Bullous Variant of Sweet’s Syndrome as a Consequence of Radioiodine Contrast Exposure

Abstract

Bullous variant of Sweet’s syndrome (SS) is a rare form of SS, which clinically presents as bullous hemorrhagic rash and demonstrates dermal neutrophilic infiltrates with segregation of dermo-epidermal junction histopathologically. We present a case of a 73-year-old patient, who initially developed a hypersensitivity reaction on exposure to a radiocontrast agent and subsequently developed blistering rashes, which were established to be from bullous SS after exclusion of other possible diagnoses. Contrast media are utilized commonly in the current era of medicine and SS is rarely identified as an adverse event from it. Bullous variant particularly presents aggressively, which when recognized early responds to steroid use with clinical recovery.

Acute onset of bullous skin rash and acute kidney injury after exposure to radiocontrast: Sweet's syndrome

“…Last, exposure to stimuli causes an immune reaction to drugs and results in a cytokine cascade resulting in SS that responds to corticosteroids [3,6] . To date, only three other cases with similar presentations of radiocontrast-induced SS have been reported; these are summarized in Table 1 [7][8][9] . Treatment for drug-induced SS consists of the administration of topical or systemic corticosteroids together with withdrawal of the drug.…”

“…Alper et al, 2008 [7] Fok et al, 2014 [8] Bhat et al, 2018 [9] Abdelnabi potassium iodide may be utilized. Recurrence is uncommon in drug-induced SS after discontinuation of the medication [4] .…”

Bullous Sweet syndrome as a presentation of chronic myelogenous leukaemia

“…Among the above-mentioned cases of CML and Sweet syndrome, two showed the causative factor to be drugs. The rare case of the bullous type of Sweet syndrome was illustrated in a case report in which initially, after being exposed to a radiocontrast agent, the patient had a hypersensitive reaction and blistering rashes which were established to be from bullous Sweet syndrome 8. Another rare case of subcutaneous Sweet syndrome in a patient with CML published in 2014 described a patient who had been under treatment with imatinib for diagnosed CML since 2009 and developed characteristic skin lesions while on supportive treatment in the hospital.…”

Bullous hemorrhagic Sweet syndrome with cryptococcoid neutrophils in patients positive for antineutrophil cytoplasmic antibody without primary vasculitis

“…The bullous variant of SS presents with hemorrhagic bullae and splitting of the dermoepidermal junction. 3 , 4 , 5 The marked activation of the neutrophils and subsequent tissue destruction may be related to the expression of CD3, CD163, tumor necrosis factor-α, interleukin (IL) 8, IL-17, matrix metalloproteinase-9, and myeloperoxidase. 4 This extensive destruction by the neutrophils may be compounded in patients positive for ANCA.…”

“… 1 , 2 Atypical bullous or hemorrhagic presentations have been described in the literature, with the majority in a setting of hematologic malignancy. 3 , 4 , 5 , 6 , 7 Twenty percent of patients with SS have an underlying malignancy that can manifest with atypical bullae. 8 …”