Angina Bullosa Haemorrhagica Of Buccal Mucosa; Case Report And Review Of The Literature

Background Angina bullosa haemorrhagica (ABH) clinically presents as acute, occasionally painful, benign submucosal, blood-filled vesicles and bullae. Rapid rupture of bulla complicates differential diagnosis with other oral lesions that cause post-bulla erosions. We report a case of recurrent ABH. Case Summary This 51-year-old Caucasian female presented for evaluation and management of oral “blisters”. She saw an oral surgeon who suspected oral lichen planus and performed a biopsy that revealed “hyperkeratosis with mild cellular atypia and chronic inflammation”. No definitive diagnosis was given or treatment rendered, so she went to her PCP, who then referred her to our service for diagnosis and management. She has been getting oral blisters after consuming salty/crunchy food. Lesions occur about once a month for the past 10 years. They generally occur in the buccal mucosa and floor of the mouth, are sometimes sensitive to certain foods after the blister pops and no interventions have been attempted. Her medical history is significant for hemochromatosis, basal cell carcinoma, and codeine allergy. She denied other bleeding disorders. All coagulation labs (available as she is being followed for hemochromatosis) were normal. She takes fluticasone as needed. Family history and social history were noncontributory. On the initial exam, multiple cherry angiomas were noted on her neck and back. No intraoral lesions were noted. However, she presented pictures of prior lesions which resembled hematomas. Differential diagnoses included vesiculobullous disease and ABH. She was instructed to return for a biopsy when new lesions presented. Biopsies (H&E and DIF) revealed “oral mucosa with mild chronic inflammation” and “no specific immunoreactants detected”. After excluding systemic conditions, bleeding disorders, and vesiculobullous disorders, a diagnosis of ABH was made. The patient was informed of the condition and instructed to avoid crunchy food. Conclusions The pathogenesis of ABH is unclear. However, trauma is reported as a major provoking factor, with lesions commonly present on palatal mucosa. Diagnosing ABH may be difficult as the clinical history and appearance are consistent with bleeding disorders, pemphigoid, oral lichen planus, and pemphigus. Clinicians should be aware of ABH presentation and diagnosis to ensure accurate diagnosis and avoid unnecessary treatments. Angina bullosa haemorrhagica (ABH) clinically presents as acute, occasionally painful, benign submucosal, blood-filled vesicles and bullae. Rapid rupture of bulla complicates differential diagnosis with other oral lesions that cause post-bulla erosions. We report a case of recurrent ABH. This 51-year-old Caucasian female presented for evaluation and management of oral “blisters”. She saw an oral surgeon who suspected oral lichen planus and performed a biopsy that revealed “hyperkeratosis with mild cellular atypia and chronic inflammation”. No definitive diagnosis was given or treatment rendered, so she went to her PCP, who then referred her to our service for diagnosis and management. She has been getting oral blisters after consuming salty/crunchy food. Lesions occur about once a month for the past 10 years. They generally occur in the buccal mucosa and floor of the mouth, are sometimes sensitive to certain foods after the blister pops and no interventions have been attempted. Her medical history is significant for hemochromatosis, basal cell carcinoma, and codeine allergy. She denied other bleeding disorders. All coagulation labs (available as she is being followed for hemochromatosis) were normal. She takes fluticasone as needed. Family history and social history were noncontributory. On the initial exam, multiple cherry angiomas were noted on her neck and back. No intraoral lesions were noted. However, she presented pictures of prior lesions which resembled hematomas. Differential diagnoses included vesiculobullous disease and ABH. She was instructed to return for a biopsy when new lesions presented. Biopsies (H&E and DIF) revealed “oral mucosa with mild chronic inflammation” and “no specific immunoreactants detected”. After excluding systemic conditions, bleeding disorders, and vesiculobullous disorders, a diagnosis of ABH was made. The patient was informed of the condition and instructed to avoid crunchy food. The pathogenesis of ABH is unclear. However, trauma is reported as a major provoking factor, with lesions commonly present on palatal mucosa. Diagnosing ABH may be difficult as the clinical history and appearance are consistent with bleeding disorders, pemphigoid, oral lichen planus, and pemphigus. Clinicians should be aware of ABH presentation and diagnosis to ensure accurate diagnosis and avoid unnecessary treatments.