Case and Research Letters
Cryptococcoid Sweet Syndrome: A Clinical and Histologic Imitator of Cryptococcosis
Síndrome de Sweet criptococoide: simulador de criptococosis tanto clínica como histológicamente
M.E. Mazzei
, A. Guerra, L. Dufrechou, M. Vola
Corresponding author
Cátedra de Dermatología, Hospital de Clínicas, Facultad de Medicina, Universidad de la República Oriental del Uruguay (UDELAR), Montevideo, Uruguay
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CD4+ lymphocytes on a skin biopsy of patient two (40x)<span class="elsevierStyleBold">. D.</span> CD8+ lymphocytes on a skin biopsy of patient two (40x). Note that in the dermal lymphocytic infiltrate, a CD8 / CD4 ratio of 2.5, less than 4 and more suggestive in this case of GHVD.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "A. Rodríguez Tejero, J. Badiola González, L. Roldán Mateo, A. Molina Leyva" "autores" => array:4 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "Rodríguez Tejero" ] 1 => array:2 [ "nombre" => "J." "apellidos" => "Badiola González" ] 2 => array:2 [ "nombre" => "L." "apellidos" => "Roldán Mateo" ] 3 => array:2 [ "nombre" => "A." 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(b) Infiltrative erythemas on the upper cheek. (c) A biopsy specimen showing epidermal atrophy, liquefaction of the basement membrane, dyskeratotic epidermal cells, and focal mononuclear cell infiltration in the dermis.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "M. Yamamoto, T. Yamamoto" "autores" => array:2 [ 0 => array:2 [ "nombre" => "M." "apellidos" => "Yamamoto" ] 1 => array:2 [ "nombre" => "T." "apellidos" => "Yamamoto" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1578219020304224?idApp=UINPBA000044" "url" => "/15782190/0000011200000001/v1_202101161046/S1578219020304224/v1_202101161046/en/main.assets" ] "en" => array:18 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Case and Research Letters</span>" "titulo" => "Cryptococcoid Sweet Syndrome: A Clinical and Histologic Imitator of Cryptococcosis" "tieneTextoCompleto" => true "saludo" => "To the Editor:" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "79" "paginaFinal" => "80" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "M.E. Mazzei, A. Guerra, L. Dufrechou, M. Vola" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M.E." "apellidos" => "Mazzei" "email" => array:1 [ 0 => "marumazzei@hotmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "A." "apellidos" => "Guerra" ] 2 => array:2 [ "nombre" => "L." "apellidos" => "Dufrechou" ] 3 => array:2 [ "nombre" => "M." "apellidos" => "Vola" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Cátedra de Dermatología, Hospital de Clínicas, Facultad de Medicina, Universidad de la República Oriental del Uruguay (UDELAR), Montevideo, Uruguay" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de Sweet criptococoide: simulador de criptococosis tanto clínica como histológicamente" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 629 "Ancho" => 750 "Tamanyo" => 110458 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Immune staining with myeloperoxidase (hematoxylin–eosin, ×400).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Cryptococcoid Sweet syndrome is a term coined recently by Wilson J et al<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> to describe a rare variety of Sweet syndrome, which has a histologic profile that is indistinguishable under hematoxylin and eosin staining from gelatinous cryptococcosis. Diagnosis is established by positivity for staining with myeloperoxidase in the presence of negative stains for fungi. To date, 8 cases of neutrophilic dermatosis have been reported with these histopathology characteristics, all of them in dermatopathology journals.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–4</span></a> We describe a new case of cryptococcoid Sweet syndrome, where infection by <span class="elsevierStyleItalic">Cryptococcus</span> spp was considered not only because of the histopathology profile but also because of the clinical characteristics of the skin lesions.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Case Description</span><p id="par0010" class="elsevierStylePara elsevierViewall">An 18-year-old woman presented a history of episodes of different neutrophilic dermatoses (pyoderma gangrenosum and Sweet syndrome) between the ages of 6 and 12 years. She subsequently developed antineutrophil cytoplasmic antibody (ANCA)-positive vasculitis, with terminal kidney failure and malignant hypertension, causing her to be put on hemodialysis. The patient was undergoing treatment with azathioprine, prednisone, losartan, valaciclovir, and trimethoprim–sulfamethoxazole. Forty-eight hours after undergoing magnetic resonance angiography, she presented molluscum-like lesions on the face (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) and blisters and erythematous lesions on the backs of the hands, accompanied by fever and neutrophilia of 9910 neutrophils/mm<span class="elsevierStyleSup">3</span> (82.5% leukocytes). An adverse reaction to the contrast medium was suspected and boluses of methylprednisone were administered; the patient was assessed by the dermatology department. An infectious process (cryptococcosis, tuberculosis, histoplasmosis, etc.) was suspected and biopsies were performed and microbiology samples were taken. The lesions resolved after a few days and the patient remained lesion-free and in excellent general health.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Histology revealed a dermal inflammatory infiltrate with neutrophils, vacuolated spaces, and yeast-like structures that appeared to have a capsule, all of which suggested gelatinous cryptococcosis (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). However, the lesions had regressed with corticosteroid treatment, the chest x-ray was normal, and cultures, special stains, and antigenemia for <span class="elsevierStyleItalic">Cryptococcus</span> spp were negative.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">In light of the history of neutrophilic dermatosis, the presence of fever and neutrophilia, resolution of symptoms with corticosteroid treatment, and negativity of the microbiology analyses, a diagnosis of cryptococcoid Sweet syndrome was reached. A positive stain for myeloperoxidase confirmed the diagnosis (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0025" class="elsevierStylePara elsevierViewall">Although the term <span class="elsevierStyleItalic">cryptococcoid Sweet syndrome</span> was coined in 2017 by Wilson et al, the first cases of this histologic phenomenon were described by Ko et al in 2013.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Those authors reported 3 cases of neutrophilic dermatosis, where the histologic profile showed a dermal inflammatory infiltrate with neutrophils, vacuolated spaces, and yeast-like basophilic structures that appeared to be encapsulated—a profile highly suggestive of disseminated cryptococcosis.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Microbiology studies and special stains did not confirm infection in any of the 3 cases. In 2 of the cases, electron microscopy revealed autolyzed neutrophils.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The cause of this phenomenon is still the subject of debate. Ko et al postulate that it may be due to autophagic programmed cell death that leads to extensive cytoplasmic vacuolization. Another possibility is that it is due to artefacts of laboratory technique. Finally, given that the 3 original patients were women over 75 years of age, the authors suggest that these phenomena may be linked to degenerative collagen alterations. This hypothesis, however, does not hold with patients of a young age, particularly our patient, who is the youngest of those reported to date (18 years).</p><p id="par0040" class="elsevierStylePara elsevierViewall">It should be noted that, to date, in all published cases, suspicion of cryptococcosis arises from a suggestive histologic profile; in no case was infection by <span class="elsevierStyleItalic">Cryptococcus</span> spp considered as a clinical diagnosis. We therefore consider cryptococcoid Sweet syndrome to be a diagnostic challenge, not only for pathologists but also for clinicians, and we highlight the idea that in all infectious processes, the definitive diagnosis must meet clinical, histopathologic, and microbiologic criteria.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Moreover, this case of cryptococcoid Sweet syndrome presented in association with an ANCA-positive vasculitis. The association between neutrophilic dermatosis and ANCA-positive vasculitis is well known, although the pathogenic link is not fully understood.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> In this context, it is important to note that histologic changes of this type have also been described in vasculitis and that entity should also be included in the differential diagnosis.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In conclusion, we present a new case of cryptococcoid Sweet syndrome, in which the differential diagnosis with disseminated cryptococcosis was due not only to the histologic profile but also to the clinical picture (sudden appearance of molluscum-like papules in an immunosuppressed patient). Thus, in the case of a clinical and histologic suspicion of disseminated cryptococcosis, we must consider Sweet syndrome in the differential diagnosis, particularly if the relevant microbiologic criteria are not met.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of Interest</span><p id="par0055" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Case Description" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of Interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-12-10" "fechaAceptado" => "2019-03-31" "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Mazzei ME, Guerra A, Dufrechou L, Vola M. Síndrome de Sweet criptococoide: simulador de criptococosis tanto clínica como histológicamente. Actas Dermosifiliogr. 2021;112:79–80.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 865 "Ancho" => 750 "Tamanyo" => 61741 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Molluscum-like lesion on the face.</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" => 633 "Ancho" => 750 "Tamanyo" => 149720 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Biopsy of molluscum-like lesion on the face showing vacuolated spaces, neutrophils, and yeast-like structures that appear to have a capsule, as is seen in gelatinous cryptococcosis (hematoxylin–eosin, ×400).</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" => 629 "Ancho" => 750 "Tamanyo" => 110458 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Immune staining with myeloperoxidase (hematoxylin–eosin, ×400).</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" => "Cryptococcoid Sweet´s syndrome: two reports of sweet´s syndrome mimicking cutaneous cryptococcosis" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "J. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 7 | 8 | 15 |
| 2024 October | 137 | 53 | 190 |
| 2024 September | 187 | 43 | 230 |
| 2024 August | 183 | 76 | 259 |
| 2024 July | 132 | 69 | 201 |
| 2024 June | 99 | 70 | 169 |
| 2024 May | 109 | 55 | 164 |
| 2024 April | 132 | 62 | 194 |
| 2024 March | 102 | 51 | 153 |
| 2024 February | 96 | 43 | 139 |
| 2024 January | 75 | 40 | 115 |
| 2023 December | 67 | 17 | 84 |
| 2023 November | 78 | 32 | 110 |
| 2023 October | 56 | 27 | 83 |
| 2023 September | 62 | 31 | 93 |
| 2023 August | 53 | 27 | 80 |
| 2023 July | 90 | 44 | 134 |
| 2023 June | 62 | 24 | 86 |
| 2023 May | 76 | 38 | 114 |
| 2023 April | 53 | 24 | 77 |
| 2023 March | 76 | 43 | 119 |
| 2023 February | 70 | 33 | 103 |
| 2023 January | 49 | 33 | 82 |
| 2022 December | 75 | 39 | 114 |
| 2022 November | 46 | 36 | 82 |
| 2022 October | 42 | 18 | 60 |
| 2022 September | 40 | 43 | 83 |
| 2022 August | 48 | 49 | 97 |
| 2022 July | 42 | 46 | 88 |
| 2022 June | 39 | 40 | 79 |
| 2022 May | 71 | 48 | 119 |
| 2022 April | 84 | 49 | 133 |
| 2022 March | 74 | 68 | 142 |
| 2022 February | 75 | 40 | 115 |
| 2022 January | 104 | 55 | 159 |
| 2021 December | 68 | 50 | 118 |
| 2021 November | 76 | 59 | 135 |
| 2021 October | 72 | 84 | 156 |
| 2021 September | 76 | 66 | 142 |
| 2021 August | 66 | 56 | 122 |
| 2021 July | 62 | 44 | 106 |
| 2021 June | 70 | 55 | 125 |
| 2021 May | 96 | 67 | 163 |
| 2021 April | 185 | 119 | 304 |
| 2021 March | 130 | 78 | 208 |
| 2021 February | 97 | 58 | 155 |
| 2021 January | 152 | 49 | 201 |
| 2020 December | 20 | 16 | 36 |
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