Angina bullosa hemorrhagica (ABH) with herpes labialis

Angina bullosa hemorrhagica (ABH), a rare disorder, is characterized by the abrupt emergence of blood-filled blisters that damage the oropharyngeal and oral mucosal surfaces. Rupture of the blister and discomfort are frequent outcomes of this condition. Palliative care and observation are treatment options [1–4]. Here, we are describing a case of ABH developed in a patient, presented with resolving herpes labialis.
A 40-year-old male, farmer by occupation, presented to us with complaints of fever along with development of painful, burning, fluid filled vesicles over the lips for last 5 days (fig. 1) and a painless hemorrhagic ruptured bulla over the left buccal mucosa for last 2 days (fig. 2 A). He is a regular smoker and chews tobacco. He had no history of similar episode in past. He had no history of any bleeding disorder or trauma.
On examination, many crusted well-defined papules and ruptured vesicles with crusting along with mild erythema were present over the right upper and lower half of lips without crossing the midline. There was a single large well-to-ill-defined ruptured hemorrhagic bulla with many small bluish blood-filled bullae present over the left palatoglossal fold, buccal mucosa and retromolar trigone with no active bleeding.
All of his vitals were within normal limits. The systemic evaluation came out as normal. The results of routine coagulation workup and haematological examinations were within normal limits. We took scrapings from the base of the ruptured vesicles over the lips and stained with Giemsa for Tzanck smear which did not reveal any multinucleate giant cells, probably due to the 5-day-old lesion. So, a clinical diagnosis of herpes labialis with ABH was made. The patient was prescribed tablets of acyclovir 400 mg thrice daily for 5 days orally along with chlorhexidine mouth wash three times daily just to avoid any secondary infection. He was reviewed after 4 days with resolution of ABH and without any new lesion or scarring along with resolved herpes labialis (fig. 2 B).
ABH is an uncommon, benign illness that manifests as well-defined, bleeding blisters that quickly enlarge and burst on their own between 24 and 48 h after they first form in the oral cavity. Since the disease’s etiopathogenesis is currently unknown, it is thought to be multifactorial. In our case, we thought herpes labialis may be a triggering event in this patient. Lesions can be solitary or many, and the disease typically affects middle-aged and elderly individuals. The soft palate is the most often impacted area [1–4]. Ordioni et al. [5] proposed a diagnostic criterion which includes clinically notable haemorrhagic bulla or erosion with a history of bleeding of the oral mucosa, exclusively oral or oropharyngeal localization, palate localization, triggering event or promoting factor, favourable evolution without a scar within a few days, normal platelet count and coagulation tests, painless lesion and tingling or burning sensation, recurrent lesions, negative direct immunofluorescence. Our patient fulfilled six criteria such as clinically notable haemorrhagic bulla, exclusively oral and palatal localization, having a triggering event prior to development of bullae, resolving without any scar in few days and normal platelet count with coagulation profile. These are sufficient for the diagnosis. In the literature, many triggering factors have been described. Here in our case, developing ABH following herpes labialis may indicate a viral infection may act as an inducing factor for development of ABH. As clinicians we should know this rare oral mucosal benign pathology which may appear after another localised disease.

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Not applicable.

Conflict of interest

The authors declare no conflict of interest.

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