Case and Research Letters
Kikuchi-Fujimoto Disease with Scalp Involvement
Enfermedad de Kikuchi-Fujimoto con compromiso de cuero cabelludo
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Durán-Vian, I. Vilanova-Udaniz, B. Castro-Gutierrez, M.A. González-López" "autores" => array:4 [ 0 => array:2 [ "nombre" => "C." "apellidos" => "Durán-Vian" ] 1 => array:2 [ "nombre" => "I." "apellidos" => "Vilanova-Udaniz" ] 2 => array:2 [ "nombre" => "B." 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Dense nodular dermal mononuclear cell rich infiltrate showing a significant number of plasma cells and numerous scattered S100 positive multinucleated histiocytes with marked emperipolesis and inconspicuous eosinophils.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "J.M. Conde, A.Y. Kim, R. de Miguel, C.H. Nousari" "autores" => array:4 [ 0 => array:2 [ "nombre" => "J.M." "apellidos" => "Conde" ] 1 => array:2 [ "nombre" => "A.Y." "apellidos" => "Kim" ] 2 => array:2 [ "nombre" => "R." "apellidos" => "de Miguel" ] 3 => array:2 [ "nombre" => "C.H." "apellidos" => "Nousari" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0001731017305999" "doi" => "10.1016/j.ad.2017.06.023" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0001731017305999?idApp=UINPBA000044" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219018301872?idApp=UINPBA000044" "url" => "/15782190/0000010900000007/v1_201809020417/S1578219018301872/v1_201809020417/en/main.assets" ] "en" => array:16 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letters</span>" "titulo" => "Kikuchi-Fujimoto Disease with Scalp Involvement" "tieneTextoCompleto" => true "saludo" => "<span class="elsevierStyleItalic">Dear Editor</span>," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "657" "paginaFinal" => "659" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "A. Combalia, X. Fustà-Novell, A. García-Herrera, J. Ferrando" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Combalia" "email" => array:1 [ 0 => "andreacombalia@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" => "X." "apellidos" => "Fustà-Novell" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "A." "apellidos" => "García-Herrera" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "J." "apellidos" => "Ferrando" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Unidad de Dermatología, Hospital Clinic de Barcelona, Barcelona, España" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Unidad de Patología, Hospital Clinic de Barcelona, Barcelona, España" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Enfermedad de Kikuchi-Fujimoto con compromiso de cuero cabelludo" ] ] "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" => 1303 "Ancho" => 1733 "Tamanyo" => 508813 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">a) The 3-cm-diameter erythematous plaque on the scalp. b) Haematoxylin and eosin staining showed mild vacuolar change in epidermal basal cells as well as perifollicular lymphohistiocytic infiltration and caryorrhexis in the reticular dermis (40x). c) The perifollicular lymphohistiocytic infiltration in haematoxylin and eosin staining (200x). d) Immunochemical analysis revealed that the lymphoid infiltrate was predominantly CD3+, with CD8 positive cells predominating over CD4.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We report the case of a 49-year-old Peruvian woman who presented with a 5-month history of cervical lymphadenopathy and neck pain. Social, family and medical past history were noncontributory. Physical examination revealed bilateral cervical lymphadenopathy, and a 3-cm-diameter erythematous plaque on her scalp (<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>a). Blood tests showed neutropenia (2.2 x 10<span class="elsevierStyleSup">9</span>/L) and positive serum antinuclear antibodies (titers 1:160). Computed tomographic scan (CTS) and 18-fluorodeoxyglucose positron emission tomography (PET) disclosed bilateral cervical lymphadenopathy with up to 2-cm enlarged lymph nodes.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Skin biopsy showed mild vacuolar change in epidermal basal cells as well as perifollicular lymphohistiocytic infiltration and caryorrhexis in the reticular dermis (<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>b, 1c). Immunochemical analysis revealed that the lymphoid infiltrate was predominantly CD3+, with CD8 positive cells predominating over CD4 (<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>d). By the other side CD68, CD163 and myeloperoxidase (MPO) immunostaining disclosed the presence of many histiocytes, and CD123 revealed that there were some plasmacytoid monocytes<span class="elsevierStyleBold">.</span> Neutrophils and eosinophils were absent, and no granulomas were evidenced. Vasculitis was not a feature. Lymph node biopsy showed paracortical hyperplasia, patchy necrosis with abundant cellular debris and profuse peripheral histiocytic cells. She was diagnosed with Kikuchi-Fujimoto disease (KFD) with cutaneous involvement.</p><p id="par0015" class="elsevierStylePara elsevierViewall">KFD, also known as histiocytic necrotizing lymphadenitis, was first described by Kikuchi<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> and Fujimoto<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> in 1972. It is a benign and self-limiting disorder characterized by lymphadenopathy associated with low-grade fever and flu-like symptoms. Unilateral and posterior cervical nodes are the commonest to be involved although it can present as generalized lymphadenopathy.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The female to male ratio is more than 4:1. The predominance of reports from Japan, and the fact that many of the patients reported in Europe and the USA have been of Asian descent, may point to a racial or genetic susceptibility.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Its etiology remains uncertain. A viral origin has long been suspected; however, the clinical course of the disease, the disappearance of lesions without any specific treatment, and some similarity with features of systemic lupus erythematosus (SLE) suggests the involvement of autoimmune mechanisms.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The diagnosis is confirmed by lymph node biopsy.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Involved lymph nodes characteristically demonstrate architecture partially effaced by confluent paracortical necrotic foci with abundant karyorrhectic debris, surrounded by CD68+ and MPO+ histiocytes, immunoblasts, CD8+ T-cells and CD123+ plasmacytoid dendritic cells.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> Neutrophils and eosinophils are absent. KFD has been classified into three histological subtypes, and is thought to progress from the proliferative type (expanded paracortex with an increase in histiocytes and plasmacytoid dendritic cells and karyorrhectic nuclear debris) to the necrotizing type (predominance of necrosis), and finally resolve into the xanthomatous type (predominance of foamy histiocytes).<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The skin is the most frequently affected extranodal organ, as cutaneous involvement has been reported in 16-40% of patients.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> Although some cases of KFD cutaneous lesions mimicking urticarial, morbilliform, rubella-like or drug-eruption-like rashes have been described, cutaneous KFD usually presents as erythematous papules and plaques, predominantly on the face, arms and upper neck. The histopathological findings of skin biopsies might mimic those of discoid lupus erythematosus; however, karyorrhectic debris without neutrophils, and the presence of CD68 and MPO positive cells are characteristic of KFD.<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> The skin lesions of KFD resolve after several weeks to months along with the resolution of lymphadenopathy.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Misdiagnosing KFD as lymphoma or SLE is not uncommon due to the similarity of clinical and histopathological features of these diseases. Identification of the characteristic histiocytes and abundant karyorrhectic debris instead of malignant lymphoma cells or SLE-pathognomonic haematoxylin bodies would provide the clue for the diagnosis.</p><p id="par0040" class="elsevierStylePara elsevierViewall">As the evidence suggests, it seems that individuals with KFD are more susceptible to SLE<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a>; in particular, KFD can precede, postdate or coincide with the diagnosis of SLE.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Although, the relationship between SLE and KFD remains a matter of debate, regular follow-up is required because SLE may develop several years after the onset of KFD.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">KDF is typically self-limiting, resolving within 1 to 4 months, though a possible recurrence rate of 3 to 4% has been reported. There is no specific treatment for KFD, although analgesics, antipyretics and nonsteroidal anti-inflammatory drugs may be used to alleviate lymph node tenderness and fever. The use of corticosteroids has been recommended in severe extranodal or generalized KFD,<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> and if symptoms persist, intravenous immunoglobulins can be prescribed.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Our patient presented with leukopenia and positive antinuclear antibodies, however, she did not fulfill the American College of Rheumatology criteria for the diagnosis of SLE. She was given oral ibuprofen daily for three months, lymphadenopathies gradually diminished and the scalp plaque disappeared. After one-year follow up, only few, small and non-specific lymph nodes remain in CTS.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Kikuchi-Fujimoto disease is rare. Clinicians should be aware of this condition as early diagnosis of the disease will avoid unnecessary medical tests and will lessen concerns of the patient and their relatives.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of Interest" ] 1 => array:1 [ "titulo" => "Bibliografía" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Combalia A, Fustà-Novell X, García-Herrera A, Ferrando J. Enfermedad de Kikuchi-Fujimoto con compromiso de cuero cabelludo. Actas Dermosifiliogr. 2018;109:657–659.</p>" ] ] "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1303 "Ancho" => 1733 "Tamanyo" => 508813 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">a) The 3-cm-diameter erythematous plaque on the scalp. b) Haematoxylin and eosin staining showed mild vacuolar change in epidermal basal cells as well as perifollicular lymphohistiocytic infiltration and caryorrhexis in the reticular dermis (40x). c) The perifollicular lymphohistiocytic infiltration in haematoxylin and eosin staining (200x). d) Immunochemical analysis revealed that the lymphoid infiltrate was predominantly CD3+, with CD8 positive cells predominating over CD4.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "Bibliografía" "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" => "Lymphadenitis showing focal reticulum cell hyperplasia with nuclear debris and phagocytosis: a clinicopathological study" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 4 | 4 | 8 |
| 2024 October | 74 | 42 | 116 |
| 2024 September | 74 | 31 | 105 |
| 2024 August | 109 | 62 | 171 |
| 2024 July | 92 | 43 | 135 |
| 2024 June | 105 | 56 | 161 |
| 2024 May | 67 | 43 | 110 |
| 2024 April | 88 | 39 | 127 |
| 2024 March | 56 | 31 | 87 |
| 2024 February | 59 | 33 | 92 |
| 2024 January | 52 | 31 | 83 |
| 2023 December | 54 | 30 | 84 |
| 2023 November | 68 | 24 | 92 |
| 2023 October | 79 | 30 | 109 |
| 2023 September | 66 | 35 | 101 |
| 2023 August | 40 | 15 | 55 |
| 2023 July | 41 | 28 | 69 |
| 2023 June | 38 | 23 | 61 |
| 2023 May | 53 | 21 | 74 |
| 2023 April | 33 | 25 | 58 |
| 2023 March | 48 | 25 | 73 |
| 2023 February | 59 | 28 | 87 |
| 2023 January | 42 | 24 | 66 |
| 2022 December | 44 | 50 | 94 |
| 2022 November | 30 | 20 | 50 |
| 2022 October | 29 | 18 | 47 |
| 2022 September | 39 | 23 | 62 |
| 2022 August | 27 | 34 | 61 |
| 2022 July | 31 | 42 | 73 |
| 2022 June | 21 | 26 | 47 |
| 2022 May | 42 | 34 | 76 |
| 2022 April | 56 | 32 | 88 |
| 2022 March | 70 | 47 | 117 |
| 2022 February | 74 | 28 | 102 |
| 2022 January | 34 | 39 | 73 |
| 2021 December | 42 | 40 | 82 |
| 2021 November | 43 | 49 | 92 |
| 2021 October | 48 | 46 | 94 |
| 2021 September | 35 | 34 | 69 |
| 2021 August | 37 | 36 | 73 |
| 2021 July | 38 | 31 | 69 |
| 2021 June | 40 | 27 | 67 |
| 2021 May | 45 | 44 | 89 |
| 2021 April | 71 | 85 | 156 |
| 2021 March | 61 | 21 | 82 |
| 2021 February | 76 | 29 | 105 |
| 2021 January | 29 | 19 | 48 |
| 2020 December | 30 | 18 | 48 |
| 2020 November | 37 | 23 | 60 |
| 2020 October | 24 | 31 | 55 |
| 2020 September | 19 | 18 | 37 |
| 2020 August | 30 | 21 | 51 |
| 2020 July | 14 | 16 | 30 |
| 2020 June | 30 | 36 | 66 |
| 2020 May | 20 | 27 | 47 |
| 2020 April | 20 | 10 | 30 |
| 2020 March | 18 | 14 | 32 |
| 2019 May | 0 | 1 | 1 |
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