Case and Research Letters
Follicular Mucinosis in Childhood: A Case Report and Review of the Literature
Mucinosis folicular en la infancia: aportación de un caso y revisión de la literatura
R. Bella-Navarro
, N. Martí-Fajardo, J.M. Martín-Hernández, E. Jordá-Cuevas
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Servicio de Dermatología, Hospital Clínico Universitario, Valencia, Spain
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Bella-Navarro, N. Martí-Fajardo, J.M. Martín-Hernández, E. Jordá-Cuevas" "autores" => array:4 [ 0 => array:4 [ "nombre" => "R." "apellidos" => "Bella-Navarro" "email" => array:1 [ 0 => "rbellanavarro@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "N." "apellidos" => "Martí-Fajardo" ] 2 => array:2 [ "nombre" => "J.M." "apellidos" => "Martín-Hernández" ] 3 => array:2 [ "nombre" => "E." "apellidos" => "Jordá-Cuevas" ] ] "afiliaciones" => array:1 [ 0 => array:1 [ "entidad" => "Servicio de Dermatología, Hospital Clínico Universitario, Valencia, Spain" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Mucinosis folicular en la infancia: aportación de un caso y revisión de la literatura" ] ] "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" => 871 "Ancho" => 2000 "Tamanyo" => 477829 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Histopathology revealed several hair follicles with abundant intraepithelial mucin in the dermis. No accompanying inflammatory infiltrate was observed. B, Detail showing intraepithelial mucin.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Follicular mucinosis is defined as the accumulation of mucin in the follicular epithelium and sebaceous glands.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It can present in isolation with an unknown etiology<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> or in association with several conditions, including various types of hematologic malignancy.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of an 8-year-old girl with no medical history of interest who was referred for a lesion on the dorsum of the nose that had appeared 2 months previously. She had been treated unsuccessfully with 1% hydrocortisone cream prescribed by her primary care physician.</p><p id="par0015" class="elsevierStylePara elsevierViewall">The lesion was a well-delimited, slow-growing, prominent, pruritic, and slightly infiltrated erythematous plaque (0.7<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>1<span class="elsevierStyleHsp" style=""></span>cm) with follicular hyperkeratosis (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The patient reported no history of injury, local infection, or insect bite and had no other lesions or palpable locoregional lymph nodes.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Histopathology revealed several hair follicles with abundant intraepithelial mucin in the dermis, although there was no evidence of an inflammatory infiltrate or other signs of malignancy. The findings were compatible with a diagnosis of follicular mucinosis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">After administration of topical 1% methylprednisolone aceponate cream twice daily for 7 days, the lesion resolved completely, with no signs of recurrence after 1 year of follow-up.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Follicular mucinosis was first reported by Pinkus<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> in 1957 in a description of the histology findings for a series of patients with characteristic cutaneous lesions and mucin deposits in the hair follicles.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The same year, Braun-Falco<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> reported the first cases of follicular mucinosis associated with hematologic malignancies, specifically lymphoreticular malignancies.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Emmerson and Coskey later established the current classification of the disease into 3 well-differentiated forms.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The first type, known as benign follicular mucinosis, usually affects children and young adults and is generally not associated with comorbidity. The lesions are usually located on the face and tend to resolve spontaneously in a few months.</p><p id="par0050" class="elsevierStylePara elsevierViewall">The second type, lymphoma-associated follicular mucinosis, which comprises conditions such as mycosis fungoides or Sézary syndrome, differs from the first type in that it affects older patients (usually aged 40-70 years) and has a chronic course; there is no underlying systemic disease.</p><p id="par0055" class="elsevierStylePara elsevierViewall">The third type is follicular mucinosis, which is a manifestation of systemic malignancy and mainly affects elderly patients.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">In the recently added fourth type, reactive follicular mucinosis, the histology findings that are typical of follicular mucinosis are observed as incidental findings in a wide variety of dermatoses or neoplasms.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Clinically, follicular mucinosis is characterized by the presence of clearly defined, slightly infiltrated erythematous plaques or papules, with follicular projections, superficial scaling, and alopecia in terminal hair–bearing areas. Other less common presentations include acneiform lesions and chronic eczema; therefore, in most cases, histopathology is necessary to confirm the diagnosis.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Histologically, follicular mucinosis is characterized by mucinous degeneration of the follicular epithelium, with the presence of dense fibrous material in the form of amorphous mucin deposits in the pilosebaceous units and a mixed inflammatory infiltrate. Despite attempts to establish distinctive criteria such as density of the deposit in the follicles, histology findings do not enable us to differentiate reliably between primary follicular mucinosis and mucinosis associated with mycosis fungoides.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">Follicular mucinosis is uncommon in children. Its lesions have been reported to mimic lichen spinulosus and keratosis pilaris, both of which are the main entities in the differential diagnosis in children with suspicious lesions.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The lesions generally have a good prognosis and resolve spontaneously.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1-5</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">Nevertheless, follicular mucinosis in children is not always self-limiting or benign, but can also be associated with malignant processes such as Hodgkin disease. Presentation on the head and neck, diagnosis in childhood, and the presence of a solitary lesion generally indicate a good prognosis.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2,5</span></a> Nonetheless, medium-term to long-term follow-up is essential, and new biopsy specimens should be taken from persistent lesions to test for lymphoproliferative disease.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,8,9</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">No standard treatment regimens have been established. Many strategies have been applied, with varying results, including observation, no treatment, corticosteroids (topical, intralesional, and systemic), psoralen-UV-A, dapsone, antimalarial agents, oral retinoids, and interferon alfa-2b.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0090" class="elsevierStylePara elsevierViewall">In conclusion, although a rare condition in childhood, follicular mucinosis should be considered in the differential diagnosis of lesions with prominent follicular hyperkeratosis. Biopsy is necessary for an early diagnosis and appropriate clinical follow-up.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Bella-Navarro R, et al. Mucinosis folicular en la infancia: aportación de un caso y revisión de la literatura. Actas Dermosifiliogr.2012;103:335-6.</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" => 713 "Ancho" => 950 "Tamanyo" => 80641 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Well-defined, slightly infiltrated, prominent erythematous plaque with follicular hyperkeratosis.</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" => 871 "Ancho" => 2000 "Tamanyo" => 477829 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A, Histopathology revealed several hair follicles with abundant intraepithelial mucin in the dermis. No accompanying inflammatory infiltrate was observed. B, Detail showing intraepithelial mucin.</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" => "Follicular mucinosis in a 4-year-old boy" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "B.N. Locksin" 1 => "A. Khachemoune" 2 => "C. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 12 | 13 | 25 |
| 2024 Octubre | 136 | 38 | 174 |
| 2024 Septiembre | 129 | 33 | 162 |
| 2024 Agosto | 145 | 67 | 212 |
| 2024 Julio | 128 | 45 | 173 |
| 2024 Junio | 139 | 58 | 197 |
| 2024 Mayo | 145 | 53 | 198 |
| 2024 Abril | 107 | 31 | 138 |
| 2024 Marzo | 134 | 35 | 169 |
| 2024 Febrero | 96 | 40 | 136 |
| 2024 Enero | 103 | 31 | 134 |
| 2023 Diciembre | 119 | 28 | 147 |
| 2023 Noviembre | 114 | 32 | 146 |
| 2023 Octubre | 123 | 29 | 152 |
| 2023 Septiembre | 170 | 48 | 218 |
| 2023 Agosto | 97 | 32 | 129 |
| 2023 Julio | 125 | 41 | 166 |
| 2023 Junio | 102 | 20 | 122 |
| 2023 Mayo | 77 | 32 | 109 |
| 2023 Abril | 86 | 40 | 126 |
| 2023 Marzo | 115 | 36 | 151 |
| 2023 Febrero | 57 | 26 | 83 |
| 2023 Enero | 36 | 30 | 66 |
| 2022 Diciembre | 36 | 44 | 80 |
| 2022 Noviembre | 39 | 23 | 62 |
| 2022 Octubre | 43 | 37 | 80 |
| 2022 Septiembre | 34 | 41 | 75 |
| 2022 Agosto | 43 | 34 | 77 |
| 2022 Julio | 36 | 34 | 70 |
| 2022 Junio | 32 | 29 | 61 |
| 2022 Mayo | 75 | 37 | 112 |
| 2022 Abril | 81 | 43 | 124 |
| 2022 Marzo | 83 | 62 | 145 |
| 2022 Febrero | 59 | 42 | 101 |
| 2022 Enero | 83 | 41 | 124 |
| 2021 Diciembre | 48 | 43 | 91 |
| 2021 Noviembre | 57 | 54 | 111 |
| 2021 Octubre | 65 | 67 | 132 |
| 2021 Septiembre | 84 | 41 | 125 |
| 2021 Agosto | 72 | 51 | 123 |
| 2021 Julio | 48 | 44 | 92 |
| 2021 Junio | 68 | 26 | 94 |
| 2021 Mayo | 50 | 50 | 100 |
| 2021 Abril | 160 | 82 | 242 |
| 2021 Marzo | 72 | 51 | 123 |
| 2021 Febrero | 57 | 55 | 112 |
| 2021 Enero | 46 | 36 | 82 |
| 2020 Diciembre | 54 | 43 | 97 |
| 2020 Noviembre | 18 | 34 | 52 |
| 2020 Octubre | 32 | 20 | 52 |
| 2020 Septiembre | 47 | 16 | 63 |
| 2020 Agosto | 25 | 25 | 50 |
| 2020 Julio | 18 | 17 | 35 |
| 2020 Junio | 33 | 32 | 65 |
| 2020 Mayo | 27 | 23 | 50 |
| 2020 Abril | 31 | 27 | 58 |
| 2020 Marzo | 27 | 24 | 51 |
| 2020 Febrero | 5 | 6 | 11 |
| 2020 Enero | 3 | 10 | 13 |
| 2019 Diciembre | 4 | 18 | 22 |
| 2019 Noviembre | 4 | 5 | 9 |
| 2019 Octubre | 0 | 18 | 18 |
| 2019 Septiembre | 4 | 18 | 22 |
| 2019 Agosto | 4 | 12 | 16 |
| 2019 Julio | 4 | 18 | 22 |
| 2019 Junio | 4 | 17 | 21 |
| 2019 Mayo | 4 | 28 | 32 |
| 2019 Abril | 2 | 44 | 46 |
| 2019 Marzo | 0 | 13 | 13 |
| 2019 Febrero | 2 | 7 | 9 |
| 2019 Enero | 0 | 7 | 7 |
| 2018 Diciembre | 3 | 5 | 8 |
| 2018 Noviembre | 1 | 0 | 1 |
| 2018 Octubre | 4 | 0 | 4 |
| 2018 Septiembre | 2 | 6 | 8 |
| 2018 Agosto | 0 | 8 | 8 |
| 2018 Julio | 0 | 9 | 9 |
| 2018 Junio | 0 | 3 | 3 |
| 2018 Mayo | 0 | 7 | 7 |
| 2018 Abril | 0 | 7 | 7 |
| 2018 Marzo | 2 | 9 | 11 |
| 2018 Febrero | 122 | 6 | 128 |
| 2018 Enero | 102 | 13 | 115 |
| 2017 Diciembre | 155 | 10 | 165 |
| 2017 Noviembre | 77 | 14 | 91 |
| 2017 Octubre | 73 | 14 | 87 |
| 2017 Septiembre | 45 | 6 | 51 |
| 2017 Agosto | 76 | 26 | 102 |
| 2017 Julio | 77 | 7 | 84 |
| 2017 Junio | 67 | 14 | 81 |
| 2017 Mayo | 44 | 10 | 54 |
| 2017 Abril | 48 | 10 | 58 |
| 2017 Marzo | 34 | 11 | 45 |
| 2017 Febrero | 114 | 9 | 123 |
| 2017 Enero | 46 | 13 | 59 |
| 2016 Diciembre | 62 | 3 | 65 |
| 2016 Noviembre | 105 | 9 | 114 |
| 2016 Octubre | 103 | 21 | 124 |
| 2016 Septiembre | 172 | 18 | 190 |
| 2016 Agosto | 98 | 21 | 119 |
| 2016 Julio | 49 | 12 | 61 |
| 2016 Junio | 6 | 17 | 23 |
| 2016 Mayo | 10 | 8 | 18 |
| 2016 Abril | 4 | 1 | 5 |
| 2016 Marzo | 8 | 2 | 10 |
| 2016 Febrero | 5 | 13 | 18 |
| 2016 Enero | 10 | 1 | 11 |
| 2015 Diciembre | 10 | 4 | 14 |
| 2015 Noviembre | 21 | 2 | 23 |
| 2015 Octubre | 22 | 12 | 34 |
| 2015 Septiembre | 11 | 6 | 17 |
| 2015 Agosto | 17 | 6 | 23 |
| 2015 Julio | 105 | 14 | 119 |
| 2015 Junio | 79 | 11 | 90 |
| 2015 Mayo | 109 | 19 | 128 |
| 2015 Abril | 98 | 19 | 117 |
| 2015 Marzo | 99 | 11 | 110 |
| 2015 Febrero | 73 | 13 | 86 |
| 2015 Enero | 122 | 20 | 142 |
| 2014 Diciembre | 85 | 18 | 103 |
| 2014 Noviembre | 69 | 16 | 85 |
| 2014 Octubre | 78 | 11 | 89 |
| 2014 Septiembre | 73 | 13 | 86 |
| 2014 Agosto | 78 | 17 | 95 |
| 2014 Julio | 88 | 23 | 111 |
| 2014 Junio | 111 | 15 | 126 |
| 2014 Mayo | 99 | 15 | 114 |
| 2014 Abril | 39 | 2 | 41 |
| 2014 Marzo | 44 | 12 | 56 |
| 2014 Febrero | 45 | 8 | 53 |
| 2014 Enero | 39 | 6 | 45 |
| 2013 Diciembre | 31 | 12 | 43 |
| 2013 Noviembre | 22 | 11 | 33 |
| 2013 Octubre | 31 | 14 | 45 |
| 2013 Septiembre | 31 | 12 | 43 |
| 2013 Agosto | 40 | 14 | 54 |
| 2013 Julio | 20 | 18 | 38 |
| 2013 Junio | 15 | 30 | 45 |
| 2013 Mayo | 19 | 20 | 39 |
| 2013 Abril | 17 | 15 | 32 |
| 2013 Marzo | 15 | 8 | 23 |
| 2013 Febrero | 34 | 6 | 40 |
| 2013 Enero | 53 | 10 | 63 |
| 2012 Diciembre | 28 | 4 | 32 |
| 2012 Noviembre | 1 | 0 | 1 |
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