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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 8  |  Issue : 2  |  Page : 124-128

Hemorrhagic bulla on the lip: A diagnostic dilemma


Department of Oral Medicine and Radiology, Swami Devi Dyal Hospital and Dental College, Barwala, Haryana, India

Date of Web Publication18-Dec-2018

Correspondence Address:
Aravinda Konidena
Department of Oral Medicine and Radiology, Swami Devi Dyal Hospital and Dental College, Barwala, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijohs.ijohs_17_18

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  Abstract 


Angina bullosa hemorrhagica (ABH) is a rare benign disorder characterized by the sudden onset of painless blood-filled blister in the oral cavity that rupture in 24–48 h. We recently encountered an interesting case of ABHon the lower lip of 68-year-old female patient referred back from the prosthodontic department following the procedure of impression making. This case will be presented along with a review of previous case summaries reporting four or more cases.

Keywords: Angina bullosa hemorrhagica, hemorrhagic blister, iatrogenic blister, oral bulla, trauma


How to cite this article:
Konidena A, Shaw H, Uppal MK. Hemorrhagic bulla on the lip: A diagnostic dilemma. Int J Oral Health Sci 2018;8:124-8

How to cite this URL:
Konidena A, Shaw H, Uppal MK. Hemorrhagic bulla on the lip: A diagnostic dilemma. Int J Oral Health Sci [serial online] 2018 [cited 2019 Mar 21];8:124-8. Available from: http://www.ijohsjournal.org/text.asp?2018/8/2/124/247798




  Introduction Top


Hemorrhagic bullae are uncommon in the oral mucosa and are frequently caused by trauma. The trauma may be inflicted either due to mechanical injuries or iatrogenic causes. Acute traumatic injuries of the mouth commonly present as ulcers, abrasions, or ecchymoses. Rarely, they may present as blood filled blisters, causing apprehension to both the clinician and the patient. Angina bullosa hemorrhagica (ABH) is characterized as the sudden onset of blisters in the oral and oropharyngeal mucosa that cannot be attributed to blood dyscrasias, vesiculobullous disorders (pemphigus), autoimmune conditions, or vascular disease.[1] It is a rare disorder of unknown etiology and was described first by Balina of Argentina in 1933 under the term traumatic oral hemophlyctenosis and in 1967, the term ABH was coined by Badham.[2] Exaggerated host susceptibility to dental or functional trauma may be a significant element in the causation of this condition.[3] We recently encountered a patient who rapidly developed a blood-filled blister while undergoing final impression procedure. The case, along with relevant literature is being presented here.


  Case Report Top


A 68-year-old female patient presented to our department with a chief complaint of missing teeth for 2–3 years. She underwent multiple extractions of teeth over the past 2–3 years followed by uneventful postoperative healing. Her edentulous state was associated with difficulty in chewing for which she was referred to the Department of Prosthodontics for fabrication of complete denture prosthesis. After a few days, while secondary impression was being made, the patient suddenly developed a blister on the lower lip and was referred back to our department for evaluation and management. She mentioned that the blister was initially small, increased to the present size within 20–30 min, causing feeling of fullness in the lower lip. The blister was painless and was not associated with any other significant finding. There was no similar history in the past or previous history of bleeding diatheses or use of anticoagulants. Her medical history was unremarkable. On extraoral examination, deepening of nasolabial fold, prominence of the chin, thinning of lips, deepening of mentolabial sulcus, hollowing of cheeks was seen. The intraoral inspection revealed a well-defined solitary bean-shaped maroon colored bulla of size 2.5 cm × 1 cm on lower labial mucosa extending superoinferiorly about 1 cm from the vermillion border up to few mm from the depth of vestibule and mediolaterally from midline to 1.5 cm from the left corner of the mouth [Figure 1]. No visible pulsation or discharging sinuses were seen. Surrounding mucosa appeared to be normal. On palpation, it was nontender, turgid and fluctuant inconsistency, slightly compressible without palpable pulsations. Translucency was absent. Based on the history and clinical examination, the provisional diagnosis of ABH was considered. Bleeding diathesis, autoimmune blistering diseases were considered under differential diagnosis and the patient's hematological profile was checked. Her platelet count was 2,00,000 cells/cu mm, bleeding time was 2 min, clotting time was 4 min, international normalized ratio was 1. On the attempt of aspiration, copious fresh blood gushed out from the site of puncture, size of lesion regressed and hemostasis could be secured by applying local pressure itself [Figure 2]. Despite extensive counseling, the patient did not consent for histopathological examination and further immunofluorescence investigations to rule out the possibility of blistering disease. The patient was instructed to follow the postoperative care and was recalled after a week. The patient reported with satisfactory healing after 10 days [Figure 3].
Figure 1: Angina bullosa hemorrhagica presenting as hemorrhagic blister on lower lip

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Figure 2: Lesion after aspiration

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Figure 3: Complete resolution of lesion after 10 days

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  Discussion Top


ABH is a rare benign disorder characterized by sudden onset of painless, blood-filled, blisters of the oral cavity that quickly expand and rupture spontaneously within 24–48 h.[1],[2],[3],[4] It is also known as localized oral purpura,[5] stomatopompholyx hemorrhagica[6] and recurrent oral hemophlyctenosis.[7]

The summary of case series reporting four or more cases is presented in [Table 1].[1],[6],[8],[9],[10],[11],[12],[13],[14] The reported causes of ABH include trauma from eating including hot, spicy, crispy or hard foods (36.3%), mechanical trauma from sharp teeth (2.9%), dental (n = 1)/endoscopic procedures, shouting/sneezing, inhaled corticosteroids, diabetes (n = 7), and high blood pressure (23%).[1],[4],[6],[8],[9],[10],[11],[12],[13],[14],[15] The chronic use of inhaled steroids may reduce collagen synthesis along with a decrease in tissue elasticity, which predisposes toward bulla formation even with minor trauma. Grinspan et al. advocated the contribution of altered glucose metabolism in the pathogenesis of ABH because they found a positive family history of diabetes mellitus or a sign of abnormal glucose metabolism in 44% of their patients. It is unclear, however, if the altered glucose metabolism contributed to the formation of ABH in their cases.[7] Systemic conditions proposed to be associated with ABH include are hypertension, asthma, diabetes, rheumatoid arthritis, gastrointestinal disturbances, renal failure, and hyperuricemia.[6],[8],[9],[10],[11],[12],[13],[14],[15],[16] Other investigators hypothesize that weakened epithelial-connective tissue junction or a weak anchorage of mucosal vessels.[3],[14],[15] Could predispose to ABH even with minor trauma. In the present case, the final impression procedure could have been the trigger, and the patient did not have any significant past medical history.
Table 1: Summary of angina bullosa hemorrhagica case series of four or more cases

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ABH most commonly affects 50– 70-year-old of both genders,[6],[8],[12],[13],[14] as was seen in the present case. It may present as solitary or multiple red or purple bullae, 2–3 cm in size, that spontaneously rupture within 24–48 h to leave a ragged ulcer.[3],[15] As our patient developed the condition in a dental setting, she presented with an intact, painless turgid bulla of size 2.5 cm, with a feeling of fullness on the lower lip. Some patients may present with pain, bleeding, burning, blood-tinged sialorrhea, hoarseness, or airway obstruction depending on the location.[1],[4],[6],[11],[13],[14] The sites reported to be involved in order of decreasing frequency include the junction of hard and soft palates (80%), tongue (n = 10), buccal mucosa, lips apart from the anterior pillar of the fauces, epiglottis, arytenoids, pharyngeal wall, and esophagus.[4],[6],[8],[9],[10],[11],[12],[13],[14]

Diagnosis of ABH depends on clinical appearance predominantly by the exclusion of other conditions. Detailed medical history taking may facilitate the diagnosis. However, achieving the diagnosis may be difficult and often delayed when the bulla has ruptured because of its resemblance to other similar-appearing conditions involving oral mucosa. Coagulation disorders can be excluded by hematological examination, as was done in the present case. The absence of characteristic lesions and systemic involvement, negative immunofluorescence studies differentiate ABH from oral lichen planus, pemphigus, pemphigoid.[13],[14],[15] In our patient, the systemic disease was ruled out only by history and clinical examination, as the patient did not consent for biopsy and further investigations. ABH might be differentiated from hemorrhagic bullae of amyloidosis by the absence of macroglossia or petechiae.[13],[14],[15] In case of doubt, histopathological examination of the lesion showing nonspecific findings such as hemorrhagic subepithelial bulla, chronic inflammatory cell infiltrate within the lamina propria and a decrease in elastic network leading to fragility of vessel walls may support the diagnosis of ABH.[4],[6],[9],[12],[14] Since the history was pointing to the diagnosis in the present case, supported by normal hematological parameters, and the patient was not willing for biopsy, the histopathological examination could not be carried out.

Treatment of ABH is aimed at providing symptomatic relief. Drainage or aspiration was performed by some authors, as in the present case.[9],[11] ABH treatment may include the use of anti-inflammatory agents, antibiotic or antisepsis with mouthwash containing 0.25% or 0.12% chlorhexidine digluconate to avoid secondary infections.[1],[10],[11] Surgical drainage may only be necessary for large lesions that functionally affect the airways. Very rarely, large-sized bulla and the blood leakage in the larynx may require intubation of the trachea by either fiberoptic endoscopy or direct laryngoscopy, or securing the airway by surgical tracheostomy.[17] Long-term follow-up is recommended to positively exclude other conditions which may present with oral blood containing bullae.[15] Diagnosis of ABH can be established only after ruling out serious medical conditions, which is important to avoid unnecessary complications. It is important for the health professional to diagnose ABH, not only to counsel the patient about the benign nature of the lesion but also to avoid subjecting the patient to unnecessary treatment procedures.[13],[16]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Martins CA, Freddo AL, Heitz C, Moresco FC, da Silveria JO. Angina bullosa hemorrhagica (ABH): Diagnosis and treatment. RFO Passo Fundo 2012;17:347-51.  Back to cited text no. 1
    
2.
Badham NJ. Blood blisters and the oesophageal cast. J Laryngol Otol 1967;81:791-803.  Back to cited text no. 2
    
3.
Shoor H, Mutalik S, Pai KM. Angina bullosa haemorrhagica. BMJ Case Rep 2013;2013. pii: bcr2013200352.  Back to cited text no. 3
    
4.
Lozano-Masdemont B, Bergón-Sendín M, Suárez-Fernández R. Two cases of spontaneous angina bullosa hemorrhagica. Acta Otorrinolaringol Esp 2016;67:300.  Back to cited text no. 4
    
5.
Scully C. The oral cavity. In: Rook AJ, editor. Textbook of Dermatology. 5th ed. Oxford: Blackwell Scientific Publications; 1992. p. 2732-3.  Back to cited text no. 5
    
6.
Deblauwe BM, van der Waal I. Blood blisters of the oral mucosa (angina bullosa haemorrhagica). J Am Acad Dermatol 1994;31:341-4.  Back to cited text no. 6
    
7.
Grinspan D, Abulafia J, Lanfranchi H. Angina bullosa hemorrhagica. Int J Dermatol 1999;38:525-8.  Back to cited text no. 7
    
8.
Stephenson P, Lamey PJ, Scully C, Prime SS. Angina bullosa haemorrhagica: Clinical and laboratory features in 30 patients. Oral Surg Oral Med Oral Pathol 1987;63:560-5.  Back to cited text no. 8
    
9.
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;8:54-8.  Back to cited text no. 9
    
10.
Horie N, Kawano R, Inaba J, Numa T, Kato T, Nasu D, et al. Angina bullosa hemorrhagica of the soft palate: A clinical study of 16 cases. J Oral Sci 2008;50:33-6.  Back to cited text no. 10
    
11.
Martini MZ, Lemos CA Jr., Shinohara EH. Angina bullosa hemorrhagica: Report of 4 cases. Minerva Stomatol 2010;59:139-42.  Back to cited text no. 11
    
12.
Serra D, De Oliveira HS, Reis JP, Vieira R, Figueiredo A. Angina bullosa haemorrhagica: A disorder to keep in mind. Eur J Dermatol 2010;20:509-10.  Back to cited text no. 12
    
13.
da Rosa AM, Pappen FG, Gomes AP. Angina bullosa hemorrhagica: A rare condition? RSBO 2012;9:190-2.  Back to cited text no. 13
    
14.
Beguerie JR, Gonzalez S. Angina bullosa hemorrhagica: Report of 11 cases. Dermatol Reports 2014;6:5282.  Back to cited text no. 14
    
15.
Singh D, Misra N, Agrawal S, Misra P. Angina bullosa haemorrhagica. BMJ Case Rep 2013;2013. pii: bcr2012008505.  Back to cited text no. 15
    
16.
Shashikumar B, Reddy RR, Harish M. Oral hemorrhagic blister: An enigma. Indian J Dermatol 2013;58:407.  Back to cited text no. 16
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17.
Pahl C, Yarrow S, Steventon N, Saeed NR, Dyar O. Angina bullosa haemorrhagica presenting as acute upper airway obstruction. Br J Anaesth 2004;92:283-6.  Back to cited text no. 17
    


    Figures

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    Tables

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