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• W2109797797 abstract "A 50-year-old Hispanic man presented with a 1-year history of recurrent, painful, edematous plaques involving the head, trunk, and extremities that vesiculated and then resolved without scarring over the course of two weeks. These manifestations were associated with fever and malaise and occurred up to four times per month. The patient denied photosensitivity, mucositis, the use of new oral medications, and occupational chemical exposure. Prior treatment with antibiotics had been of no benefit, but improvement was noted after short courses of oral corticosteroids. The patient was taking no other medications. Physical examination revealed discrete and confluent, edematous, smooth, erythematous plaques on the right distal forearm with numerous overlying tense vesicles and bullae (Fig. 1). Laboratory investigation was notable for leukocytosis with absolute neutrophilia of 14,500 cells/μl (normal range: 1700–7000 cells/μl). Connective tissue serologies, serum and urine porphyrins, blood and tissue cultures, and perilesional direct immunofluorescence testing were negative. Hepatitis C antibodies were positive with an undetectable viral load. Histopathologic examination of a shave biopsy showed marked reticular degeneration of the epidermis with extensive vesiculation, severe papillary dermal edema, and a diffuse dermal infiltrate of neutrophils without vasculitis (Fig. 2). Bullous Sweet's syndrome. Acute febrile neutrophilic dermatosis, or Sweet's syndrome (SS), is distinguished by acute eruptive lesions involving the face, torso, and upper extremities associated with fever and peripheral neutrophilia.1 The classic presenting morphology is of variably agminated red papules that coalesce into well-defined, smooth, brightly erythematous plaques. Atypical morphologies include ill-defined nodules with subcutaneous involvement and panniculitis, superficial ulcers localized to the dorsal hands resembling pyoderma gangrenosum, and bullous lesions.2 Bullous SS is uncommon. It clinically resembles acute contact dermatitis, autoimmune bullous disease, bullous fixed drug eruption, bullous cellulitis, acute porphyria, and other photo-exacerbated dermatoses.3 Its histopathology, however, is similar to that of conventional cases in that it shows a diffuse neutrophilic dermal infiltrate with papillary dermal edema. Leukoclastic vasculitis may be present focally. In bullous presentations, there is extensive neutrophilic exocytosis and extreme dermal edema which produces both the intraepidermal and subepidermal vesiculation seen clinically. Sweet's syndrome is conventionally classified as idiopathic, malignancy-associated, or drug-induced, but it also occurs with infection, connective tissue disease, inflammatory bowel disease, pregnancy, and trauma. Most presentations are acute solitary events that reflect self-limiting upper respiratory tract infections and show a female predominance. Recurrent presentations are less common and should raise diagnostic suspicion of a drug-associated cause, malignancy, or chronic infectious etiology.2, 4 An increased prevalence of bullous lesions in hematologic and solid malignancy-associated SS has been reported, although workup with bone marrow biopsy and positron emission tomography (PET) and computed tomography (CT) was negative in this case.3 The pathogenesis of SS is thought to involve a hypersensitivity reaction with cytokine-induced neutrophil activation to a systemic antigen.4 Rarely, hepatitis C has been reported to trigger both classic and localized SS,4, 5 and thus we postulate variable viremia may account for the relapsing and episodic presentation seen in this patient. Conventional cases of SS respond promptly to oral corticosteroids and are frequently self-limiting. Idiopathic recurrent cases can be a treatment challenge. In these circumstances, anti-neutrophilic agents such as dapsone, colchicine, and potassium iodide (KI) can be used to minimize the number of symptomatic outbreaks, and oral corticosteroids can be used to break recurrence.6 Clearance in the present patient was achieved with 10 drops of saturated solution KI and colchicine 0.6 mg, both administered three times per day. Although it is generally well tolerated, the long-term administration of KI can induce hypothyroidism which necessitates the periodic monitoring of thyroid function." @default.
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• W2109797797 date "2015-08-12" @default.
• W2109797797 modified "2023-09-26" @default.
• W2109797797 title "A recurrent vesiculobullous eruption on the head, trunk, and extremities" @default.
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• W2109797797 doi "https://doi.org/10.1111/ijd.13001" @default.
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