 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A, Multiple minute nonconfluent red-to-yellow papules on the chest and abdomen. B, The axillae were not involved.</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" => 1991 "Ancho" => 1665 "Tamanyo" => 1014894 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Dilatation and hyperkeratotic plugging of the follicular infundibulum surrounded by an abundance of xanthomized macrophages (hematoxylin-eosin, original magnification ×50).</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" => 2082 "Ancho" => 1665 "Tamanyo" => 1001142 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Chronic periadnexal lymphocytic inflammatory infiltrate and dilated clusters of apocrine cells (hematoxylin-eosin, original magnification ×50).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Two cases of a rare papular disease affecting the axillary region" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "G.H. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 10 | 6 | 16 |
| 2024 Octubre | 84 | 47 | 131 |
| 2024 Septiembre | 94 | 33 | 127 |
| 2024 Agosto | 131 | 70 | 201 |
| 2024 Julio | 102 | 60 | 162 |
| 2024 Junio | 110 | 65 | 175 |
| 2024 Mayo | 86 | 39 | 125 |
| 2024 Abril | 66 | 34 | 100 |
| 2024 Marzo | 101 | 31 | 132 |
| 2024 Febrero | 84 | 35 | 119 |
| 2024 Enero | 86 | 31 | 117 |
| 2023 Diciembre | 83 | 18 | 101 |
| 2023 Noviembre | 81 | 39 | 120 |
| 2023 Octubre | 81 | 25 | 106 |
| 2023 Septiembre | 72 | 36 | 108 |
| 2023 Agosto | 44 | 45 | 89 |
| 2023 Julio | 92 | 60 | 152 |
| 2023 Junio | 54 | 48 | 102 |
| 2023 Mayo | 90 | 59 | 149 |
| 2023 Abril | 71 | 38 | 109 |
| 2023 Marzo | 83 | 54 | 137 |
| 2023 Febrero | 67 | 42 | 109 |
| 2023 Enero | 73 | 50 | 123 |
| 2022 Diciembre | 72 | 55 | 127 |
| 2022 Noviembre | 34 | 49 | 83 |
| 2022 Octubre | 38 | 36 | 74 |
| 2022 Septiembre | 37 | 49 | 86 |
| 2022 Agosto | 46 | 53 | 99 |
| 2022 Julio | 32 | 28 | 60 |
| 2022 Junio | 35 | 37 | 72 |
| 2022 Mayo | 72 | 45 | 117 |
| 2022 Abril | 67 | 28 | 95 |
| 2022 Marzo | 76 | 41 | 117 |
| 2022 Febrero | 44 | 20 | 64 |
| 2022 Enero | 54 | 43 | 97 |
| 2021 Diciembre | 53 | 39 | 92 |
| 2021 Noviembre | 74 | 57 | 131 |
| 2021 Octubre | 68 | 49 | 117 |
| 2021 Septiembre | 43 | 40 | 83 |
| 2021 Agosto | 97 | 29 | 126 |
| 2021 Julio | 69 | 42 | 111 |
| 2021 Junio | 64 | 41 | 105 |
| 2021 Mayo | 54 | 45 | 99 |
| 2021 Abril | 93 | 62 | 155 |
| 2021 Marzo | 87 | 30 | 117 |
| 2021 Febrero | 88 | 33 | 121 |
| 2021 Enero | 42 | 18 | 60 |
| 2020 Diciembre | 43 | 20 | 63 |
| 2020 Noviembre | 32 | 17 | 49 |
| 2020 Octubre | 34 | 13 | 47 |
| 2020 Septiembre | 29 | 18 | 47 |
| 2020 Agosto | 35 | 23 | 58 |
| 2020 Julio | 36 | 19 | 55 |
| 2020 Junio | 38 | 26 | 64 |
| 2020 Mayo | 36 | 23 | 59 |
| 2020 Abril | 27 | 17 | 44 |
| 2020 Marzo | 31 | 16 | 47 |
| 2020 Febrero | 4 | 3 | 7 |
| 2020 Enero | 0 | 1 | 1 |
| 2019 Diciembre | 0 | 8 | 8 |
| 2019 Octubre | 0 | 5 | 5 |
| 2019 Septiembre | 0 | 8 | 8 |
| 2019 Agosto | 0 | 6 | 6 |
| 2019 Julio | 1 | 25 | 26 |
| 2019 Junio | 10 | 42 | 52 |
| 2019 Mayo | 2 | 52 | 54 |
| 2019 Abril | 9 | 65 | 74 |
| 2019 Marzo | 1 | 33 | 34 |
| 2019 Febrero | 5 | 21 | 26 |
| 2019 Enero | 2 | 3 | 5 |
| 2018 Diciembre | 3 | 6 | 9 |
| 2018 Noviembre | 5 | 2 | 7 |
| 2018 Octubre | 15 | 2 | 17 |
| 2018 Septiembre | 4 | 4 | 8 |
| 2018 Agosto | 0 | 15 | 15 |
| 2018 Julio | 0 | 13 | 13 |
| 2018 Junio | 0 | 7 | 7 |
| 2018 Mayo | 0 | 10 | 10 |
| 2018 Abril | 0 | 3 | 3 |
| 2018 Marzo | 17 | 2 | 19 |
| 2018 Febrero | 40 | 5 | 45 |
| 2018 Enero | 45 | 8 | 53 |
| 2017 Diciembre | 50 | 9 | 59 |
| 2017 Noviembre | 41 | 5 | 46 |
| 2017 Octubre | 58 | 2 | 60 |
| 2017 Septiembre | 47 | 8 | 55 |
| 2017 Agosto | 39 | 4 | 43 |
| 2017 Julio | 27 | 6 | 33 |
| 2017 Junio | 53 | 29 | 82 |
| 2017 Mayo | 23 | 5 | 28 |
| 2017 Abril | 23 | 7 | 30 |
| 2017 Marzo | 25 | 28 | 53 |
| 2017 Febrero | 82 | 8 | 90 |
| 2017 Enero | 29 | 11 | 40 |
| 2016 Diciembre | 58 | 12 | 70 |
| 2016 Noviembre | 89 | 10 | 99 |
| 2016 Octubre | 122 | 19 | 141 |
| 2016 Septiembre | 166 | 9 | 175 |
| 2016 Agosto | 132 | 18 | 150 |
| 2016 Julio | 50 | 8 | 58 |
| 2016 Junio | 6 | 17 | 23 |
| 2016 Mayo | 6 | 11 | 17 |
| 2016 Abril | 4 | 1 | 5 |
| 2016 Marzo | 5 | 1 | 6 |
| 2016 Febrero | 11 | 4 | 15 |
| 2016 Enero | 13 | 3 | 16 |
| 2015 Diciembre | 7 | 0 | 7 |
| 2015 Noviembre | 6 | 2 | 8 |
| 2015 Octubre | 4 | 2 | 6 |
| 2015 Septiembre | 4 | 2 | 6 |
| 2015 Agosto | 5 | 2 | 7 |
| 2015 Julio | 33 | 3 | 36 |
| 2015 Junio | 28 | 6 | 34 |
| 2015 Mayo | 46 | 14 | 60 |
| 2015 Abril | 28 | 7 | 35 |
| 2015 Marzo | 30 | 7 | 37 |
| 2015 Febrero | 29 | 2 | 31 |
| 2015 Enero | 37 | 3 | 40 |
| 2014 Diciembre | 44 | 2 | 46 |
| 2014 Noviembre | 21 | 1 | 22 |
| 2014 Octubre | 43 | 8 | 51 |
| 2014 Septiembre | 41 | 5 | 46 |
| 2014 Agosto | 23 | 7 | 30 |
| 2014 Julio | 27 | 5 | 32 |
| 2014 Junio | 29 | 3 | 32 |
| 2014 Mayo | 44 | 10 | 54 |
| 2014 Abril | 36 | 5 | 41 |
| 2014 Marzo | 34 | 7 | 41 |
| 2014 Febrero | 28 | 4 | 32 |
| 2014 Enero | 29 | 4 | 33 |
| 2013 Diciembre | 22 | 4 | 26 |
| 2013 Noviembre | 9 | 2 | 11 |
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