Bullous Pemphigoid Triggered by Thermal Burn Under Medication With a Dipeptidyl Peptidase-IV Inhibitor: A Case Report and Review of the Literature

Abstract

Bullous pemphigoid (BP) is a common autoimmune blistering disease in which autoantibodies mainly target the hemidesmosomal component BP180 (also known as type XVII collagen) in basal keratinocytes. Various triggering factors are known to induce BP onset, including radiotherapy, burns, ultraviolet exposure, surgery, and the use of dipeptidyl peptidase-IV inhibitors (DPP4i), which are widely used antihyperglycemic drugs. Here, we present a case of BP triggered by a thermal burn under medication with DPP4i. A 60-year-old man with type II diabetes had been treated with the DPP4i linagliptin for 1 year. After the right forearm experienced a thermal burn, blisters developed around the burned area and gradually spread over the whole body with the production of autoantibodies targeting the non-NC16A domain of BP180. The diagnosis of BP was confirmed by immunohistopathological examination. Upon withdrawal of linagliptin and treatment with topical steroid and minocycline, complete remission was achieved after 4 months. Previously, 13 cases of BP that developed after thermal burns have been reported, and our case shared some of the clinical features of these thermal burn-induced BP cases. Interestingly, the present case also showed the typical clinical, histopathological, and immunological features of the non-inflammatory type of DPP4i-associated BP (DPP4i-BP). Although the pathogenesis of BP remains uncertain, the present case suggests that DPP4i may trigger the onset of BP similarly to a thermal burn. In addition, the clinical and histopathological features of DPP4i-BP may be distinct from other types of BP.

Bullous pemphigoid developed on nummular eczema lesions

“…1,2 Its location in the hand is infrequent (only six cases described so far), but unlike other locations, where it is usually an incidental histological finding, PD of the palm typically presents as hyperpigmented macules. [1][2][3] We present four additional cases corroborating that this entity has a well-defined clinical-dermoscopic pattern. Four women aged 25, 28, 39 and 64 years were assessed.…”

“…To date, there have been several reports focusing on the prodrome of BP. [1][2][3] The precipitating factors of BP are ultraviolet light, radiation therapy, thermal burn, surgical wounds, surgical grafts, ostomy and several traumatic triggers (Koebner phenomenon). 4 In addition, BP is occasionally preceded by non-specific lesions such as erythema multiforme and urticarial or eczematous rash with weeks' or months' duration.…”

Bullous Pemphigoid: Trigger and Predisposing Factors

“…As for diagnosis, we examined DIF results of 229 DIBP patients discussed in two retrospective case-control studies and in 42 case reports, and found 186 DIF positive patients, 15 DIF negative, and 28 with an unclear DIF result. Although in the literature some reviews and retrospective case-control studies do not always report DIF results, in the vast majority of DIBP patients, the diagnosis was obtained through the gold standard approach: DIF [ 9 , 10 , 11 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 ].…”

Radiation-Associated Pemphigus Vulgaris in a Patient With Preceding Malignancy: Treatment With Rituximab as a Valuable Option

“…Another autoimmune skin blistering disorder that is pathogenetically often connected to malignancy or RT is bullous pemphigoid (BP). The so far published cases comprise similarly to RT-associated pemphigus more than 30 (42), including a stage IV melanoma patient with dual exposure to PD-1 checkpoint inhibition and RT therapy (43). Nguyen et al have summarized 29 patients with BP following radiation for malignancy treatment.…”