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World J Stomatol. Feb 20, 2015; 4(1): 1-7
Published online Feb 20, 2015. doi: 10.5321/wjs.v4.i1.1
Angina bullosa hemorrhagica an enigmatic oral disease
Javier Alberdi-Navarro, María Luisa Gainza-Cirauqui, María Prieto-Elías, José Manuel Aguirre-Urizar
Javier Alberdi-Navarro, María Luisa Gainza-Cirauqui, María Prieto-Elías, José Manuel Aguirre-Urizar, Oral Medicine and Oral and Maxillofacial Pathology Units, Dental Clinic Service, Master in Oral Pathology, Department of Stomatology II, UFI 11/25, University of the Basque Country/EHU, 48950 Leioa, Spain
María Luisa Gainza-Cirauqui, Department of Dental Surgery, Faculty of Dental Surgery, University of Malta, 2080 Msida, Malta
Author contributions: Alberdi-Navarro J and Aguirre-Urizar JM designed study; Alberdi-Navarro J, Gainza-Cirauqui ML and Prieto-Elías M contributed to bibliographic research; Alberdi-Navarro J and Prieto-Elías M drafted paper; Gainza-Cirauqui ML and Aguirre-Urizar JM contributed to critical review.
Conflict-of-interest: The authors have no conflict of interest to declare.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: José Manuel Aguirre-Urizar, PhD, Oral Medicine and Oral and Maxillofacial Pathology Units, Dental Clinic Service, Master in Oral Pathology, Department of Stomatology II, UFI 11/25, University of the Basque Country/EHU, Barrio Sarrera s/n, 48950 Leioa, Spain. josemanuel.aguirre@ehu.es
Telephone: +34-94-6015711
Received: October 15, 2014
Peer-review started: October 20, 2014
First decision: December 17, 2014
Revised: December 28, 2014
Accepted: January 9, 2015
Article in press: January 12, 2015
Published online: February 20, 2015
Processing time: 138 Days and 22 Hours
Abstract

Angina bullosa hemorrhagica (ABH) is an enigmatic oral disorder described for the first time by Badham in 1967 to define blisters with a hematic content in the oral cavity and oropharynx unrelated to any hematological, dermatological or systemic disease. The ABH is an uncommon disease of the oral cavity distinctively affecting adults, with the highest incidence over the 5th decade of life. This process is considered nowadays to have a multifactorial etiopathogenesis, where mild oral traumatisms can trigger the blisters in susceptible individuals. Certain association on the onset of the lesion with the chronic use of inhaled steroids and, more controversially, with triggering systemic disorders, such as, diabetes or hypertension has been described. Characteristically, the ABH blisters are acute and are located on the lining mucosa, more frequently on the soft palate. Usually, the lesions are solitary and rupture easily, resulting in a superficial ulceration that heals quickly without scarring. The histopathological analysis shows a subepithelial blister containing blood and direct immunofluorescence on the epithelium is negative. The differential diagnosis should consider all oral vesiculo-bullous disorders with hematic content, including mucocutaneos, hematological or cystic pathology. The diagnosis of ABH is clearly clinical, although the biopsy might be helpful on atypical or abnormally recurrent cases. The general prognosis of ABH is good and the treatment is symptomatic.

Keywords: Angina; Bullosa; Hemorrhagica; Traumatic; Blister

Core tip: Although it is an uncommon disease, the angina bullosa hemorrhagica should be considered in the differential diagnosis of oral vesiculo-bullous processes. Acknowledging this entity will help in differentiating it from important mucocutaneous and hematological diseases such as pemphigus vulgaris, mucous membrane pemphigoid or coagulation disorders. In this review we analyze the main etiopathogenic, clinicopathological, diagnostic and therapeutic aspects of this enigmatic oral condition.