Is inhaled glucocorticoids the only culprit in angina bullosa hemorrhagica?

Abstract

Angina bullosa hemorrhagica (ABH) is an infrequent dermatosis characterized by acute onset of hemorrhagic bulla in the oral cavity. Clinical presentation of ABH may be quite worrisome, and clinicians often feel skeptical regarding their clinical diagnosis and lack confidence in managing this distinct entity. Indeed, ABH is a completely benign and self-limited disorder. The exact etiopathogenesis of ABH is still unknown. There have been reports in the literature addressing a central role for mechanical instability of the epithelial-connective tissue connection in the pathogenesis of ABH. Moreover, it has been claimed that long-term usage of inhaled glucocorticoids (GCs) is involved in the development of ABH, since most of the reported cases are asthmatic patients, who were treated with inhaled GCs, and GCs are well known for their degradative activities on collagen formation. Here, we describe a case with ABH, who had a drug history of inhaled GCs and nonsteroidal anti-inflammatory drugs (NSAIDs). We assume that our case not only supports the association of inhaled GCs with ABH but also suggests a possible role for NSAIDs in the pathogenesis of ABH.

Recurrent episodes of angina bullosa hemorrhagica of the tongue

“…Several causative factors have been suggested like trauma due to sharp cusps, hard/course food, trauma during treatment procedures such as dental procedures, endoscopy, local anesthesia, genetic predisposition with increased mucosal vascular fragility, shouting/sneezing, long-term use of corticosteroid inhalers as in asthmatics, systemic diseases such as diabetes-mellitus, hypertension, chronic renal failure, rheumatoid arthritis, gastrointestinal disturbances, hyperuricemia and localized amyloidosis. [ 3 4 5 6 7 ] An imbalance in glucose metabolism as in the familial occurrence of diabetes mellitus has been linked with the development of ABH though the mechanism is not clear. [ 3 ] The use of corticosteroids for more than 5 years is said to cause changes in the collagen, a decrease in elastic fibers and epithelium atrophy resulting in weakened epithelial-connective tissue junction or a weak anchorage of mucosal vessels.…”

“…[ 3 4 ] Some authors report female preponderance with episodes during premenstruation. [ 3 6 ] ABH is not common in children <10 years. [ 3 ] There may be a single episode or recurrent episodes as seen in the present case.…”

“…Few authors have reported tongue involvement. [ 4 5 6 ] The gingiva and lips are less commonly affected. [ 3 ] It is believed that during mastication there is increased blood flow to the soft palate via parasympathetic reflex vasodilatation which might cause ABH more frequently in the soft palate.…”

Variations of oral anatomy and common oral lesions

“…It can manifest itself in four different forms: ulcerative (it is the most common and appears as an ulcerated and erythematous lesion on the mucosa), hemorrhagic (bleeding, mainly in the gingiva), hyperkeratotic (the least common form, occurring in cases of renal failure of long-standing) and erythemato-pultaceous (pseudomembrane formation, usually in patients with controlled renal disease). 36 , 37 It may mimic oral hairy leukoplakia. The diagnosis is mainly based on clinical history, oral examination, and laboratory tests.…”