Angina Bullosa Hemorrhagica: A Rare Case Involving the Ventral Tongue

ABSTRACT

Angina bullosa hemorrhagica (ABH) is a rare benign condition characterized by the sudden onset of hemorrhagic blisters in the oral mucosa, often triggered by minor trauma. We report a case of ABH in a 55‐year‐old woman with a history of asthma managed with inhaled corticosteroids who presented with a painful blister on the ventral tongue, an uncommon site. The lesion was associated with chewing trauma and resolved spontaneously within seven days without scarring. Laboratory investigations excluded systemic causes, and symptomatic treatment with benzydamine hydrochloride and 0.12% chlorhexidine mouthwashes provided relief from discomfort. This case underscores the clinical features, diagnostic approach, and management of ABH, particularly highlighting its rare localization to the ventral tongue.

Key Clinical Message

Angina bullosa hemorrhagica (ABH) is a rare, benign oral hemorrhagic blistering disorder. This case on the ventral tongue highlights mechanical trauma and inhaled corticosteroids as triggers. Rapid identification of ABH can prevent invasive diagnostic procedures and ensure effective conservative treatment, thereby minimizing patient anxiety and avoiding unnecessary biopsies or surgical interventions.

1. Introduction

Angina bullosa hemorrhagica (ABH), first described in 1933, is a rare, benign condition characterized by the sudden appearance of blood‐filled blisters on the oral or oropharyngeal mucosa. Badham coined the term ABH in 1967 [1]. It predominantly affects middle‐aged and older adults and is rare in children [2, 3, 4]. While the exact prevalence of angina bullosa hemorrhagica (ABH) remains unclear, a retrospective cross‐sectional study conducted in Brazil on patients with oral and maxillofacial lesions documented a prevalence rate of 0.18% among a total of 12,727 lesions [5].

Common triggers include mechanical trauma from chewing or dental procedures [6, 7], with inhaled corticosteroids [8] and systemic conditions such as diabetes as potential risk factors [9]. This case report describes ABH presenting on the ventral tongue, an uncommon location, highlighting its clinical presentation and management.

2. Case Presentation

A 55‐year‐old woman presented to our dental clinic complaining of a painful erosive lesion accompanied by a burning sensation on the ventral surface of the tongue. Through a thorough review of her medical history, clinical examination, and paraclinical tests, we identified several key diagnostic indicators:

2.1. History of a Preceding Hemorrhagic Blister

The patient described the lesion as having developed after dinner the previous evening. It began as a sudden, blood‐filled blister that ruptured spontaneously within a few hours, resulting in an erosive ulcer (Figures 1 and 2: intact blood‐filled blister; post‐rupture erosion). Notably, she had encountered two similar episodes at the same location during the past 6 months, each triggered by the mechanical trauma of chewing hard foods.

FIGURE 1.

Intact blood‐filled blister on the ventral tongue before rupture.

FIGURE 2.

Erosive lesion on the ventral tongue following spontaneous rupture of a blood‐filled blister.

2.2. Characteristic Clinical Course

On initial examination, we observed an intact, tense, dark red‐to‐purple blood‐filled blister. Following spontaneous rupture, a shallow, irregular erosion with a fibrinous base developed, healing completely without scarring within 7 days.

2.3. Lesion Site and Mucosal Type

The lesion was confined to the non‐keratinized mucosa of the ventral tongue—a vulnerable area prone to trauma‐induced blistering owing to its thin epithelium and exposure to high mechanical stresses during mastication.

2.4. Medical History

The patient had a documented history of asthma, managed with inhaled corticosteroids for 2 years until discontinuation 1 year prior; this prior therapy may have rendered the mucosa more fragile. She reported no hypertension, diabetes, systemic bleeding disorders, or use of any concurrent medications.

2.5. Paraclinical Evaluation

Laboratory results, including complete blood count (CBC), prothrombin time (PT), and partial thromboplastin time (PTT), fell within normal ranges, effectively ruling out thrombocytopenia, von Willebrand disease, or any coagulopathy. The absence of extraoral skin or mucosal lesions excluded pemphigus vulgaris, mucous membrane pemphigoid, and erythema multiforme. Furthermore, the lack of fever or lymphadenopathy helped dismiss infectious or malignant processes.

The spontaneous rupture of the blood‐filled blister precluded histopathologic examination. Nevertheless, we confidently diagnosed angina bullosa hemorrhagica (ABH) based on fulfillment of 7 out of 9 diagnostic criteria proposed by Ordioni et al. [10], supported by comprehensive clinical and historical findings, systematic exclusion of differential diagnoses, and the characteristic disease course (Table 1).

TABLE 1.

Diagnostic criteria for angina bullosa hemorrhagica by Ordioni et al. [10].

Main criteria

1. Clinically noticeable hemorrhagic bulla or erosion with a history of bleeding of the oral mucosa

2. Exclusively oral or oropharyngeal localization

Additional criteria

3. Palatal localization

4. Triggering event or promoting factor (food intake)

5. Recurrent lesions

6. Favorable evolution without leaving a scar in a few days

7. Painless lesion, tingling, or burning sensation

8. Normal platelet count and coagulation profile

9. Negative direct immunofluorescence

We initiated supportive therapy with benzydamine hydrochloride and 0.12% chlorhexidine mouthwashes to alleviate symptoms and prevent secondary infection. The patient's pain subsided within 3 days, and full epithelialization without scarring was achieved by day seven. At the one‐month follow‐up, no recurrence was noted.

3. Discussion

3.1. Etiology and Pathogenesis

ABH typically presents as subepithelial hemorrhagic bullae, frequently precipitated by mechanical trauma during chewing [6, 7]. In this patient, the prior chronic use of inhaled corticosteroids likely heightened mucosal fragility through impaired collagen synthesis [8]. Although systemic factors such as diabetes or hypertension have been linked to ABH in other reports [9], these were notably absent here.

3.2. Clinical Features

Patients with ABH commonly develop solitary or multiple hemorrhagic blisters, most often on the buccal mucosa or soft palate, with less frequent extension to the tongue, gingivobuccal sulcus, or lips [11]. The ventral tongue involvement in our case is particularly uncommon [12]. This site's non‐keratinized mucosa appears especially vulnerable to trauma due to reduced epithelial adhesion. Whereas ABH blisters are generally painless, our patient reported discomfort, attributable to the sensitive location. The blisters ruptured promptly within hours, yielding an ulcerated base that healed scar‐free in 7–14 days [13]—a pattern mirrored in this instance. The reported recurrence rates of 25%–100% align closely with the patient's prior episodes [14].

3.3. Diagnostic Approach

Differential considerations encompassed hematologic disorders (e.g., thrombocytopenia) and vesiculobullous diseases (e.g., pemphigus). However, normal laboratory findings and the lack of systemic signs clearly differentiated ABH. Histologically, ABH features subepithelial separation with nonspecific immunofluorescence, but biopsy was unnecessary due to the classic presentation [15]. While the clinical and laboratory findings strongly supported the diagnosis of ABH, the absence of histopathologic confirmation represents a limitation of this report.

3.4. Management and Prognosis

Our approach was conservatively supportive, prioritizing analgesia and antimicrobial prophylaxis. The benzydamine and chlorhexidine rinses proved highly effective. Overall, ABH carries an outstanding prognosis, with complete, scarless healing in 7–14 days [13]. We recommend ongoing surveillance to detect any potential recurrences.

This table (Table 2) summarizes the clinical features of angina bullosa hemorrhagica (ABH) in the reported case compared to findings in the literature.

TABLE 2.

Comparison of clinical features of ABH in this case and reported cases.

Feature	This case	Literature
Age/Sex	55/Female	Middle‐aged/elderly [4], no sex bias [2]
Location	Ventral tongue	Ventral [12]/lateral tongue [14]
Trigger	Chewing trauma	Trauma [4], inhaled corticosteroids [8]
Recurrence	Twice in 6 months	25–100% recurrence rate [14]
Healing Time	7 days, no scarring	7–14 days, no scarring [13]
Management	Benzydamine, chlorhexidine	Supportive (analgesics, antiseptics) [4]

4. Conclusion

This case highlights ABH as a benign, trauma‐induced condition with an excellent prognosis, notable for its rare ventral tongue presentation. Clinicians should consider ABH in patients with sudden‐onset oral hemorrhagic blisters, particularly with a history of trauma or corticosteroid use. Diagnostic evaluation should exclude hematologic and vesiculobullous disorders. Supportive treatment with benzydamine and chlorhexidine is effective, with lesions healing within 7–14 days.

Author Contributions

Fatemeh Khajehzadeh: conceptualization, data curation, formal analysis, funding acquisition, investigation, methodology, project administration, resources, software, supervision, validation, visualization, writing – original draft, writing – review and editing.

Funding

The author has nothing to report.

Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images.

Conflicts of Interest

The author declares no conflicts of interest.

Khajehzadeh F., “Angina Bullosa Hemorrhagica: A Rare Case Involving the Ventral Tongue,” Clinical Case Reports 14, no. 1 (2026): e71748, 10.1002/ccr3.71748.

Data Availability Statement

Data sharing does not apply to this article as no datasets were generated or analyzed during the current study.