Angina bullosa hemorrhagica: report of 11 cases

Abstract

Angina bullosa hemorrhagica is a rare and benign disorder, usually localized in the subepithelial layer of the oral, pharyngeal and esophageal mucosa. The lesions are characterized by their sudden onset. They appear as a painless, tense, dark red and blood-filled blister in the mouth that rapidly expand and rupture spontaneously in 24-48 hours. The underlying etiopathology remains ill defined, although it may be a multifactorial phenomenon including diabetes, and steroid inhalers. The condition is not attributable to blood dyscrasias, nor other vesicular-bullous disorders. In this study, eleven patients with such blisters are described. Physical examination of the patients revealed a single blister with hemorrhagic content localized in the oral mucosa. Biopsy of the lesions showed sub epithelial blisters with a mild infiltrate. In general practice, dermatologists could face a blood-filled bullous lesion of the oral mucosa. Recognition is, therefore, of great importance for dermatologists.

Angina Bullosa Hemorrhagica: A Case Report

“…Otros nombres que ha recibido esta lesión han sido: estomatitis hemorrágica bullosa benigna y hemofiltenosis oral traumática o recurrente. 3 Clínicamente, la ABH se caracteriza por ser una lesión única o múltiple, generalmente asintomática (aunque en ocasiones llega a ser dolorosa), que aparece repentinamente, 3,4 cuyo color varía entre el rojo oscuro y el púrpura, la cual puede hacer ruptura y expandirse rápidamente en un término de 24 a 48 horas; 4 sin embargo, se han encontrado casos que reportan una duración de más de cuatro meses hasta años. 5 Se pueden presentar múltiples ampollas de dos a tres centímetros de diámetro, con ruptura espontánea y curación sin dejar cicatriz, aunque pueden ser recurrentes.…”

“…8 La zona más afectada es el paladar blando, 3 aunque en la literatura se han reportado casos en otras zonas menos frecuentes, como la mucosa bucal, el borde lateral de la lengua, 3 el pilar anterior de las fauces y los cartílagos aritenoides; 7 de hecho, también se ha localizado en la zona del piso de la boca y en la mucosa faríngea y esofágica, 2, 4 y se han reportado casos inusuales en mucosa queratinizada, como el paladar duro. 9 La ABH no está asociada a desórdenes sanguíneos o hemorrágicos; 1,3,4 su etiopatología permanece aún sin definir, y el factor relacionado más importante es el trauma local del tejido, 1,3,6 causado por el consumo de bebidas calientes, procedimientos dentales, maltrato de las mucosas, o como resultado de la anestesia local. 2 Otras condiciones, como la diabetes mellitus y los esteroides inhalados, el salbutamol y el bromuro de ipratropio appear in the literature as predisposing factors.…”

“…Other names to this lesion include benign bullosa hemorrhagic stomatitis and traumatic or recurrent oral hemophiltenosis. 3 Clinically, ABH is characterized by a single or multiple lesion, asymptomatic for their most part (but painful in some cases), appearing suddenly, 3,4 with color ranging from dark red to purple, which can break and expand rapidly in a term of 24 to 48 hours; 4 however, there are reports of cases lasting more than four months to years. 5 It may appear as multiple blisters of two to three centimeters in diameter that break spontaneously and heal leaving no scars, although may be recurrent.…”

“…6,7 It has no sex predilection, and the age it most frequently occurs is between 50 and 70 years; 3 however, it can occur in younger patients. 8 The most commonly affected area is the soft palate, 3 although the literature has reported cases in other less common areas such as the buccal mucosa, the lateral border of the tongue, 3 the anterior pillar of the jaws and the arytenoid cartilages; 7 in fact, it has also been located in the area of the floor of the mouth and in the pharyngeal and esophageal mucosa; 2,4 unusual cases have been reported in keratinized mucosa, such as the hard palate.…”

“…ABH is not associated with blood disorders or hemorrhagic conditions; 1,3,4 its pathogenesis remains unknown, and the most important related factor is local tissue trauma, 1,3,6 caused by the consumption of hot drinks, dental procedures, harm to mucous membranes, or as a result of local anesthesia. 2 Other conditions, such as diabetes mellitus and inhaled steroids, salbutamol and ipratropium bromide,…”