Case and Research Letters
Scarring Alopecia in Classic Adult Type I Pityriasis Rubra Pilaris
Alopecia cicatricial en pitiriasis rubra pilaris tipo I clásica del adulto
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Grandes placas de alopecia cicatricial con descamación folicular y eritema perifolicular parcheado que evolucionan a placas nacaradas atróficas con ausencia de eritema y descamación.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "C. Martín Callizo, J. Molinero Caturla, J. Sánchez Sánchez, R.M. Penín Mosquera" "autores" => array:4 [ 0 => array:2 [ "nombre" => "C." "apellidos" => "Martín Callizo" ] 1 => array:2 [ "nombre" => "J." "apellidos" => "Molinero Caturla" ] 2 => array:2 [ "nombre" => "J." "apellidos" => "Sánchez Sánchez" ] 3 => array:2 [ "nombre" => "R.M." 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Martín Callizo, J. Molinero Caturla, J. Sánchez Sánchez, R.M. Penín Mosquera" "autores" => array:4 [ 0 => array:4 [ "nombre" => "C." "apellidos" => "Martín Callizo" "email" => array:1 [ 0 => "claramartincallizo@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "J." "apellidos" => "Molinero Caturla" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "J." "apellidos" => "Sánchez Sánchez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "R.M." "apellidos" => "Penín Mosquera" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Dermatología, Hospital Universitari de Bellvitge, Barcelona, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitari de Bellvitge, Barcelona, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Alopecia cicatricial en pitiriasis rubra pilaris tipo I clásica del adulto" ] ] "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" => 1168 "Ancho" => 1600 "Tamanyo" => 616465 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A and B, Skin biopsy of a pityriasis rubra pilaris lesion showing hyperkeratosis alternating with parakeratosis. A, Hematoxylin-eosin, original magnification ×4. B, Hematoxylin-eosin, original magnification ×10. C and D, Scalp biopsy with stellate perifollicular dermal fibrosis. No perifollicular inflammatory infiltrate is seen. C, Hematoxylin-eosin, original magnification ×10. D, Hematoxylin-eosin, original magnification ×20.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Pityriasis rubra pilaris (PRP) is an inflammatory disease of unknown etiology<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> with an estimated incidence ranging from 1 in 5000 to 1 in 50 000 dermatology patients. The established Griffiths classification recognizes 5 groups (types I-V) according to clinical features and patient age.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> More recently, Miralles et al.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> added a sixth group after observing clinical differences in cases associated with human immunodeficiency virus (HIV) infection. Type I (classic adult PRP) is the most common subtype. Complete remission is achieved within 3 years of onset in 80% of cases,<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> whereas recurrence occurs in 20% of cases. Systemic retinoids, specifically acitretin, are the current first-line treatment.<span class="elsevierStyleVsp" style="height:0.5px"></span></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report for the first time a case of classic adult type I PRP associated at first recurrence with rapidly developing disseminated scarring alopecia of the scalp. The patient was a 41-year-old woman with no relevant past history who presented with a pruritic rash that had appeared 3 weeks earlier at the same time as an orange-hued waxy palmoplantar keratoderma (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>, A). The trunk and the proximal extremities had orange-red lesions with some apparently healthy areas of skin (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>, B-D). Skin biopsy confirmed the suspected diagnosis of classic adult type I PRP. Findings included alternating hyperkeratosis and parakeratosis as well as psoriasiform acanthosis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>, A and B). Treatment was started with 25 mg of oral acitretin daily. After 8 months of treatment the rash had cleared completely; we thus decided to discontinue treatment 2 months later. The patient returned after 10 lesion-free months complaining of a new crop of skin lesions similar to the original ones. This new bout occurred together with what appeared to be a severe form of scarring alopecia forming irregular patches on the scalp. The hairless patches were pearly and shiny, with fine scaling and perifollicular erythema. The clinical course was fulminant, and after 2 weeks the only scalp finding was atrophic alopecia patches with no erythema or scaling (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). A biopsy specimen obtained from the periphery of one of the hairless patches and comprising some follicles and perifollicular erythema exhibited significant perifollicular stellate fibrosis replacing the follicles, with no signs of inflammatory infiltrate (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>, C and D). Direct immunofluorescence was negative. Blood tests showed no abnormalities; test results for antinuclear antibodies, cryoglobulins, and antineutrophil cytoplasmic antibodies were all negative, and hepatitis B and C, HIV, and syphilis were ruled out. The rash cleared after 4 months of oral acitretin treatment, but the scarring alopecia persisted without change. The patient has worn a wig since then and has had no further bouts of PRP. Her scarring alopecia could not be reversed and remains stable at the time of writing in 2013, after 3 years of follow-up from initial presentation.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">The onset of scarring alopecia of the scalp in conjunction with a second bout of type I PRP suggested to us that alopecia might have been induced by the follicular involvement characteristic of PRP.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Although fine scaling of the scalp and face is frequent in type I PRP,<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and is sometimes accompanied by nonscarring alopecia,<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> an association of type I PRP with scarring alopecia has not been described before. In 1968, Bergeron and Stone<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> reported the case of a patient with PRP who developed acne conglobata, hidradenitis suppurativa, and dissecting cellulitis of the scalp. Acitretin-induced scarring alopecia has never before been reported,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> and in any case, our patient was no longer taking acitretin at the onset of the second bout and of scarring alopecia. The follicular hyperkeratosis and perifollicular erythema of the scalp present during both bouts of type I PRP could have disturbed our patient's hair follicles enough to trigger fulminant scarring alopecia.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The clinical appearance of our patient's scarring alopecia is reminiscent of pseudopelade of Brocq in large, scattered patches. It is unclear whether pseudopelade is a disease in itself or the final stage of other, primary forms of scarring alopecia. In the initial stage, our patient's condition resembled lichen planopilaris or cutaneous lupus erythematosus. Nevertheless, the absence of any perifollicular lymphocytic inflammatory infiltrate, the replacement of hair follicles with dermal fibrosis, and the negative direct immunofluorescence test results make lichen, lupus, and pseudopelade unlikely diagnoses.</p><p id="par0030" class="elsevierStylePara elsevierViewall">We therefore consider this to be a case of scarring alopecia of an unusually fulminant course and severity associated with a bout of type I PRP, and with histologic findings that rule out a primary classic form of scarring alopecia. The alopecia underwent rapid clinical transformation into generalized pseudopelade in large patches, but the only histologic finding was fibrosis.</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: Martín Callizo C. Alopecia cicatricial en pitiriasis rubra pilaris tipo I clásica del adulto. Actas Dermosifiliogr. 2014;105:955–957.</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" => 920 "Ancho" => 1500 "Tamanyo" => 267708 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">First bout of type I pityriasis rubra pilaris. A, Waxy palmar keratoderma. B-D, Scaly, orange-red patches on the trunk and limbs with islands of sparing and marked 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" => 1168 "Ancho" => 1600 "Tamanyo" => 616465 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A and B, Skin biopsy of a pityriasis rubra pilaris lesion showing hyperkeratosis alternating with parakeratosis. A, Hematoxylin-eosin, original magnification ×4. B, Hematoxylin-eosin, original magnification ×10. C and D, Scalp biopsy with stellate perifollicular dermal fibrosis. No perifollicular inflammatory infiltrate is seen. C, Hematoxylin-eosin, original magnification ×10. D, Hematoxylin-eosin, original magnification ×20.</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" => 807 "Ancho" => 900 "Tamanyo" => 191931 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Second bout of type I pityriasis rubra pilaris. Large patches of scarring alopecia with follicular flaking and patchy perifollicular erythema that developed into atrophic pearly patches without erythema or scaling.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:6 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pityriasis rubra pilaris: a review of diagnosis and treatment" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "A. Klein" 1 => "M. Landthaler" 2 => "S. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 12 | 7 | 19 |
| 2024 October | 78 | 40 | 118 |
| 2024 September | 107 | 32 | 139 |
| 2024 August | 130 | 54 | 184 |
| 2024 July | 114 | 37 | 151 |
| 2024 June | 130 | 26 | 156 |
| 2024 May | 129 | 32 | 161 |
| 2024 April | 112 | 38 | 150 |
| 2024 March | 117 | 131 | 248 |
| 2024 February | 110 | 47 | 157 |
| 2024 January | 112 | 39 | 151 |
| 2023 December | 114 | 16 | 130 |
| 2023 November | 161 | 31 | 192 |
| 2023 October | 148 | 23 | 171 |
| 2023 September | 195 | 35 | 230 |
| 2023 August | 132 | 25 | 157 |
| 2023 July | 121 | 37 | 158 |
| 2023 June | 81 | 35 | 116 |
| 2023 May | 133 | 21 | 154 |
| 2023 April | 189 | 18 | 207 |
| 2023 March | 162 | 26 | 188 |
| 2023 February | 120 | 31 | 151 |
| 2023 January | 100 | 40 | 140 |
| 2022 December | 99 | 42 | 141 |
| 2022 November | 57 | 27 | 84 |
| 2022 October | 57 | 17 | 74 |
| 2022 September | 92 | 46 | 138 |
| 2022 August | 78 | 54 | 132 |
| 2022 July | 73 | 39 | 112 |
| 2022 June | 61 | 24 | 85 |
| 2022 May | 116 | 45 | 161 |
| 2022 April | 140 | 33 | 173 |
| 2022 March | 114 | 46 | 160 |
| 2022 February | 142 | 29 | 171 |
| 2022 January | 191 | 35 | 226 |
| 2021 December | 112 | 28 | 140 |
| 2021 November | 114 | 43 | 157 |
| 2021 October | 117 | 51 | 168 |
| 2021 September | 95 | 33 | 128 |
| 2021 August | 81 | 24 | 105 |
| 2021 July | 99 | 22 | 121 |
| 2021 June | 124 | 28 | 152 |
| 2021 May | 146 | 24 | 170 |
| 2021 April | 429 | 42 | 471 |
| 2021 March | 174 | 14 | 188 |
| 2021 February | 116 | 20 | 136 |
| 2021 January | 72 | 22 | 94 |
| 2020 December | 117 | 14 | 131 |
| 2020 November | 69 | 22 | 91 |
| 2020 October | 77 | 15 | 92 |
| 2020 September | 61 | 17 | 78 |
| 2020 August | 56 | 15 | 71 |
| 2020 July | 60 | 21 | 81 |
| 2020 June | 36 | 22 | 58 |
| 2020 May | 40 | 21 | 61 |
| 2020 April | 36 | 18 | 54 |
| 2020 March | 23 | 21 | 44 |
| 2020 February | 8 | 9 | 17 |
| 2020 January | 0 | 3 | 3 |
| 2019 December | 4 | 3 | 7 |
| 2019 November | 0 | 12 | 12 |
| 2019 October | 0 | 3 | 3 |
| 2019 September | 3 | 6 | 9 |
| 2019 August | 0 | 14 | 14 |
| 2019 July | 4 | 13 | 17 |
| 2019 June | 0 | 12 | 12 |
| 2019 May | 2 | 8 | 10 |
| 2019 April | 0 | 9 | 9 |
| 2019 March | 0 | 6 | 6 |
| 2019 February | 2 | 6 | 8 |
| 2019 January | 0 | 6 | 6 |
| 2018 December | 1 | 9 | 10 |
| 2018 November | 1 | 3 | 4 |
| 2018 October | 1 | 1 | 2 |
| 2018 September | 3 | 2 | 5 |
| 2018 July | 0 | 1 | 1 |
| 2018 May | 0 | 5 | 5 |
| 2018 April | 0 | 13 | 13 |
| 2018 March | 1 | 0 | 1 |
| 2018 February | 69 | 6 | 75 |
| 2018 January | 105 | 11 | 116 |
| 2017 December | 51 | 9 | 60 |
| 2017 November | 43 | 6 | 49 |
| 2017 October | 42 | 4 | 46 |
| 2017 September | 27 | 8 | 35 |
| 2017 August | 38 | 10 | 48 |
| 2017 July | 30 | 5 | 35 |
| 2017 June | 55 | 19 | 74 |
| 2017 May | 22 | 21 | 43 |
| 2017 April | 33 | 15 | 48 |
| 2017 March | 13 | 7 | 20 |
| 2017 February | 24 | 8 | 32 |
| 2017 January | 13 | 19 | 32 |
| 2016 December | 27 | 8 | 35 |
| 2016 November | 36 | 10 | 46 |
| 2016 October | 25 | 20 | 45 |
| 2016 September | 0 | 8 | 8 |
| 2016 August | 0 | 2 | 2 |
| 2016 July | 4 | 1 | 5 |
| 2016 June | 11 | 6 | 17 |
| 2016 May | 16 | 1 | 17 |
| 2016 April | 19 | 1 | 20 |
| 2016 March | 19 | 1 | 20 |
| 2016 February | 6 | 13 | 19 |
| 2016 January | 12 | 2 | 14 |
| 2015 December | 13 | 4 | 17 |
| 2015 November | 9 | 2 | 11 |
| 2015 October | 17 | 3 | 20 |
| 2015 September | 15 | 1 | 16 |
| 2015 August | 15 | 2 | 17 |
| 2015 July | 26 | 3 | 29 |
| 2015 June | 21 | 3 | 24 |
| 2015 May | 6 | 8 | 14 |
| 2015 April | 5 | 9 | 14 |
| 2015 March | 15 | 4 | 19 |
| 2015 February | 7 | 2 | 9 |
| 2015 January | 3 | 5 | 8 |
| 2014 December | 4 | 2 | 6 |
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