INTRODUCTION

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Hemorrhagic bullous angina (HBA) is a disorder not related to the systemic causes or disease vesiculobullous characterized by one or more subepithelial vesicle with blood in the oral mucosa and oropharyngeal, more common in the soft palate[1]. It can occur in men and women, especially above middle age, that fact there has never been a report in children under 10-year-old[1].

According to Bahdam (1967) cited by Santos et al.,[2] blood blisters are not attributed to blood dyscrasia, systemic disorder, or vesiculobullous disorder.

The HBA usually develops through local trauma but could be related to the prolonged use of inhaled steroids and diabetes mellitus. Its etiology remains uncertain. The condition appears to be associated with an individual's predisposition, such as poor adhesion between the epithelium and the connective tissue of the mucosal or even poor adhesion of the mucosal vessels[3].

CASE REPORT

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A 58-year-old female patient, sought the Dentistry Service at the University of West of Santa Catarina- SC, with the chief complaint of replacement of the upper and lower total dentures. Anamnesis was performed and the patient reported use of formoterol fumarate dihydrate + budesonide (Foraseq) and salbutamol sulfate (Aerolin). The clinical examination indicated the need for pre-prosthetic surgeries such as correction and reduction of soft tissue of the right and left maxillary tuberosity besides and frenectomy. Fasting blood glucose and complete blood count were requested.

In the second session, with the examinations into the normal standards (hemogram, prothrombin time 11.5 s and partial activated thromboplastin time 25.0 s), were planned the surgeries of correction of left tuberosity and frenectomy. During the first procedure (frenectomy), bleeding remained normal and blood pressure was stable. After the upper lip frenulum was sutured, the left tuberosity was anesthetized. At this time, blood bubbles were observed on the soft palate[1,2] of various sizes [Figure 1], which were not present at the beginning of the first surgical procedure. The bubbles arise despite the little use of the suction unit. The session was interrupted and the patient was followed up on the days that followed and the blisters burst and disappeared without sequelae at the end of 10 days.

Figure 1

DISCUSSION

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HBA is characterized by the rapid formation of a blood bubble/ blister. Lesions may occur in the mucosa, lip, and lateral surface of the tongue, especially in the soft palate region. It can affect men and women, usually older. The term "angina" comes from the sensation of shock that the appearance of the bubbles can cause. The main causes are associated on traumas and the prolonged use of inhaled steroids, but some studies relate diabetes mellitus as a possible cause. The differential diagnosis should be performed with complementary examinations, for example, hematological examinations and some histological studies to rule out pemphigus and pemphigoid hypothesis[1]. In this case, any other pathology than asthma was excluded from the study, the patient was healthy and exhibited all the main normal complementary examinations (fasting glycemia, coagulogram, and hemogram).

Chronic use of inhaled corticosteroids is known to possibly affect collagen synthesis and reduces its total mucosal content causing atrophy of the mucosal epithelium. In addition, the elasticity of the tissue may decrease with maturation of these fibers. This would reduce the blood supply in these regions, which would explain the development of HBA, even without major on -site trauma. Some authors still associate HBA with the use of inhaled corticosteroids by patients with asthma and chronic obstructive pulmonary disease based on studies with patients who have used it for more than 5 years[4]. In fact, the patient reported in the study using medication (Foraseq and Aerolin) for regular treatment of asthma, which caused the appearance of blisters on the soft palate.

In a reported case by Garlick and Calderon,[5] trauma during routine odontological procedures could occur as a precipitating factor in the appearance of HBA. This fact is reinforced by Daly (1988) who describes the appearance of the bubbles after contact of the mirror used in photographic documentation, appearing almost instantaneously. These reports are in agreement with our study, which the patient also developed HBA fast[2], however, without having received mechanical trauma at the site.

There are case reports associating the occurrence of these lesions in patients submitted to hemodialysis,[6]hypertension, and even the ingestion of hot or very spicy foods[7]. The latter, however, described two cases of HBA without any evidence of trauma. In turn, there are also reports of HBA after the ingestion of hard or crunchy foods as triggers of the lesions[8].

Although the risk of choking may be remote, blisters on the palate or oropharyngeal region should be ruptured to prevent possible airway obstruction[3].

The rupture usually occurs during meals and healing takes normally of 7-10 days. As reported by Rai et al.,[9] the blisters lasted only a few minutes and then ruptured, leaving shallow ulcers that healed without pain in both cases. The treatment is basically palliative, with the use of chlorhexidine mouthwash 0.12-0.20% twice a day for 1 min as prevention of secondary infections. However, for painful ulcers,[8] they recommend the use of benzydamine hydrochloride for the relief of symptoms. In general, the prognosis is good. In this case, the patient evolved well without any complications.

Abrupt onset, spontaneous origin, constant presence of blood within the blister, rapid recovery, and absence of local specific cause, or other systemic disorder distinguishes HBA from membranous pemphigoid, bullous pemphigoid, bullous lichen planus, epidermolysis bullosa, dermatitis herpetiformis, dermatosis by linear IgA, and oral amyloidosis. [6] According to Rai et al.,[7] thrombocytopenia may also be a differential diagnosis.

The classic evolution, the morphology of the lesions, anamnesis well performed, and absence of other symptoms were essential for the correct diagnosis in this case.

REFERENCES

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1. Volkweis MR, Galeazzi S. Angina bullous hemorrhagic - one review of 14 cases. RFO, Passo Fundo 2010;15:298-301.
2. Santos BR, Figueiredo MA, Oliveira PT, Yurgel LS. Angina hemorrhagic bullous: An unusual case. Rev Fac Odontol Univ Passo Fundo 2001;6:7-10.
3. Beguerie JR, Gonzalez S. Angina bullosa hemorrhagica: Report of 11 cases. Dermatol Rep 2014;6:5282.
4. Martins CA, Gomes FV, Freddo AL, Heitz C, Moresco FC, Silveira JO. Angina bullosa haemorrhagica (ABH): Diagnosis and treatment. RFO, Passo Fundo 2012;17:347-51.
5. Garlick JA, Calderon S. Oral blood blisters in angina bullosa haemorrhagica secondary to trauma of eating and dental injection. Br Dent J 1988;22165:286-7.
6. Shashikumar B, Reddy RR, Harish M. Oral hemorrhagic blister: An enigma. Indian J Dermatol 2013;58:407.
7. Masdemont BL, Munoz LG, Santullano AV, Blanco VP, Dominguez MC. Langerhans cell histiocytosis mimicking lichen nitidus with bone involvement. Australas J Dermatol 2016. DOI: 10.1111/ajd.12467.
8. Horie N, Kawano R, Inaba J, Numa T, Kato T, Nasu D, et al. Angina bullosa hemorrhagica of the Angina bullosa hemorrhagica of the soft palate, a clinical study of 16 cases. J Oral Sci 2008;50:33-6.
9. Rai S, Kaur M, Goel S. Angina bullosa hemorrhagica: Report of two cases. Indian J Dermatol 2012;57:503.