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Band&#44; 21&#37; Seg&#44; 9&#37; Lym&#44; 7&#37; Mono&#44; and 57&#37; Blast&#44; decreased levels of hemoglobin &#40;8&#46;6&#160;g&#47;dL&#41; and platelets &#40;108&#44;000&#47;&#956;L&#41;&#44; and increased levels of C-reactive protein &#40;26&#46;02&#160;mg&#47;dL&#41;&#46; Blood culture was sterile&#46; A biopsy specimen from the peripheral edge of the bulla on the neck showed a subepidermal bulla and dense neutrophilic infiltration in the upper to mid-dermis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>a&#44; b&#41;&#46; Immature myeloid cell infiltration was not detected&#46; Infiltration of histiocytes&#47;histiocytoid cells was not observed&#44; and immunohistological examination showed scattered CD68- and MPO-positive cells which were not prominent&#46; Additionally&#44; another biopsy taken from the lower leg showed infiltration of inflammatory cells&#44; mainly lymphocytes&#44; around the vessels in the septa of the subcutaneous fat tissue &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>c&#41;&#46; Hematological examination and bone marrow biopsy revealed transformation from MDS into acute myelocytic leukemia&#44; and chemotherapy including prednisolone was immediately started in the hematology department&#46; After about one week&#44; all of the bullous lesions were re-epithelialized&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">The present case developed a sudden onset of skin lesions&#44; which histopathologically showed a predominant neutrophilic infiltration without leukocytoclastic vasculitis&#46; In addition&#44; fever&#44; elevated C-reactive protein&#44; and rapid response to systemic steroids were observed&#46; White blood cell count was within the upper limit of the normal range but two times higher than the usual value&#46; Thus&#44; the patient fulfilled the criteria for Sweet syndrome &#40;SS&#41;&#46; Cutaneous lesions of SS associated with myelodysplasia tend to be vesicular&#44; bullous&#44; or erosive&#46; The present case developed bullous SS&#44; simultaneously with the blastic crisis of MDS&#46; Of interest&#44; the lesions showed a peculiar distribution of well-circumscribed erythema in the V-neck area not protected by clothes during the season when the dose of ultraviolet radiation is increased in Japan&#44; which was considered to be a photo-Koebner phenomenon&#46; Only several cases of photo-induced or photo-distributed SS have been reported&#44; in which both ultraviolet A and ultraviolet B were candidates for induction of skin lesions&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> Although phototesting was not performed in the present case&#44; the patient had no previous history of photosensitive dermatitis or of taking drugs that can induce photosensitive eruptions&#46; To our knowledge&#44; the current report is the first case of photolocalized bullous SS&#46; The direct action of ultraviolet is speculated to activate and recruit neutrophils via interleukin-8&#44; tumor necrosis factor-&#945;&#44; E-selectin&#44; interleukin-1&#945;&#44; and G-CSF<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a> as well as ultraviolet-induced local immunosuppression&#46; Finally&#44; the patient concurrently developed infiltrative erythematous nodules on the face and extremities&#46; Biopsy showed septal panniculitis with lymphocytic infiltration without neutrophil infiltration in either dermis or subcutis&#59; however&#44; all of the concurrent lesions were considered to be part of the same spectrum&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0015" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#39; contributions</span><p id="par0020" class="elsevierStylePara elsevierViewall">Mai Endo&#58; Designed the study&#59; performed the research and contributed to analysis and interpretation of data&#59; wrote the initial draft of the manuscript&#59; read and approved the final version of the manuscript&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Miyuki Yamamoto&#58; Performed the research and contributed to analysis and interpretation of data&#59; read and approved the final version of the manuscript&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Mikio Ohtsuka&#58; Performed the research and contributed to analysis and interpretation of data&#59; read and approved the final version of the manuscript&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Toshiyuki Yamamoto&#58; Designed the study&#59; assisted in the preparation of the manuscript&#59; read and approved the final version of the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0040" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Case Letter
Bullous Sweet syndrome with a unique photodistributed pattern
Mai Endo
Autor para correspondência
enmai04@fmu.ac.jp

Corresponding author.
, Miyuki Yamamoto, Mikio Ohtsuka, Toshiyuki Yamamoto
Department of Dermatology, Fukushima Medical University, Fukushima, Japan
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 54-year-old female was admitted to our department&#44; complaining of a 4-day history of up to 40&#46;3&#160;&#176;C fever and cutaneous eruptions on the neck&#46; The patient had been suffering from myelodysplastic syndrome &#40;MDS&#41;&#44; for which no drugs&#44; including granulocyte-colony stimulating factor &#40;G-CSF&#41;&#44; were administered&#46; Physical examination showed numerous concrescences of blisters clearly localized in the V-neck area &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>a&#44; b&#41;&#46; Painful erythematous nodules were scattered on the cheeks&#44; but the tip of the nose was spared&#46; Furthermore&#44; there was one blister on the right forearm as well as painful erythematous nodules on the upper and lower extremities&#46; She did not have any mucosal lesions or a prior history of recurrent oral aphthae&#46; Laboratory examination showed a white blood cell count of 8&#44;100&#47;&#956;L with 3&#37; Band&#44; 21&#37; Seg&#44; 9&#37; Lym&#44; 7&#37; Mono&#44; and 57&#37; Blast&#44; decreased levels of hemoglobin &#40;8&#46;6&#160;g&#47;dL&#41; and platelets &#40;108&#44;000&#47;&#956;L&#41;&#44; and increased levels of C-reactive protein &#40;26&#46;02&#160;mg&#47;dL&#41;&#46; Blood culture was sterile&#46; A biopsy specimen from the peripheral edge of the bulla on the neck showed a subepidermal bulla and dense neutrophilic infiltration in the upper to mid-dermis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>a&#44; b&#41;&#46; Immature myeloid cell infiltration was not detected&#46; Infiltration of histiocytes&#47;histiocytoid cells was not observed&#44; and immunohistological examination showed scattered CD68- and MPO-positive cells which were not prominent&#46; Additionally&#44; another biopsy taken from the lower leg showed infiltration of inflammatory cells&#44; mainly lymphocytes&#44; around the vessels in the septa of the subcutaneous fat tissue &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>c&#41;&#46; Hematological examination and bone marrow biopsy revealed transformation from MDS into acute myelocytic leukemia&#44; and chemotherapy including prednisolone was immediately started in the hematology department&#46; After about one week&#44; all of the bullous lesions were re-epithelialized&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">The present case developed a sudden onset of skin lesions&#44; which histopathologically showed a predominant neutrophilic infiltration without leukocytoclastic vasculitis&#46; In addition&#44; fever&#44; elevated C-reactive protein&#44; and rapid response to systemic steroids were observed&#46; White blood cell count was within the upper limit of the normal range but two times higher than the usual value&#46; Thus&#44; the patient fulfilled the criteria for Sweet syndrome &#40;SS&#41;&#46; Cutaneous lesions of SS associated with myelodysplasia tend to be vesicular&#44; bullous&#44; or erosive&#46; The present case developed bullous SS&#44; simultaneously with the blastic crisis of MDS&#46; Of interest&#44; the lesions showed a peculiar distribution of well-circumscribed erythema in the V-neck area not protected by clothes during the season when the dose of ultraviolet radiation is increased in Japan&#44; which was considered to be a photo-Koebner phenomenon&#46; Only several cases of photo-induced or photo-distributed SS have been reported&#44; in which both ultraviolet A and ultraviolet B were candidates for induction of skin lesions&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> Although phototesting was not performed in the present case&#44; the patient had no previous history of photosensitive dermatitis or of taking drugs that can induce photosensitive eruptions&#46; To our knowledge&#44; the current report is the first case of photolocalized bullous SS&#46; The direct action of ultraviolet is speculated to activate and recruit neutrophils via interleukin-8&#44; tumor necrosis factor-&#945;&#44; E-selectin&#44; interleukin-1&#945;&#44; and G-CSF<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a> as well as ultraviolet-induced local immunosuppression&#46; Finally&#44; the patient concurrently developed infiltrative erythematous nodules on the face and extremities&#46; Biopsy showed septal panniculitis with lymphocytic infiltration without neutrophil infiltration in either dermis or subcutis&#59; however&#44; all of the concurrent lesions were considered to be part of the same spectrum&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0015" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#39; contributions</span><p id="par0020" class="elsevierStylePara elsevierViewall">Mai Endo&#58; Designed the study&#59; performed the research and contributed to analysis and interpretation of data&#59; wrote the initial draft of the manuscript&#59; read and approved the final version of the manuscript&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Miyuki Yamamoto&#58; Performed the research and contributed to analysis and interpretation of data&#59; read and approved the final version of the manuscript&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Mikio Ohtsuka&#58; Performed the research and contributed to analysis and interpretation of data&#59; read and approved the final version of the manuscript&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Toshiyuki Yamamoto&#58; Designed the study&#59; assisted in the preparation of the manuscript&#59; read and approved the final version of the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0040" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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