Oral ulcerations after placement of orthodontic braces and skin pustules after laser hair removal: novel inducers of pathergy reactions in new-onset Behçet's disease

Abstract

A 25-year-old man was admitted to our hospital, with fever, severe ulceration of the oral and pharyngeal mucosa, and pustules on his skin. These pathergy-like lesions had emerged after placement of orthodontic braces and laser therapy for hair removal, respectively. The patient's clinical condition, together with his ethnic background, pointed to the diagnosis of Behçet's disease. Treatment with colchicine and prednisone resulted in rapid improvement of his symptoms. Pathergy reactions in response to placement of dental braces and laser hair removal are a rare first presentation of Behçet's disease.

Pathergy Phenomenon

“…Angiographic interventions or vascular surgery may lead to arterial thrombus or aneurysm, venipuncture may induce superficial thrombophlebitis, segmental bowel resection may trigger intestinal ulcers (39)(40)(41)(42). Other examples of pathergy phenomenon are, appearance of oral ulcers following dental therapuetic interventions and placement of othodontic braces and also pustular lesions after laser hair removal (43,44). These findings support the notion that hyperinflammatory response triggered by trauma is a feature of the disease itself, rather than being an organ-specific phenomenon and investigating SPT sites, may help in understanding inflammatory pathways involved in etiopathogenesis of BD.…”

“…The hypothesis on the role of microbial antigens as triggers is supported by decline in response when skin is surgically cleansed by 100% chlorhexidine or povidone iodide. Also exacerbation of oral aphthae, seen following dental invasive treatments can be due to oral microorganisms ( 44 ). Increased response to self saliva containing various microorganisms and significantly higher sensitivity with pneumococcal vaccine also support the notion of microbial elements as the inducers of SPR ( 21 ).…”

Behçet’s syndrome: focus on pathogenetic background, clinical phenotypes and specific treatments

“…Stress and fatigue have been independently reported by a French and a Turkish study as a common self-triggering factor for most BS patients [20,21]. Different foods have been correlated with the occurrence of oral ulcers (OU), possibly through an histamine-related irritative mechanism [20,21], together with oral and skin trauma [20,[22][23][24]. On the other hand, smoking habit seems to be protective for OU development [25,26].…”

“…Among the infectious agents, bacteria (and Streptococci in particular) have been suggested as triggering factors in the pathogenesis of BS, mainly for mucocutaneous manifestations [22,24,30,31].…”

A Case Report of Pathergy-Like Reaction in a Patient with Behçet's Disease Triggered by Wax Epilation

“…Pathergy test is usually performed on the flexor forearm by at least two intradermal punctures with a 20~22 gauge needle, and the test is considered positive if an erythematous papule or pustule is seen at the puncture site after 24~48 hours. Although it is considered as hyper-reactivity of the skin to intradermal prick, papulopustular lesions after laser epilation and oral aphthous lesions developing right after orthodontic treatment were also regarded as a manifestation of pathergy reaction 2 . Here we present a patient with diffuse papulopustular lesions after wax epilation, a pathergy-like reaction which has not been reported previously in the literature.…”