Case and Research Letter
Folliculitis Decalvans and Lichen Planopilaris Phenotypic Spectrum With Pityriasis Amiantacea: A Challenging Diagnosis
Espectro fenotípico de foliculitis decalvante y liquen planopilar con pitiriasis amiantácea: un diagnóstico desafiante
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Bilateral and symmetrical whitish velvety plaques on the buccal, internal labial, and lateral lingual mucosae (B).</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "A. Llull-Ramos, I. Gracia-Darder, A. Martín-Santiago" "autores" => array:3 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "Llull-Ramos" ] 1 => array:2 [ "nombre" => "I." "apellidos" => "Gracia-Darder" ] 2 => array:2 [ "nombre" => "A." "apellidos" => "Martín-Santiago" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731024006446?idApp=UINPBA000044" "url" => "/00017310/0000011500000009/v2_202410160548/S0001731024006446/v2_202410160548/en/main.assets" ] "asociados" => array:1 [ 0 => array:18 [ "pii" => "S0001731024005258" "issn" => "00017310" "doi" => "10.1016/j.ad.2023.01.030" "estado" => "S300" "fechaPublicacion" => "2024-10-01" "aid" => "3997" "copyright" => "AEDV" "documento" => "simple-article" "crossmark" => 1 "subdocumento" => "crp" "cita" => "Actas Dermosifiliogr. 2024;115:T922-T924" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "es" => array:11 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Carta científico-clínica</span>" "titulo" => " Espectro fenotípico de foliculitis decalvante y liquen planopilar con pitiriasis amiantácea: un diagnóstico desafiante" "tienePdf" => "es" "tieneTextoCompleto" => "es" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "T922" "paginaFinal" => "T924" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Folliculitis Decalvans and Lichen Planopilaris Phenotypic Spectrum With Pityriasis Amiantacea: A Challenging Diagnosis" ] ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figura 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1868 "Ancho" => 1874 "Tamanyo" => 891538 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Tricoscopia: (a) Eritema intenso, menor densidad de folículos pilosos, descamación blanca perifolicular e interfolicular, penachos foliculares, costras hemorrágicas, zonas de color rojo lechoso, vasos sanguíneos dilatados. 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"apellidos" => "dos Anjos Bortolini" ] ] ] ] ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731024005258?idApp=UINPBA000044" "url" => "/00017310/0000011500000009/v2_202410160548/S0001731024005258/v2_202410160548/es/main.assets" ] ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letter</span>" "titulo" => "Folliculitis Decalvans and Lichen Planopilaris Phenotypic Spectrum With Pityriasis Amiantacea: A Challenging Diagnosis" "tieneTextoCompleto" => true "saludo" => "To the Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "922" "paginaFinal" => "924" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "E. Neves Souza, L. Martins Diniz, L. Amaral de Moura, B. dos Anjos Bortolini" "autores" => array:4 [ 0 => array:4 [ "nombre" => "E." "apellidos" => "Neves Souza" "email" => array:1 [ 0 => "neves.emilly@hotmail.com" ] "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "L." "apellidos" => "Martins Diniz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "L." "apellidos" => "Amaral de Moura" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "B." "apellidos" => "dos Anjos Bortolini" "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Universidade Federal do Espírito Santo (UFES), Vitória, ES, Brazil" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Hospital Universitário Cassiano Antônio Moraes (HUCAM), Vitória, ES, Brazil" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Espectro fenotípico de foliculitis decalvante y liquen planopilar con pitiriasis amiantácea: un diagnóstico desafiante" ] ] "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" => 570 "Ancho" => 1007 "Tamanyo" => 192372 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) A large patch of cicatricial alopecia on the vertex and crown, extending to left and right parietal scalp; intense erythema, adherent scales, yellow/hemorrhagic crusts, pustules and polytrichia. (b) Satisfactory improvement after therapeutical approach: reduction of erythema, pustules and scales, hair growth, and no progression of alopecia.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Folliculitis decalvans (FD) and lichen planopilaris (LPP) are primary cicatricial alopecias classified according to the predominant cell-type infiltrate on histopathology. FD is characterized by a neutrophilic infiltrate, while LPP is marked by a lymphocytic inflammation.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Clinically, FD presents as a patch cicatricial alopecia with tufted hairs, pustules, follicular papules and crusts, while LPP mainly exhibits perifollicular erythema and perifollicular scales.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Recently, an overlap of FD and LPP has been described as Folliculitis Decalvans and Lichen Planopilaris Phenotypic Spectrum (FDLPPPS), which combines clinical, trichoscopic and histopathological features of both diseases.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2–9</span></a> We report the case of a 21-year-old female patient with FDLPPPS associated with pityriasis amiantacea (PA), misdiagnosed as refractory seborrheic dermatitis.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A healthy 21-year-old female presented with a 13-year history of intense desquamation, pruritus and alopecia on her scalp. She referred multiple consultations with general practitioners, being repeatedly treated for seborrheic dermatitis, without improvement. The patient had no relevant family medical history. A year ago, she experienced a worsening of symptoms with pain and hair loss. She also had erythema, pustules and adherent crusts on her scalp. Dermatological examination revealed a large patch of cicatricial alopecia on the vertex and crown, extending to left and right parietal scalp; intense erythema, adherent scales, yellow/hemorrhagic crusts, pustules and polytrichia (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>a). Trichoscopy indicated decreased hair follicles density, perifollicular and interfollicular white scales, hair tufts, hemorrhagic crusts, milky-red areas, dilated blood vessels, and interfollicular erythema (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Histopathological study identified hyperkeratosis, absence of sebaceous glands, interstitial lymphoplasmacytic infiltrates, scarce neutrophils, plasma cells dermal aggregates, perifollicular concentric fibrosis, and granulomas around fragmented hair shafts (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). These findings suggested FDLPPPS associated with PA. We prescribed trimethoprim/sulfamethoxazole 160–800<span class="elsevierStyleHsp" style=""></span>mg twice a day, clobetasol propionate emulsion 0.05% daily, salicylic acid shampoo 2% in alternate days and oral prednisone 20<span class="elsevierStyleHsp" style=""></span>mg daily for 2 months. After this therapeutical approach, the lesions showed satisfactory improvement (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>b). Maintenance treatment was adopted with doxycycline 100<span class="elsevierStyleHsp" style=""></span>mg daily and oral prednisone 40<span class="elsevierStyleHsp" style=""></span>mg/day in weekend pulse. There was no progression of alopecia after 4 months of follow-up.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">In 2017, Morais et al.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> reported a case series of “LPP with pustules” revealing an atypical LPP presentation with crusts, follicular tufts and pustules. Later, the existence of FD-LPP phenotypic spectrum has been proposed.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,3</span></a> This disease can appear as a biphasic alopecia with FD preceding LPP or as the concomitant presence of both,<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,4</span></a> as we described. The pathogenesis is still unclear, but Yip et al.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> proposed that a microbiome dysbiosis may stimulate an abnormal cell recruitment and collapse hair follicle immune privilege, exposing follicular neoantigens and promoting an exacerbated inflammatory response.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The phenotypic spectrum usually affects the vertex through hairless patches and can be asymptomatic or exhibit pruritus, burning and pain.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> Trichoscopic findings include hair tufts (2–5 hairs), loss of follicular openings, milky red areas, perifollicular and interfollicular scale, erythema, yellow/hemorrhagic crusts, and pustules.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2–4</span></a> The childhood-onset of FDLPPPS reported herein is rare, and we were able to find only three cases in the literature.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,4</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Histopathological analysis reveals loss of sebaceous glands, multicompound follicular structures, perifollicular fibrosis, atrophy of the follicular epithelium, lymphohistiocytic infiltrate with granulomas, prominent plasma cells, and scarce or absent neutrophils.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2–4</span></a> Differential diagnosis includes tinea capitis, psoriasis and central centrifugal cicatricial alopecia.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4,8</span></a> PA is commonly associated with psoriasis and seborrheic dermatitis, but can also appear on primary cicatricial alopecias.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> The onset of PA secondary to FDLPPPS had not been described yet, but it can occur due to the severe inflammatory condition and long duration until diagnosis. There is no standard treatment, but some cases have improved with the combination of antibacterial and oral, intralesional or topical corticosteroids.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,4,9</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In conclusion, scarring alopecias are responsible for a strongly negative impact on patients’ quality of life, considering the physical and psychological implications associated with the condition. Further studies are needed to elucidate the pathogenesis of this distinctive alopecia and allow the establishment of treatment guidelines.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interests" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "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" => 570 "Ancho" => 1007 "Tamanyo" => 192372 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(a) A large patch of cicatricial alopecia on the vertex and crown, extending to left and right parietal scalp; intense erythema, adherent scales, yellow/hemorrhagic crusts, pustules and polytrichia. (b) Satisfactory improvement after therapeutical approach: reduction of erythema, pustules and scales, hair growth, and no progression of alopecia.</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" => 1868 "Ancho" => 1874 "Tamanyo" => 893576 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Trichoscopy: (a) Intense erythema, decreased hair follicles density, perifollicular and interfollicular white scales, hair tufts, hemorrhagic crusts, milky-red areas, dilated blood vessels. (b) Intense erythema, decreased hair follicles density, perifollicular white scales, hair tufts, yellow/hemorrhagic crusts, milky-red areas. (c) Hair tufts, adherent scales, yellow/hemorrhagic crusts. (d) Perifollicular white scales, hair tufts, adherent yellow crusts.</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" => 1324 "Ancho" => 1874 "Tamanyo" => 730727 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Histopathological study: (a, b) Vertical section showing absence of sebaceous glands, interstitial lymphoplasmacytic infiltrates, scarce neutrophils, plasma cells dermal aggregates, perifollicular concentric fibrosis, and granulomas around fragmented hair shafts (hematoxylin–eosin stain; ×100). (c) Horizontal section showing hyperkeratosis, perifollicular and interstitial lymphoplasmacytic infiltrates, scarce neutrophils, and a granuloma around the hair follicle (hematoxylin–eosin stain; ×40). (d) Horizontal section showing perifollicular and interstitial lymphoplasmacytic infiltrates, scarce neutrophils, and a granuloma around the hair follicle (hematoxylin–eosin stain, ×100).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Summary of North American Hair Research Society (NAHRS)-sponsored Workshop on Cicatricial Alopecia, Duke University Medical Center, February 10 and 11, 2001" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "E.A. Olsen" 1 => "W.F. Bergfeld" 2 => "G. Cotsarelis" 3 => "V.H. Price" 4 => "J. Shapiro" 5 => "R. 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