Case and Research Letters
Distal Digital Keratoacanthoma in Patients With Incontinentia Pigmenti
Queratoacantoma digital distal en paciente con incontinencia pigmenti
A. Martínez-López
, G. Blasco-Morente, I. Pérez-López, E. García-Durá
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Unidad de Gestión Clínica de Dermatología Médico-Quirúrgica y Venereología, Hospital Universitario Virgen de las Nieves, Granada, Spain
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Martínez-López, G. Blasco-Morente, I. Pérez-López, E. García-Durá" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Martínez-López" "email" => array:1 [ 0 => "antoniomartinezlopez@aol.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "G." "apellidos" => "Blasco-Morente" ] 2 => array:2 [ "nombre" => "I." "apellidos" => "Pérez-López" ] 3 => array:2 [ "nombre" => "E." "apellidos" => "García-Durá" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Unidad de Gestión Clínica de Dermatología Médico-Quirúrgica y Venereología, Hospital Universitario Virgen de las Nieves, Granada, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Queratoacantoma digital distal en paciente con incontinencia <span class="elsevierStyleItalic">pigmenti</span>" ] ] "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" => 1180 "Ancho" => 1667 "Tamanyo" => 138507 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Isoechogenic oval lesion beneath the nail plate. There is no central vascularization.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Distal digital keratoacanthoma (DKA) or subungual keratoacanthoma is a rare and aggressive variant of keratoacanthoma (KA) that typically arises in the distal subungual tissue or affects the proximal nail fold, giving rise to paronychia. It presents as a painful, expanding subungual tumor that frequently recurs.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> We present a patient with incontinentia pigmenti who developed DKA.</p><p id="par0010" class="elsevierStylePara elsevierViewall">A 59-year-old woman with a past history of incontinentia pigmenti consulted for the appearance 2 months earlier of a very tender lesion under the nail of her left great toe. There was no history of trauma. Physical examination revealed a hyperkeratotic tumor that was destroying the distal lateral border of the nail of the left great toe, with mild inflammation (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Plain x-ray of the foot showed no bone involvement. Histopathology of the lesion after surgical excision showed a hyperplastic epithelium surrounding central cystic areas; there were dyskeratotic keratinocytes with very little atypia. Staining for oncoprotein p53 was negative; staining for Ki-67 revealed occasional cells in division above the basal layer of the epidermis. These findings were compatible with subungual DKA. One-and-a-half months later, the patient returned due to the appearance of a lesion with similar characteristics in the area of the previous surgery, suggestive of tumor recurrence. Nail ultrasound showed thickening of the nail plate, under which was an isoechogenic, oval lesion measuring 0.71<span class="elsevierStyleHsp" style=""></span>cm in its longest axis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>), with no increase in central vascularization on Doppler study. Based on these findings, a second surgical excision was performed, with the same diagnosis.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">DKA is a rare destructive subtype of KA. It is more common in men, and particularly affects the first 3 digits of the hand.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> DKA typically develops as a rapidly growing and very painful verrucous or hyperkeratotic nodule that arises at the distal digital border of the nail, separating the nail plate from the bed, and it is usually associated with onycholysis.<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">4,5</span></a> The rapid growth of DKA often provokes bone erosion by compression; radiographically this appears as a clearly defined cup-shaped defect of the distal phalanx, with no associated sclerosis or periosteal reaction.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,4,5</span></a> Ultrasound usually demonstrates a mass of intermediate echogenicity with posterior acoustic enhancement due to the cortical erosion of the distal phalanx.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> The diagnosis is confirmed histologically by the presence of areas of epidermal hyperkeratosis and parakeratosis associated with a central crater full of keratin<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a>; the presence of dyskeratotic eosinophils is very common.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,7</span></a> An entity with similar clinical and histologic characteristics is the painful subungual tumor in incontinentia pigmenti, which can occasionally lead to diagnostic error because of its similar clinical appearance, although this latter lesion is more common on the fingers. The painful subungual tumor in incontinentia pigmenti occurs exclusively in women, predominantly between the ages of 15 and 30 years; a hormonal influence has been suggested because these tumors characteristically disappear during pregnancy and reappear postpartum.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Histologically, subungual tumor in incontinentia pigmenti presents a hyperplastic epidermis with papillomatosis, acanthosis, and prominent dyskeratotic keratinocytes. Immunohistochemistry usually shows mild-to-moderate positivity for p53, Ki-67, and PCNA (proliferating cell nuclear antigen). Although these lesions do not recur after surgical excision, the appearance of multiple subungual lesions at other sites is common.<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">8,9</span></a> To avoid this situation, some authors have proposed the use of systemic retinoids, with good results.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,8</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">In the differential diagnosis it is especially important to exclude squamous cell carcinoma (SCC), because of its therapeutic and prognostic implications. Epidemiologically, DKA usually arises in younger patients than SCC. In addition, DKA is characterized by rapid growth over a few weeks or months until it stabilizes (although, in contrast to KA at other anatomic sites, spontaneous regression is very rare), whereas SCC grows slowly but relentlessly.<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3,5</span></a> The use of immunohistochemistry stains for p53 and Ki-67 is useful for histologic differentiation of the 2 tumors, as these markers are diffusely positive in SCC but are negative in DKA.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> Other diseases that must be included in the differential diagnosis are common warts, subungual epidermal cysts, and onychomycosis. The most widely used initial treatment for DKA is conservative surgery, although recurrence is common, as in our case. Amputation is reserved for cases with multiple recurrences or when histologic differentiation from SCC is not conclusive.</p><p id="par0030" class="elsevierStylePara elsevierViewall">In conclusion, DKA is a rare tumor, but it tends to recur and produce bone erosion. Its relevance derives from the importance of the differential diagnosis with SCC, a neoplasm with a poorer prognosis and that requires a more aggressive treatment than was used in our case.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Funding</span><p id="par0035" class="elsevierStylePara elsevierViewall">The authors declare that they have received no funding for this study.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflicts of Interest</span><p id="par0040" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Funding" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflicts of Interest" ] 2 => 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: Martínez-López A, Blasco-Morente G, Pérez-López I, García-Durá E. Queratoacantoma digital distal en paciente con incontinencia <span class="elsevierStyleItalic">pigmenti</span>. Actas Dermosifiliogr. 2016;107:257–258.</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" => 650 "Ancho" => 1659 "Tamanyo" => 152675 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hyperkeratotic tumor that has destroyed the distal lateral border of the nail of the left great toe: A, Superior view. B, Inferior-lateral view.</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" => 1180 "Ancho" => 1667 "Tamanyo" => 138507 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Isoechogenic oval lesion beneath the nail plate. There is no central vascularization.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Distal digital keratoacanthoma: A report of 12 cases and a review of the literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "R. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 9 | 6 | 15 |
| 2024 Octubre | 67 | 41 | 108 |
| 2024 Septiembre | 82 | 23 | 105 |
| 2024 Agosto | 83 | 61 | 144 |
| 2024 Julio | 79 | 35 | 114 |
| 2024 Junio | 105 | 19 | 124 |
| 2024 Mayo | 89 | 24 | 113 |
| 2024 Abril | 78 | 28 | 106 |
| 2024 Marzo | 70 | 25 | 95 |
| 2024 Febrero | 58 | 29 | 87 |
| 2024 Enero | 66 | 29 | 95 |
| 2023 Diciembre | 87 | 22 | 109 |
| 2023 Noviembre | 74 | 23 | 97 |
| 2023 Octubre | 86 | 21 | 107 |
| 2023 Septiembre | 87 | 27 | 114 |
| 2023 Agosto | 61 | 18 | 79 |
| 2023 Julio | 66 | 30 | 96 |
| 2023 Junio | 68 | 23 | 91 |
| 2023 Mayo | 60 | 21 | 81 |
| 2023 Abril | 68 | 21 | 89 |
| 2023 Marzo | 69 | 27 | 96 |
| 2023 Febrero | 39 | 23 | 62 |
| 2023 Enero | 41 | 23 | 64 |
| 2022 Diciembre | 54 | 38 | 92 |
| 2022 Noviembre | 32 | 16 | 48 |
| 2022 Octubre | 23 | 21 | 44 |
| 2022 Septiembre | 47 | 37 | 84 |
| 2022 Agosto | 28 | 26 | 54 |
| 2022 Julio | 22 | 24 | 46 |
| 2022 Junio | 18 | 17 | 35 |
| 2022 Mayo | 56 | 27 | 83 |
| 2022 Abril | 35 | 35 | 70 |
| 2022 Marzo | 35 | 39 | 74 |
| 2022 Febrero | 31 | 21 | 52 |
| 2022 Enero | 47 | 34 | 81 |
| 2021 Diciembre | 35 | 30 | 65 |
| 2021 Noviembre | 57 | 43 | 100 |
| 2021 Octubre | 53 | 49 | 102 |
| 2021 Septiembre | 40 | 52 | 92 |
| 2021 Agosto | 59 | 29 | 88 |
| 2021 Julio | 34 | 26 | 60 |
| 2021 Junio | 44 | 22 | 66 |
| 2021 Mayo | 31 | 32 | 63 |
| 2021 Abril | 64 | 57 | 121 |
| 2021 Marzo | 60 | 35 | 95 |
| 2021 Febrero | 45 | 26 | 71 |
| 2021 Enero | 29 | 12 | 41 |
| 2020 Diciembre | 22 | 10 | 32 |
| 2020 Noviembre | 22 | 30 | 52 |
| 2020 Octubre | 20 | 9 | 29 |
| 2020 Septiembre | 20 | 15 | 35 |
| 2020 Agosto | 21 | 22 | 43 |
| 2020 Julio | 18 | 16 | 34 |
| 2020 Junio | 37 | 30 | 67 |
| 2020 Mayo | 26 | 25 | 51 |
| 2020 Abril | 24 | 19 | 43 |
| 2020 Marzo | 28 | 22 | 50 |
| 2020 Febrero | 2 | 2 | 4 |
| 2020 Enero | 0 | 2 | 2 |
| 2019 Diciembre | 4 | 9 | 13 |
| 2019 Noviembre | 0 | 2 | 2 |
| 2019 Octubre | 0 | 3 | 3 |
| 2019 Septiembre | 4 | 4 | 8 |
| 2019 Julio | 0 | 1 | 1 |
| 2019 Junio | 0 | 14 | 14 |
| 2019 Mayo | 3 | 12 | 15 |
| 2019 Abril | 1 | 2 | 3 |
| 2019 Marzo | 2 | 4 | 6 |
| 2018 Diciembre | 5 | 0 | 5 |
| 2018 Noviembre | 1 | 0 | 1 |
| 2018 Octubre | 3 | 0 | 3 |
| 2018 Septiembre | 4 | 0 | 4 |
| 2018 Febrero | 21 | 7 | 28 |
| 2018 Enero | 30 | 2 | 32 |
| 2017 Diciembre | 26 | 8 | 34 |
| 2017 Noviembre | 23 | 5 | 28 |
| 2017 Octubre | 20 | 4 | 24 |
| 2017 Septiembre | 18 | 7 | 25 |
| 2017 Agosto | 35 | 7 | 42 |
| 2017 Julio | 18 | 9 | 27 |
| 2017 Junio | 26 | 12 | 38 |
| 2017 Mayo | 27 | 8 | 35 |
| 2017 Abril | 26 | 6 | 32 |
| 2017 Marzo | 9 | 13 | 22 |
| 2017 Febrero | 16 | 9 | 25 |
| 2017 Enero | 13 | 8 | 21 |
| 2016 Diciembre | 22 | 16 | 38 |
| 2016 Noviembre | 29 | 20 | 49 |
| 2016 Octubre | 21 | 14 | 35 |
| 2016 Agosto | 1 | 1 | 2 |
| 2016 Junio | 0 | 1 | 1 |
| 2016 Mayo | 0 | 1 | 1 |
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