Case Letter
Idiopathic follicular mucinosis in childhood
Fernanda José Bauer
, José Roberto Paes de Almeida, Angelo Sementilli, Sandra Lopes Mattos e Dinato
Corresponding author
Department of Dermatology, Centro Universitário Lusíada, Santos, SP, Brazil
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=> true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "pt" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2666275220300989?idApp=UINPBA00008Z" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0365059620300489?idApp=UINPBA00008Z" "url" => "/03650596/0000009500000002/v1_202004180640/S0365059620300489/v1_202004180640/en/main.assets" ] "en" => array:17 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Letter</span>" "titulo" => "Idiopathic follicular mucinosis in childhood" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "268" "paginaFinal" => "270" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Fernanda José Bauer, José Roberto Paes de Almeida, Angelo Sementilli, Sandra Lopes Mattos e Dinato" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Fernanda José" "apellidos" => "Bauer" "email" => array:1 [ 0 => "fer_bauer_90@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "José Roberto Paes de" "apellidos" => "Almeida" ] 2 => array:2 [ "nombre" => "Angelo" "apellidos" => "Sementilli" ] 3 => array:2 [ "nombre" => "Sandra Lopes" "apellidos" => "Mattos e Dinato" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology, Centro Universitário Lusíada, Santos, SP, Brazil" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "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" => 1139 "Ancho" => 855 "Tamanyo" => 119903 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hypochromic lesion topped by shiny papules.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Follicular mucinosis is a rare condition, belonging to the group of cutaneous mucinoses, characterized by localized or diffuse mucin deposits in the skin or within hair follicles. Two forms were described: primary (or idiopathic) and secondary form, which may be associated with benign or malignant conditions.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The presence of well-defined papular, erythematous or reddish-brown erythema papules or plaques clinically characterize the condition. Follicular keratosis or alopecic patches could also be observed. Other less common forms have been described, such as acneiform, eczematous, cystic or nodular.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The patient is an 11-year-old white male, with no relevant personal or family history, with a 2 year history of asymptomatic cutaneous lesion on the face.</p><p id="par0020" class="elsevierStylePara elsevierViewall">On the dermatological examination: hypochromic lesion topped by follicular and nonfollicular shiny papules on the nasal, malar, zygomatic and left periorbital region and papular lesions on the upper right eyelid and mild left eyelid edema (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Sensory evaluation results are normal.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">A routine anatomopathological examination (Hematoxylin & eosin staining) reveals preserved epidermis. Dermis presents some hair follicles containing fibromyxoid stroma and mixed pattern inflammatory cells (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Alcian Blue stain reveals built-up mucin in the outer root sheath of the hair follicle (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Laboratory tests were requested to investigate associated diseases; all results were normal.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Treatment with daily application of high-potency topical corticosteroid showed improvement; however, recurrence occurred when discontinued.</p><p id="par0040" class="elsevierStylePara elsevierViewall">In 1957, Hermann Pinkus described a group of 6 patients with localized alopecia, histopathologically characterized by mucin deposits in the hair follicles, which he named alopecia mucinosa. Jablonska et al. proposed in 1959 to change the name to follicular mucinosis, a term accepted to this day.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Its cause is still unknown. Today, it is considered a standard reaction of the follicular epithelium to various factors.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">This dermatosis can have two clinical forms:<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2,4</span></a><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">1.</span><p id="par0055" class="elsevierStylePara elsevierViewall">Primary form: an idiopathic, benign and transient form, which commonly occurs in children and adults. In younger patients, it usually affects the head and neck, receding spontaneously after 2–24 months in most cases. Some rare cases of developing Hodgkin's disease, other lymphomas and leukemia have been recorded. Adult patients present generalized lesions that may last indefinitely.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">2.</span><p id="par0060" class="elsevierStylePara elsevierViewall">Secondary form: normally affects adults and older patients, and is associated with an underlying inflammatory or neoplastic condition. The most common is mycosis fungoides.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a></p></li></ul></p><p id="par0065" class="elsevierStylePara elsevierViewall">In the reported case, clinical and lab analysis allowed to exclude associated diseases and diagnose the condition as the idiopathic form of follicular mucinosis.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Histopathology is essential for diagnosis since it shows mucin deposits on the outer root sheath of the hair follicle, in addition to inflammatory infiltrates composed of lymphocytes, macrophages and eosinophils with folliculotropic lymphocytes. The benign form is determined by the extension of the eosinophilic inflammatory infiltrate and significant mucinous alterations in the follicular epithelium.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> Similar findings were observed in the patient‘s results. Presence of epidermotropic lymphocytes and dense perifollicular infiltrate of atypical cells suggest a malignant form of the condition; however, no evidence of it was found in the studied patient.</p><p id="par0075" class="elsevierStylePara elsevierViewall">It's interesting to mention that the chances of lymphomas appearing in follicular mucinosis patients varies greatly. The criteria adopted in the different evaluations change ranging from 14% to 32%. For example, Coskey and Mehregan detected lymphoma in 7 out of 50 follicular mucinosis patients (14%), Emmerson in 8 out of 47 patients (17%), Logan and Headington in 21 out of 80 patients (26%), Mehregan et al. in 9 out of 33 patients (27.2%) and Gibson et al. in 19 out of 59 patients (32%).<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">On the other hand, the frequency of the disease among patients with cutaneous lymphomas has not been established in literature. Marti et al. found the disease in 5 out of 43 patients (11%).<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">Regarding treatment, there is no specific medication for idiopathic follicular mucinosis. However, topical, intralesional and systemic corticosteroids, in addition to dapsone, antimalarial, isotretinoin and minocycline, can be used.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> Treatment was done using topical corticosteroids; the lesions improved but had a slight recurrence. These patients require periodic monitoring.</p><p id="par0090" class="elsevierStylePara elsevierViewall">The importance of the report is that is shows a rare incidence of follicular mucinosis in childhood. It is imperative to emphasize the obligation of long-term monitoring in order to completely rule out the connection with associated neoplasias.</p><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Financial support</span><p id="par0115" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Authors’ contributions</span><p id="par0095" class="elsevierStylePara elsevierViewall">Fernanda José Bauer: Conception and planning of the study; elaboration and writing of the manuscript; obtaining, analysis, and interpretation of the data; critical review of the literature; critical review of the manuscript.</p><p id="par0100" class="elsevierStylePara elsevierViewall">José Roberto Paes de Almeida: Approval of the final version of the manuscript; conception and planning of the study; effective participation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of the manuscript.</p><p id="par0105" class="elsevierStylePara elsevierViewall">Angelo Sementilli: Obtaining, analysis, and interpretation of the data.</p><p id="par0110" class="elsevierStylePara elsevierViewall">Sandra Lopes Mattos e Dinato: Approval of the final version of the manuscript.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0120" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0010" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0005" "titulo" => "Authors’ contributions" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-01-14" "fechaAceptado" => "2019-06-18" "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Bauer FJ, Almeida JRP, Sementilli A, Mattos e Dinato SL. Idiopathic follicular mucinosis in childhood. An Bras Dermatol. 2020;95:268–70.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Hospital Guilherme Álvaro, Santos, SP, Brazil.</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" => 1139 "Ancho" => 855 "Tamanyo" => 119903 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hypochromic lesion topped by shiny papules.</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" => 1188 "Ancho" => 855 "Tamanyo" => 242228 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Hair follicles containing fibromyxoid stroma (Hematoxylin & eosin, ×100).</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" => 1135 "Ancho" => 855 "Tamanyo" => 144578 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Mucin in the outer root sheath of the hair follicle (Alcian Blue, ×100).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Follicular mucinosis: literature review and case report" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "A.P.M. 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| 2022 November | 73 | 62 | 135 |
| 2022 October | 61 | 57 | 118 |
| 2022 September | 47 | 62 | 109 |
| 2022 August | 34 | 49 | 83 |
| 2022 July | 41 | 51 | 92 |
| 2022 June | 46 | 51 | 97 |
| 2022 May | 27 | 55 | 82 |
| 2022 April | 32 | 54 | 86 |
| 2022 March | 39 | 76 | 115 |
| 2022 February | 20 | 34 | 54 |
| 2022 January | 45 | 75 | 120 |
| 2021 December | 31 | 57 | 88 |
| 2021 November | 43 | 59 | 102 |
| 2021 October | 50 | 70 | 120 |
| 2021 September | 42 | 56 | 98 |
| 2021 August | 30 | 39 | 69 |
| 2021 July | 42 | 49 | 91 |
| 2021 June | 28 | 45 | 73 |
| 2021 May | 56 | 94 | 150 |
| 2021 April | 112 | 202 | 314 |
| 2021 March | 58 | 45 | 103 |
| 2021 February | 29 | 14 | 43 |
| 2021 January | 21 | 10 | 31 |
| 2020 December | 19 | 20 | 39 |
| 2020 November | 13 | 11 | 24 |
| 2020 October | 8 | 5 | 13 |
| 2020 September | 13 | 11 | 24 |
| 2020 August | 9 | 8 | 17 |
| 2020 July | 11 | 3 | 14 |
| 2020 June | 19 | 14 | 33 |
| 2020 May | 14 | 12 | 26 |
| 2020 April | 3 | 4 | 7 |
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