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Angina bullosa haemorrhagica: a systematic review and proposal for diagnostic criteria
U Ordioni1, M Hadj Saïd2, G Thiery3
1Department of Oral Surgery and Odontology, APHM, Timone Hospital, Marseille, France; Centre Massilien de la Face, Marseille, France.
Insights
Angina bullosa haemorrhagica (ABH) is poorly understood, with limited evidence. This review proposes diagnostic criteria to improve patient care for this condition.
Area of Science:
- Oral Medicine
- Dermatology
- Pathology
Background:
- Angina bullosa haemorrhagica (ABH) is a rare oral condition characterized by spontaneous blistering and hemorrhage.
- Limited high-quality evidence exists regarding its epidemiological and etiological characteristics.
Purpose of the Study:
- To critically review and synthesize existing literature on the epidemiological, etiological, clinical, histological, biological, and therapeutic aspects of ABH.
- To propose diagnostic criteria for ABH based on a systematic literature review.
Main Methods:
- Systematic literature search of PubMed, Science Direct, Web of Science, and Cochrane Library databases.
- Eligibility assessment according to PRISMA statement.
- Inclusion of 40 articles (1985-2016) describing 225 ABH cases.
Main Results:
- Mean patient age was 55.4 years, with a female predominance (M:F ratio 0.7).
- Palate was the most common site (66%); 82% had a triggering factor.
- Symptomatic treatment yielded favorable outcomes, but recurrences were frequent (62%).
Conclusions:
- ABH is characterized by a lack of robust scientific documentation and low-level evidence studies.
- No clear aetiopathogenic associations with general pathologies or treatments were identified.
- Proposed diagnostic criteria aim to enhance the management of ABH patients.
Abstract:
The aim of this study was to perform a critical review of published data on the epidemiological, aetiological, clinical, histological, biological, and therapeutic characteristics of patients with angina bullosa haemorrhagica (ABH). A literature search was conducted in the PubMed, Science Direct, Web of Science, and Cochrane Library databases. All publications fulfilling the selection criteria were included in the eligibility assessment according to the PRISMA statement. The full texts of 54 retrieved articles were screened. Forty articles published between 1985 and 2016 describing 225 cases of ABH were finally selected. The mean age of the patients was 55.4 years; the male to female ratio was 0.7. The predominant localization was the palate (66%). A third of patients had no medical history. When specified, a triggering event or promoting factor was frequently found (82%). Biological tests were normal. A biopsy was performed on 35% of the patients. Treatment was symptomatic with a favourable outcome. Recurrences were frequent (62%). In conclusion, ABH is poorly documented and only by studies of low-level evidence. This review did not allow any aetiopathogenic association to be made with a general pathology or treatment. On the basis of this systematic review of the literature, diagnostic criteria aiming to improve the care of patients presenting with ABH are proposed.
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