Case and Research Letters
Fox-Fordyce Disease in a Pediatric Patient
Enfermedad de Fox-Fordyce en edad infantil
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Huerta-Vena, J.C. Tardío, A. Hernández-Núñez, J. Borbujo" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Huerta-Vena" "email" => array:1 [ 0 => "andrea.huerta@salud.madrid.org" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "J.C." "apellidos" => "Tardío" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "A." "apellidos" => "Hernández-Núñez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "J." "apellidos" => "Borbujo" "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 Universitario de Fuenlabrada, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario de Fuenlabrada, 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" => "Enfermedad de Fox-Fordyce en edad infantil" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 605 "Ancho" => 755 "Tamanyo" => 176101 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Accumulation of foam cells adjacent to a hair follicle.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">An 8-year-old girl was referred by endocrinology to the dermatology department for assessment of mildly pruritic lesions in both axillae. The lesions had developed over a long period of time and were more severe in the left armpit. The patient, whose parents were natives of Romania, was in follow-up by pediatric endocrinology due to precocious puberty, with slightly elevated testosterone levels (normal Synacthen test). She had also presented with an episode of acute gastroenteritis with liver failure, which required follow-up for 3 years.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Physical examination revealed yellowish elastic papules measuring between 1 and 2 mm, with a perifollicular distribution, grouped in localized plaques in both axillae, with more patchy grouping on the left side (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Histology of a punch biopsy of one of the lesions revealed dilation of the follicular infundibulum and a perifollicular aggregation of foam cells (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). These results were compatible with a diagnosis of Fox-Fordyce disease (FFD).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">The clinical course was favorable, with spontaneous improvement of the lesions. The only relevant family history was that her mother reported hyperhidrosis and axillary bromhidrosis, and she was therefore discharged with local antiperspirant on demand (aluminum chlorohydrate, 15%). It was not possible to establish a temporal relationship between the patient’s underlying endocrinologic disease and the appearance or improvement of the lesions.</p><p id="par0025" class="elsevierStylePara elsevierViewall">FFD, also known as apocrine miliaria, was first described in 1902 by George Henry Fox and John Addison Fordyce. It is a rare chronic disease that typically appears in postpubescent women (15–35 years of age).<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Presentation in childhood is exceptional.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The etiology is unknown and the cause is probably multifactorial.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Its association with hormonal factors has been postulated, given the age range in which it appears and the frequent remission of the disease during pregnancy and menopause, although it has not been possible to show any hormone abnormality in patients.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> We do not therefore know whether an association exists with the precocious puberty in our patient. We have found no previous association with liver failure in the literature.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Appearance of the disease as an adverse effect of laser or intense pulsed light (IPL) depilation techniques has been reported.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> In this case, the clinical and histologic findings are very similar to primary FFD.</p><p id="par0035" class="elsevierStylePara elsevierViewall">It presents clinically as small dome-shaped pruritic perifollicular papules the color of yellowish-healthy skin. The most common location is the axillae but the lesions may also present in the periareolar or anogenital region, and, more rarely, on the thighs, the periumbilical region, and sternum. The disease may be associated with hair loss in the affected areas and hypohidrosis.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The lesions improve with pregnancy and the use of hormonal contraceptives, and worsen with heat, hot water, stress, and the menstrual cycle.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The clinical presentation of the disease is constant and similar. Histological findings, however, vary widely.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Formation of a keratin plug in the follicular infundibulum, which also obstructs the apocrine duct may be the trigger.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The most consistent feature is dilation of the infundibulum and hyperkeratosis of the infundibular epithelium.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The most specific finding is perifollicular xanthomatosis.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">It was traditionally believed that this disease involved only the apocrine glands. Recent histological findings, however, suggest that eccrine and nonfollicular variants (involving the apocrine glands) may exist.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The clinical differential diagnosis of this entity should basically include eruptive syringoma, lichen nitidus, trichostasis spinulosa, Graham-Little-Piccardi-Lasseur syndrome, and Darier disease.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,7,8</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Because it is a rare disease, comparative studies are not available, and treatment remains unclear. Multiple treatments have been described, all of them with variable results. Topical or intralesional corticosteroids are considered to be the treatment of choice. Topical and systemic retinoids, topical antibiotics such as clindamycin, antiperspirants, oral contraceptives, and topical calcineurin inhibitors have also been used. Curettage and electrocoagulation, modified liposuction techniques, and, more recently, fractional CO<span class="elsevierStyleInf">2</span> laser, microwaves, and botulinum toxin A have been used in refractory cases.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9–11</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Spontaneous remission, as in the case of our patient, is anecdotal.</p><p id="par0070" class="elsevierStylePara elsevierViewall">In summary, we report a case of FFD that is exceptional due to onset in prepubertal age and its atypical course, with spontaneous resolution of the lesions. We cannot state the causal relationship with the patient’s endocrinologic abnormalities, although they probably play a major role in the development of the disease.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of interest</span><p id="par0075" 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: Huerta-Vena A, Tardío JC, Hernández-Núñez A, Borbujo J. Enfermedad de Fox-Fordyce en edad infantil. Actas Dermosifiliogr. 2020. <span class="elsevierStyleInterRef" id="intr0005" href="https://doi.org/10.1016/j.ad.2019.03.016">https://doi.org/10.1016/j.ad.2019.03.016</span></p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 505 "Ancho" => 755 "Tamanyo" => 35493 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Millimeter-sized confluent perifollicular papules with a yellowish color and elastic consistency, in the left armpit.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 505 "Ancho" => 755 "Tamanyo" => 32634 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Less severe lesions in the right armpit.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 605 "Ancho" => 755 "Tamanyo" => 176101 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Accumulation of foam cells adjacent to a hair follicle.</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" => "Fox-Fordyce disease (apocrine miliaria)" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "J. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 4 | 2 | 6 |
| 2024 Octubre | 80 | 42 | 122 |
| 2024 Septiembre | 94 | 27 | 121 |
| 2024 Agosto | 108 | 50 | 158 |
| 2024 Julio | 81 | 40 | 121 |
| 2024 Junio | 120 | 36 | 156 |
| 2024 Mayo | 96 | 41 | 137 |
| 2024 Abril | 102 | 21 | 123 |
| 2024 Marzo | 93 | 36 | 129 |
| 2024 Febrero | 81 | 30 | 111 |
| 2024 Enero | 75 | 45 | 120 |
| 2023 Diciembre | 86 | 22 | 108 |
| 2023 Noviembre | 108 | 37 | 145 |
| 2023 Octubre | 75 | 20 | 95 |
| 2023 Septiembre | 83 | 31 | 114 |
| 2023 Agosto | 56 | 13 | 69 |
| 2023 Julio | 74 | 51 | 125 |
| 2023 Junio | 66 | 25 | 91 |
| 2023 Mayo | 65 | 33 | 98 |
| 2023 Abril | 54 | 18 | 72 |
| 2023 Marzo | 71 | 34 | 105 |
| 2023 Febrero | 63 | 26 | 89 |
| 2023 Enero | 40 | 31 | 71 |
| 2022 Diciembre | 69 | 48 | 117 |
| 2022 Noviembre | 35 | 25 | 60 |
| 2022 Octubre | 34 | 25 | 59 |
| 2022 Septiembre | 27 | 36 | 63 |
| 2022 Agosto | 29 | 48 | 77 |
| 2022 Julio | 35 | 43 | 78 |
| 2022 Junio | 21 | 41 | 62 |
| 2022 Mayo | 71 | 56 | 127 |
| 2022 Abril | 105 | 32 | 137 |
| 2022 Marzo | 93 | 48 | 141 |
| 2022 Febrero | 89 | 32 | 121 |
| 2022 Enero | 87 | 31 | 118 |
| 2021 Diciembre | 89 | 32 | 121 |
| 2021 Noviembre | 83 | 39 | 122 |
| 2021 Octubre | 94 | 57 | 151 |
| 2021 Septiembre | 51 | 43 | 94 |
| 2021 Agosto | 54 | 40 | 94 |
| 2021 Julio | 68 | 30 | 98 |
| 2021 Junio | 61 | 44 | 105 |
| 2021 Mayo | 52 | 47 | 99 |
| 2021 Abril | 121 | 51 | 172 |
| 2021 Marzo | 86 | 39 | 125 |
| 2021 Febrero | 75 | 41 | 116 |
| 2021 Enero | 74 | 21 | 95 |
| 2020 Diciembre | 72 | 24 | 96 |
| 2020 Noviembre | 73 | 18 | 91 |
| 2020 Octubre | 56 | 33 | 89 |
| 2020 Septiembre | 15 | 10 | 25 |
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