Case Letter
Treatment of Kimura's disease with oral corticosteroid and methotrexate
Han Ma
Department of Dermatology, the Fifth Affiliated Hospital, Sun Yat-sen University, Zhuhai, Guangdong, China
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"idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case Letter</span>" "titulo" => "Treatment of Kimura's disease with oral corticosteroid and methotrexate" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "115" "paginaFinal" => "117" ] ] "autores" => array:1 [ 0 => array:3 [ "autoresLista" => "Han Ma" "autores" => array:1 [ 0 => array:3 [ "nombre" => "Han" "apellidos" => "Ma" "email" => array:1 [ 0 => "drmahan@sina.com" ] ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Department of Dermatology, the Fifth Affiliated Hospital, Sun Yat-sen University, Zhuhai, Guangdong, China" "identificador" => "aff0005" ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 518 "Ancho" => 758 "Tamanyo" => 104194 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Nodular lymphocytic infiltrate with germinal centers involving the dermis and subcutaneous tissue, and reactive germinal centers surrounded by small mature lymphocytes and eosinophils (arrow) (Hematoxylin & eosin, ×100).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Kimura's disease (KD) was initially described by Kim and Szeto in 1937, and became better known after a systematic description provided by Kimura as a chronic inflammatory disease.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Most cases reported occurred in Asian men between 20 and 30 years of age.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> Therapeutic modalities for KD include surgical excision, radiotherapy, and various immunomodulating agents, such as oral corticosteroids, cyclosporine, leflunomide, and mycophenolate mofetil.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> We report a case of KD with an excellent and sustained response to oral corticosteroid and intravenous methotrexate. A 51-year-old man presented with a history of fullness of the bilateral upper eyelids and a similar swelling in the bilateral parotid regions for seven years (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>); itching or pain symptoms. Physical examination revealed soft, pendular, non-tender mass lesions on both lateral upper eyelids, resulting in mechanical ptosis. The remainder of the ocular examination was within normal limits. His past medical history was unremarkable. Complete rheumatologic and immunologic workup was performed. Complete blood count showed the total number of white blood cells was 8.3<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L, neutrophils 4.35<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L (accounting for 52.4%), lymphocytes 2.50<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L (accounting for 30.1%), and eosinophils 1.01<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleSup">9</span>/L (accounting for 12.2%). Serum IgE was 205<span class="elsevierStyleHsp" style=""></span>IU/mL (normal, <100). Remaining laboratory results were normal. Computed tomography scan revealed soft-tissue lesions involving both the upper eyelid and parotid regions. A post-contrast study showed intense homogeneous enhancement on delayed scans (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Histopathology of the lesion excised from the left upper eyelid showed lymphoid tissue hyperplasia, with lymphoid nodules containing germinal centers that were scattered in the dermis and subcutaneous tissue, with scattered eosinophilic infiltration (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Based on the clinical manifestations and histopathological features, KD was then diagnosed. The therapeutic regimen comprised a tapering dose of oral prednisone (initial dose 40<span class="elsevierStyleHsp" style=""></span>mg/d) and intravenous methotrexate at 15<span class="elsevierStyleHsp" style=""></span>mg/week for two months. The patient had complete resolution after treatment and there was no recurrence in the next two years of follow-up. KD is a chronic inflammatory disease that manifests as a triad of subcutaneous nodules in the head and neck region, peripheral blood eosinophilia, and elevated serum IgE.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> It may also involve extracutaneous sites, such as regional lymph nodes, major salivary glands, and the kidneys. However, renal involvement is not uncommon and most frequently results in nephritic syndrome.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> The patient presented all the three typical elements to fulfill the diagnostic criteria and both sides of salivary glands had been involved. Thus, KD was the first diagnosis considered. This disease must be distinguished from angiolymphoid hyperplasia with eosinophilia (ALHE) because of several overlapping clinical and histologic features. KD occurs mainly in young men of Asian descent with one or multiple asymptomatic masses involving the subcutaneous tissue and salivary glands. It is often accompanied by regional lymph node involvement, peripheral blood eosinophilia, and elevated IgE. In contrast, ALHE occurs predominantly in middle-aged women, presenting with multiple small papules or erythematous nodules associated with itching.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> In the histopathologic features, KD displays the presence of numerous lymphoid follicles and the absence of irregular, dilated blood vessels,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">2</span></a> just like what was observed in this case. The pathogenesis of KD remains unknown, but allergy, atopy, autoimmunity, and parasite infestation are considered possible risk factors.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> Previous studies have found increased levels of interleukin-4, interleukin-5, and interleukin-13 in the peripheral blood of affected individuals, suggesting a role for type 2 T-helper cytokines.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> Therapeutic methods reported in the literature are heterogeneous, but surgical excision and oral corticosteroids represent the most frequently used strategies.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> To avoid recurrence in the course of tapering steroids, various immunomodulating agents should be added in the treatment plan. Leflunomide and mycophenolate mofetil have shown promise effective in some reported cases.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a> But the two drugs are still expensive, so we chose methotrexate as the combined drug, which exhibits immunomodulatory effects in a similar fashion by inhibiting <span class="elsevierStyleItalic">de novo</span> purine synthesis <span class="elsevierStyleItalic">via</span> inosine monophosphate dehydrogenase. Although recurrence is very common, it did not occurred in the present patient within the next two years of follow-up. The author feels that methotrexate may be a promising therapy for KD.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0010" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Author's contribution</span><p id="par0015" class="elsevierStylePara elsevierViewall">Han Ma: Approval of the final version of the manuscript; elaboration and writing of the manuscript.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0020" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Author's contribution" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-02-25" "fechaAceptado" => "2019-03-01" "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Ma H. Treatment of Kimura's disease with oral corticosteroid and methotrexate. An Bras Dermatol. 2020;95:115–7.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Department of Dermatology, the Fifth Affiliated Hospital, Sun Yat-sen University, Zhuhai, Guangdong, China.</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" => 989 "Ancho" => 756 "Tamanyo" => 64931 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Fullness of the bilateral upper eyelids and swelling in the bilateral parotid regions.</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" => 823 "Ancho" => 758 "Tamanyo" => 78776 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Soft-tissue lesions involving both the upper eyelid and parotid regions.</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" => 518 "Ancho" => 758 "Tamanyo" => 104194 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Nodular lymphocytic infiltrate with germinal centers involving the dermis and subcutaneous tissue, and reactive germinal centers surrounded by small mature lymphocytes and eosinophils (arrow) (Hematoxylin & eosin, ×100).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Angiolymphoid hyperplasia with eosinophilia versus Kimura's disease: a case report and a clinical and histopathological comparison" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "J.T. 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| Ano/Mês | Html | Total | |
|---|---|---|---|
| 2024 Novembro | 34 | 8 | 42 |
| 2024 Outubro | 177 | 72 | 249 |
| 2024 Setembro | 207 | 88 | 295 |
| 2024 Agosto | 211 | 110 | 321 |
| 2024 Julho | 158 | 114 | 272 |
| 2024 Junho | 138 | 68 | 206 |
| 2024 Maio | 112 | 59 | 171 |
| 2024 Abril | 158 | 86 | 244 |
| 2024 Março | 93 | 63 | 156 |
| 2024 Fevereiro | 86 | 70 | 156 |
| 2024 Janeiro | 75 | 47 | 122 |
| 2023 Dezembro | 58 | 60 | 118 |
| 2023 Novembro | 85 | 70 | 155 |
| 2023 Outubro | 47 | 76 | 123 |
| 2023 Setembro | 56 | 60 | 116 |
| 2023 Agosto | 51 | 31 | 82 |
| 2023 Julho | 49 | 37 | 86 |
| 2023 Junho | 50 | 32 | 82 |
| 2023 Maio | 48 | 27 | 75 |
| 2023 Abril | 34 | 16 | 50 |
| 2023 Março | 47 | 44 | 91 |
| 2023 Fevereiro | 36 | 31 | 67 |
| 2023 Janeiro | 43 | 45 | 88 |
| 2022 Dezembro | 41 | 30 | 71 |
| 2022 Novembro | 57 | 43 | 100 |
| 2022 Outubro | 94 | 46 | 140 |
| 2022 Setembro | 40 | 58 | 98 |
| 2022 Agosto | 32 | 40 | 72 |
| 2022 Julho | 36 | 58 | 94 |
| 2022 Junho | 28 | 65 | 93 |
| 2022 Maio | 32 | 45 | 77 |
| 2022 Abril | 41 | 50 | 91 |
| 2022 Março | 42 | 70 | 112 |
| 2022 Fevereiro | 23 | 31 | 54 |
| 2022 Janeiro | 40 | 74 | 114 |
| 2021 Dezembro | 39 | 51 | 90 |
| 2021 Novembro | 28 | 46 | 74 |
| 2021 Outubro | 49 | 66 | 115 |
| 2021 Setembro | 24 | 43 | 67 |
| 2021 Agosto | 36 | 46 | 82 |
| 2021 Julho | 38 | 43 | 81 |
| 2021 Junho | 26 | 44 | 70 |
| 2021 Maio | 41 | 60 | 101 |
| 2021 Abril | 159 | 198 | 357 |
| 2021 Março | 53 | 50 | 103 |
| 2021 Fevereiro | 27 | 16 | 43 |
| 2021 Janeiro | 23 | 12 | 35 |
| 2020 Dezembro | 18 | 21 | 39 |
| 2020 Novembro | 15 | 10 | 25 |
| 2020 Outubro | 15 | 7 | 22 |
| 2020 Setembro | 11 | 10 | 21 |
| 2020 Agosto | 15 | 8 | 23 |
| 2020 Julho | 12 | 11 | 23 |
| 2020 Junho | 13 | 14 | 27 |
| 2020 Maio | 12 | 12 | 24 |
| 2020 Abril | 2 | 1 | 3 |
| 2020 Março | 8 | 1 | 9 |
| 2020 Fevereiro | 0 | 2 | 2 |
| 2020 Janeiro | 0 | 6 | 6 |
| 2019 Dezembro | 0 | 3 | 3 |
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