Angina Bullosa Hemorrhagica: A Case Report
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Case Report
P: 103-105
December 2023

Angina Bullosa Hemorrhagica: A Case Report

J Turk Acad Dermatol 2023;17(4):103-105
1. Gazi University Faculty of Medicine, Department of Dermatology, Ankara, Turkey
2. Gazi University Faculty of Medicine, Medical Student, Ankara, Turkey
No information available.
No information available
Received Date: 06.06.2022
Accepted Date: 11.04.2023
Publish Date: 20.12.2023
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ABSTRACT

Angina bullosa hemorrhagica (ABH) is a self-limited oral blistering disorder that heals spontaneously within a few weeks. ABH has a benign nature and clinical diagnosis is usually straightforward. However, it should be differentiated from other bullous disorders affecting oral mucosa in some cases. In this case report, we presented a 57-year-old male patient diagnosed with ABH and revisited this rare entity with its diagnostic and clinical features.

Introduction

Angina bullosa hemorrhagica (ABH) is a benign disorder characterized by sudden onset hemorrhagic bullous lesions that heal spontaneously within 1-2 weeks [1,2]. In this paper, we report a case of ABH presented to our clinic with recurrent hemorrhagic blisters in oral mucosa. Informed consent was obtained from the patient for this study.

Case Report

A 57-year-old man presented to our clinic with blood-filled blisters on the left side of his tongue. His lesions appeared as hemorrhagic bullae and slight erythema a few days ago without subjective symptoms. He had experienced similar lesions for the last 10 years, occurring 1-2 times per year and healing spontaneously. His lesions were triggered by hot drinks and smoking in previous episodes. In his past medical history; hypertension, ear eczema and left-sided direct inguinal hernia was present. Dermatologic examination revealed multiple hemorrhagic ulcers on the left side of tongue (Figure 1). To exclude other diagnoses; complete blood count, biochemistry, coagulation parameters, anti-nuclear antibody profile, erythrocyte sedimentation rate, complement levels, urinalysis tests were performed. His laboratory values were within normal range.

Figure 1

Based on his typical clinical history and examination findings, we diagnosed the patient as ABH. We prescribed benzydamine hydrochloride mouthwash twice a day for 1 week and advised him to avoid hot, spicy, crispy foods and quit smoking. Ten days later, his lesions healed completely (Figure 2).

Figure 2

Discussion

ABH is a self-limited disorder characterized by sudden onset hemorrhagic bullous lesions. Soft palate is the most commonly affected area; followed by buccal mucosa, lateral side of tongue and lip [1,2,3]. Very rarely, gingiva can be affected [4]. Lesions are usually painless; whereas, secondary to rupture of bullae, ulceration and pain may occur [2,5]. Oral cavity floor, esophagus, pharynx, epiglottis involvement can also be seen [5,6,7]. İncision of blisters may be necessary to prevent airway obstruction [2,8]. ABH is seen mainly in middle-aged people. It is reported almost equally in both genders, mean age of diagnosis is 54 [7,8].

Etiology of ABH is obscure. Loosening of cohesion between epithelium and mucosal dermis, and mucosal vascular abnormalities are proposed to play a role in pathogenesis. Hot, spicy foods; dental trauma, intubation, local anesthesia application, endoscopy, air travel are reported as triggering factors [3,5,7,8]. ABH is also more commonly reported in the premenstruation period of women [7]. Inhaled steroid use (especially more than 5 years), hypertension, diabetes mellitus are major predisposing factors of ABH [7,8]. Long term use of inhaled corticosteroids can disrupt collagen and elastin formation, cause epithelial atrophy; and this can cause weakening and breaking down of capillaries [2,3].

Diagnosis can be made easily by typical clinical history. In some situations, exclusion of other disorders presenting with bullous lesions is necessary. Sudden onset, blood-filled tense blisters in soft palate are very typical findings of ABH. Most of the time, biopsy is unnecessary [1]. In order to diagnose ABH, nine criteria are proposed by Ordoni et al. [3] (Table 1) [9].

Table 1

If biopsy is performed from blood-filled bulla, most common anatomic localization of detachment is at the subepithelial area. However, intramucosal and intradermal detachments are also reported in the literature [4]. At the surrounding tissue parakeratosis can be shown. In direct immunofluorescence immunoglobulin (Ig) G, IgA and C3 staining is not observed, which is helpful in differentiating from autoimmune bullous disorders [6].

There is no specific treatment of disorder. Mouthwashes or sprays containing chlorhexidine can be used for symptomatic relief. Ascorbic acid and citroflavonoid containing tablets can also be helpful [3,7].

Conclusion

ABH should be considered in patients presenting with sudden onset bullous lesions that heal spontaneously. Although this may be perceived as a fearful condition at first glance for both patient and clinician, ABH has a benign nature and heals spontaneously. Clinicians should be aware of this benign disorder and differentiate it from other oral blistering disorders.

Ethics

Informed Consent: Informed consent was obtained from the patient for this study.

Peer-review: Internally and externally peer-reviewed.

Authorship Contributions

Surgical and Medical Practices: O.A., M.E.T., E.A., Concept: O.A., M.E.T., E.A., Design: O.A., M.E.T., E.A., Data Collection or Processing: O.A., M.E.T., İ.A.K., E.A., Analysis or Interpretation: O.A., M.E.T., İ.A.K., E.A., Literature Search: O.A., M.E.T., İ.A.K., E.A., Writing: O.A., M.E.T., E.A.

Conflict of Interest: No conflict of interest was declared by the authors.

Financial Disclosure: The authors declared that this study received no financial support.

References

1
Horie N, Kawano R, Inaba J, Numa T, Kato T, Nasu D, Kaneko T, Kudo I, Shimoyama T. Angina bullosa hemorrhagica of the soft palate: a clinical study of 16 cases. J Oral Sci 2008;50:33-36.
2
Yamamoto K, Fujimoto M, Inoue M, Maeda M, Yamakawa N, Kirita T. Angina bullosa hemorrhagica of the soft palate: report of 11 cases and literature review. J Oral Maxillofac Surg 2006;64:1433-1436.
3
Alberdi-Navarro J, García-García A, Cardona-Tortajada F, Gainza-Cirauqui ML, Aguirre-Urizar JM. Angina bullosa hemorrhagica, an uncommon oral disorder. Report of 4 cases. J Clin Exp Dent 2020;12:e509-e513.
4
Kurban M, Kibbi AG, Ghosn S. Expanding the histologic spectrum of angina bullosa hemorrhagica: report of one case. Am J Dermatopathol 2007;29:477-479.
5
Beguerie JR, Gonzalez S. Angina bullosa hemorrhagica: report of 11 cases. Dermatol Reports 2014;6:5282.
6
Giuliani M, Favia GF, Lajolo C, Miani CM. Angina bullosa haemorrhagica: presentation of eight new cases and a review of the literature. Oral Dis 2002;8:54-58.
7
Grinspan D, Abulafia J, Lanfranchi H. Angina bullosa hemorrhagica. Int J Dermatol. 1999;38:525-528.
8
Shoor H, Mutalik S, Pai KM. Angina bullosa haemorrhagica. BMJ Case Rep 2013;2013:bcr2013200352.
9
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 2019;48:28-39.