What is your diagnosis?
Case for diagnosis. Ichthyosiform mycosis fungoides
Luciana Baptista Pereira
, Natália de Paiva Sobreira, Vanessa Barreto Rocha
Corresponding author
Dermatology Service, Faculty of Medicine, Hospital das Clínicas, Universidade Federal de Minas Gerais, Belo Horizonte, MG, Brazil
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Ichthyosiform mycosis fungoides" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "795" "paginaFinal" => "797" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Luciana Baptista Pereira, Natália de Paiva Sobreira, Vanessa Barreto Rocha" "autores" => array:3 [ 0 => array:4 [ "nombre" => "Luciana Baptista" "apellidos" => "Pereira" "email" => array:1 [ 0 => "lucianabaptistapereira@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Natália de Paiva" "apellidos" => "Sobreira" ] 2 => array:2 [ "nombre" => "Vanessa Barreto" "apellidos" => "Rocha" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Dermatology Service, Faculty of Medicine, Hospital das Clínicas, Universidade Federal de Minas Gerais, Belo Horizonte, MG, Brazil" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 1050 "Ancho" => 1675 "Tamanyo" => 193574 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0020" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Immunohistochemistry showing CD5 positivity in epidermotropic atypical lymphocytes.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case report</span><p id="par0005" class="elsevierStylePara elsevierViewall">A previously healthy 30-year-old female patient had presented pruritic lesions in the lower limbs that extended to the trunk and upper limbs for two years. She reported mild weight loss (<10% of baseline weight in 6 months), with no fever or night sweats. She denied using any medications. On physical examination, she had hyperchromic plaques with ichthyosiform desquamation on the trunk, axillae, abdomen, thighs and feet (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). The peripheral lymph nodes, liver and spleen were not palpable. No other changes were observed on physical examination.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Cervical, chest, abdominal, and pelvic CT scans showed no changes. Complete blood count, platelet levels, liver and kidney function, lactic dehydrogenase, beta-2-microglobulin, and TSH levels were within the normal limits; antinuclear factor (ANF) was 1/640, with nuclear coarse stippled or reticulated appearance; serology for cytomegalovirus and Epstein-Barr showed negative IgM and positive IgG; HTLV, HIV, viral hepatitis B and C serologies were non-reactive</p><p id="par0015" class="elsevierStylePara elsevierViewall">A skin biopsy showed hyperkeratosis with orthokeratosis, thinning of the granular layer, and exocytosis of atypical lymphocytes, grouped in Pautrier microabscesses. (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The lymphocytes were reactive with CD3, CD5 and CD8, but there was loss of CD7 reactivity (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia></span><span id="sec1010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">What is your diagnosis?</span><p id="par0020" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><p id="par0025" class="elsevierStylePara elsevierViewall"><ul class="elsevierStyleList" id="lis0010"><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">a)</span><p id="par0030" class="elsevierStylePara elsevierViewall">Ichthyosiform mycosis fungoides?</p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">b)</span><p id="par0035" class="elsevierStylePara elsevierViewall">Acquired ichthyosis associated with systemic malignancy (ovarian, thyroid, breast, lung cancer)?</p></li><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">c)</span><p id="par0040" class="elsevierStylePara elsevierViewall">Ichthyosis associated with the use of medications (hydroxyurea, clofazimine, statins)?</p></li><li class="elsevierStyleListItem" id="lsti0025"><span class="elsevierStyleLabel">d)</span><p id="par0045" class="elsevierStylePara elsevierViewall">Ichthyosis associated with infectious disease (leprosy, AIDS, HTLV-1 infection)?</p></li></ul></p></li></ul></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect00010">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">Ichthyoses can be hereditary or acquired. The hereditary forms are usually present at birth and their onset may occur later until the age of 13.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Acquired ichthyoses (AI) can be associated with different etiologies, such as malignant, infectious, autoimmune diseases, endocrinological and nutritional disorders, kidney and liver failure, drug reactions and sarcoidosis. They may manifest before or after the identification of the systemic disease.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Mycosis fungoides (MF) is the most common form of primary cutaneous T-cell lymphoma and presents a variety of clinical manifestations.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> An ichthyosiform eruption as the only manifestation of MF is a rare event (1.8% of cases).<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Ichthyosiform mycosis fungoides (IMF) is characterized by an indolent course of the disease and good prognosis, with a mean age of onset at 32 years.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> It may coexist with other subtypes of MF, especially the folliculotropic one, or manifest only as ichthyosiform lesions,<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> with a clinical course similar to ichthyosis vulgaris or other forms of ichthyosis.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a> They are preferentially located on the trunk and extremities, but the entire body surface may be affected.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Serology for HTLV is important to rule out the diagnosis of adult T-cell lymphoma/leukemia in these cases.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Although the clinical characteristics of IMF are indistinguishable from other causes of AI, histopathology discloses epidermotropic lymphocytic infiltrates, associated with typical characteristics of acquired ichthyosis such as orthokeratotic hyperkeratosis and thinning of the granular layer.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> Immunohistochemical evaluation of IMF usually shows CD3+ and CD4+ lymphocytes but there have been reports of a predominance of CD8+ lymphocytes, as in the reported case.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Although ichthyosiform lesions can coexist with typical MF lesions, the diagnosis of IMF should be considered when ichthyosis is the sole manifestation of MF.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Therefore, patients with acquired ichthyosis should be carefully evaluated to rule out this peculiar clinical-pathological variant.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Financial support</span><p id="par0075" class="elsevierStylePara elsevierViewall">None declared</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Authors’ contributions</span><p id="par0080" class="elsevierStylePara elsevierViewall">Luciana Baptista Pereira: Conception and patient diagnosis, data collection, article writing, approval of the final version of the manuscript.</p><p id="par0085" class="elsevierStylePara elsevierViewall">Natália de Paiva Sobreira: Conception of the article, data collection, writing of the article, approval of the final version of the manuscript.</p><p id="par0090" class="elsevierStylePara elsevierViewall">Vanessa Barreto Rocha: Conception of the article, writing of the manuscript, approval and review of the final version of the manuscript.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Conflicts of interest</span><p id="par0095" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case report" ] 1 => array:2 [ "identificador" => "sec1010" "titulo" => "What is your diagnosis?" ] 2 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 3 => array:2 [ "identificador" => "sec0015" "titulo" => "Financial support" ] 4 => array:2 [ "identificador" => "sec0020" "titulo" => "Authors’ contributions" ] 5 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflicts of interest" ] 6 => array:2 [ "identificador" => "xack673264" "titulo" => "Acknowledgments" ] 7 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2021-08-17" "fechaAceptado" => "2021-10-01" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Study conducted at the Dermatology Service, Cutaneous Lymphoma Outpatient Clinic, Hospital das Clínicas, Universidade Federal de Minas Gerais, Belo Horizonte, MG, Brazil.</p>" ] ] "multimedia" => array:4 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1601 "Ancho" => 1333 "Tamanyo" => 416103 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hyperchromic plaques with ichthyosiform desquamation on the posterior trunk region.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1770 "Ancho" => 1333 "Tamanyo" => 434712 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Hyperchromic plaques with ichthyosiform desquamation on the anterior trunk region.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1258 "Ancho" => 1675 "Tamanyo" => 522919 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Orthokeratotic hyperkeratosis hypogranulosis and exocytosis of atypical lymphocytes (Hematoxylin & eosin, ×40).</p>" ] ] 3 => array:8 [ "identificador" => "fig0020" "etiqueta" => "Figure 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 1050 "Ancho" => 1675 "Tamanyo" => 193574 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0020" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Immunohistochemistry showing CD5 positivity in epidermotropic atypical lymphocytes.</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" => "Acquired ichthyosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "N. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 19 | 14 | 33 |
| 2024 October | 97 | 72 | 169 |
| 2024 September | 130 | 116 | 246 |
| 2024 August | 163 | 131 | 294 |
| 2024 July | 125 | 118 | 243 |
| 2024 June | 95 | 94 | 189 |
| 2024 May | 100 | 60 | 160 |
| 2024 April | 98 | 95 | 193 |
| 2024 March | 94 | 74 | 168 |
| 2024 February | 114 | 86 | 200 |
| 2024 January | 70 | 62 | 132 |
| 2023 December | 81 | 61 | 142 |
| 2023 November | 87 | 89 | 176 |
| 2023 October | 73 | 74 | 147 |
| 2023 September | 72 | 68 | 140 |
| 2023 August | 67 | 31 | 98 |
| 2023 July | 66 | 31 | 97 |
| 2023 June | 64 | 40 | 104 |
| 2023 May | 62 | 26 | 88 |
| 2023 April | 46 | 18 | 64 |
| 2023 March | 55 | 50 | 105 |
| 2023 February | 45 | 44 | 89 |
| 2023 January | 65 | 50 | 115 |
| 2022 December | 105 | 53 | 158 |
| 2022 November | 108 | 93 | 201 |
| 2022 October | 142 | 91 | 233 |
| 2022 September | 32 | 44 | 76 |
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