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Angina Bullosa Haemorrhagica in COVID 19: A Diagnostic Conundrum. Case Report and Review of Literature. Indian J Otolaryngol Head Neck Surg 2023. [PMCID: PMC9979882 DOI: 10.1007/s12070-023-03584-w] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 03/06/2023] Open
Abstract
Oral manifestations of COVID-19 are amongst the most obscure and ill-reported. Of these, angina bullosa haemorrhagica is amongst the rarest. Only 2 cases of angina bullosa haemorrhagica in COVID-19 patients have been reported in literature. Angina bullosa haemorrhagica (ABH) is an enigmatic, abstruse condition represented by sudden onset of painful subepithelial, mucosal blood-filled vesicles and bullae in the oral cavity. It is not attributed to any systemic conditions, blood dyscracias or other well-known dermatological pathologies. The occurrence of these lesions in patients of COVID-19 suggests that the underlying pathology of the latter may predispose to ABH and thus help in shedding some light onto the pathogenesis of this obscure disease. Herein we present 2 cases of ABH in patients of COVID-19 within a few weeks of the resolution of the latter. Both patients reported that they had never had this condition before and that this was the first presentation of the symptom. A review of literature shows that the etiopathogenesis of ABH is ambiguous at best and that the pathology underlying the oral manifestation of COVID-19 may well be applicable to ABH as well. Various mechanisms have been proposed to cause oral manifestations in COVID-19 patients. These include imbalance in the RAS pathway causing mucosal disruption, immune dysregulation, deranged cellular immune mechanism and disruption of local immune mechanisms. Since ABH has been reported in COVID 19, it is plausible that some of the mechanisms underlying the pathogenesis of oral manifestations may explain the pathogenesis of ABH.
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Borovikov YE, Bukin YK. Angina bullosa hemorrhagica. VESTNIK DERMATOLOGII I VENEROLOGII 2019. [DOI: 10.25208/0042-4609-2019-95-2-50-54] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/02/2022] Open
Abstract
Angina bullosa hemorrhagica (ABH) is a term used to describe acute, subepithelial oral mucosal blisters filled with blood without any systemic disorder or hemostatic defect. The diagnosis of ABH is largely clinical, and includes elimination of other diseases at histology. We present a 24-year-old patient suffering from ABH with some clinical peculiarities. Recognition of ABH is of great importance to dermatologists, to avoid misdiagnosis.
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Affiliation(s)
| | - Yu. K. Bukin
- Kaliningrad Regional Center of Specialized Types of Medical Care
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Ordioni U, Hadj Saïd M, Thiery G, Campana F, Catherine JH, Lan R. Angina bullosa haemorrhagica: a systematic review and proposal for diagnostic criteria. Int J Oral Maxillofac Surg 2018; 48:28-39. [PMID: 30032974 DOI: 10.1016/j.ijom.2018.06.015] [Citation(s) in RCA: 17] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/24/2017] [Revised: 06/19/2018] [Accepted: 06/29/2018] [Indexed: 01/17/2023]
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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Affiliation(s)
- U Ordioni
- Department of Oral Surgery and Odontology, APHM, Timone Hospital, Marseille, France; Centre Massilien de la Face, Marseille, France.
| | - M Hadj Saïd
- Department of Oral Surgery and Odontology, APHM, Timone Hospital, Marseille, France; Faculty of Medicine, Aix-Marseille University, EFS/CNRS, APHM, Marseille, France
| | - G Thiery
- Centre Massilien de la Face, Marseille, France
| | - F Campana
- Department of Oral Surgery and Odontology, APHM, Timone Hospital, Marseille, France; Centre Massilien de la Face, Marseille, France
| | - J-H Catherine
- Department of Oral Surgery and Odontology, APHM, Timone Hospital, Marseille, France; Faculty of Medicine, UMR 7268 ADES, Aix-Marseille University/EFS/CNRS, Marseille, France
| | - R Lan
- Department of Oral Surgery and Odontology, APHM, Timone Hospital, Marseille, France; Faculty of Medicine, UMR 7268 ADES, Aix-Marseille University/EFS/CNRS, Marseille, France
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Yorulmaz A, Yalcin B. Is inhaled glucocorticoids the only culprit in angina bullosa hemorrhagica? Indian J Pharmacol 2018; 50:91-93. [PMID: 30100658 PMCID: PMC6044132 DOI: 10.4103/ijp.ijp_19_17] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/10/2017] [Accepted: 05/27/2018] [Indexed: 11/27/2022] Open
Abstract
Angina bullosa hemorrhagica (ABH) is an infrequent dermatosis characterized by acute onset of hemorrhagic bulla in the oral cavity. Clinical presentation of ABH may be quite worrisome, and clinicians often feel skeptical regarding their clinical diagnosis and lack confidence in managing this distinct entity. Indeed, ABH is a completely benign and self-limited disorder. The exact etiopathogenesis of ABH is still unknown. There have been reports in the literature addressing a central role for mechanical instability of the epithelial-connective tissue connection in the pathogenesis of ABH. Moreover, it has been claimed that long-term usage of inhaled glucocorticoids (GCs) is involved in the development of ABH, since most of the reported cases are asthmatic patients, who were treated with inhaled GCs, and GCs are well known for their degradative activities on collagen formation. Here, we describe a case with ABH, who had a drug history of inhaled GCs and nonsteroidal anti-inflammatory drugs (NSAIDs). We assume that our case not only supports the association of inhaled GCs with ABH but also suggests a possible role for NSAIDs in the pathogenesis of ABH.
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Affiliation(s)
- Ahu Yorulmaz
- Department of Dermatology, Ankara Numune Research and Education Hospital, Ankara, Turkey
| | - Basak Yalcin
- Department of Dermatology, Ankara Numune Research and Education Hospital, Ankara, Turkey
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Hernández Aragüés I, Vilas Boas P, Sánchez Herrero A, Suárez Fernández R. [Blood-filled blister on tongue]. Semergen 2017; 44:e74-e75. [PMID: 28655439 DOI: 10.1016/j.semerg.2017.05.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/08/2016] [Revised: 04/23/2017] [Accepted: 05/10/2017] [Indexed: 10/19/2022]
Affiliation(s)
- I Hernández Aragüés
- Servicio de Dermatología, Hospital General Universitario Gregorio Marañón, Madrid, España.
| | - P Vilas Boas
- Servicio de Dermatología, Hospital General Universitario Gregorio Marañón, Madrid, España
| | - A Sánchez Herrero
- Servicio de Dermatología, Hospital General Universitario Gregorio Marañón, Madrid, España
| | - R Suárez Fernández
- Servicio de Dermatología, Hospital General Universitario Gregorio Marañón, Madrid, España
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Alberdi-Navarro J, Gainza-Cirauqui ML, Prieto-Elías M, Aguirre-Urizar JM. Angina bullosa hemorrhagica an enigmatic oral disease. World J Stomatol 2015; 4:1-7. [DOI: 10.5321/wjs.v4.i1.1] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/28/2014] [Revised: 12/01/2014] [Accepted: 01/12/2015] [Indexed: 02/06/2023] Open
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.
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Brajon D, Bagnères D, Berbis P, Rossi P. Angina bullosa haemorragica. J Eur Acad Dermatol Venereol 2014; 30:190-1. [PMID: 25174662 DOI: 10.1111/jdv.12705] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/29/2022]
Affiliation(s)
- D Brajon
- Dermatology department, Hopital Nord, Marseille, France
| | - D Bagnères
- Internal medicine department, Hopital Nord, Marseille, France
| | - P Berbis
- Dermatology department, Hopital Nord, Marseille, France
| | - P Rossi
- Internal medicine department, Hopital Nord, Marseille, France
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