• Users Online: 138
  • Print this page
  • Email this page


 
 
Table of Contents
CASE REPORT
Year : 2022  |  Volume : 15  |  Issue : 4  |  Page : 230-232

Multiple angina bullosa haemorrhagica like oropharyngeal lesions associated with severe acute respiratory syndrome coronavirus 2-induced immune thrombocytopenia: A rare case report


Department of Oral Medicine and Radiology, Faculty of Dentistry, Jamia Millia Islamia, New Delhi, India

Date of Submission06-Aug-2022
Date of Decision06-Sep-2022
Date of Acceptance08-Sep-2022
Date of Web Publication22-Dec-2022

Correspondence Address:
Mandeep Kaur
Department of Oral Medicine and Radiology, Faculty of Dentistry, Jamia Millia Islamia, New Delhi - 110 025
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/hmj.hmj_63_22

Rights and Permissions
  Abstract 


Rationale: Angina bullosa hemorrhagica (ABH) or blood blisters have an acute onset and occur independently of other disturbances of the human body like vesiculo-bullous disorders, vascular disorders, systemic causes etc. ABH is sometimes painful in the oral cavity, most commonly affects the soft palate and less commonly affects the masticatory mucosa. Within a short period of time, these blood filled lesions burst spontaneously resulting in superficial erosions and healing without scarring within 1-2 weeks. ABH has a good prognosis and the only complication reported in the literature has been of airway obstruction. Patient Concerns: A 50 yr old female patient presented with the chief complaint of blood filled blisters and bleeding gums since one week. Diagnosis: Blood filled blisters were noted at multiple sites in the oral mucosa. Blood investigation revealed mild alteration in platelet count and endoscopy displayed involvement of the pharyngeal mucosa. A provisional diagnosis of ABH-like lesion as a covid -19 related delayed inflammatory reaction to LA was given. Interventions: Symptomatic management with topical agents was done. Outcomes: The lesions had completely healed by the seventh day and she remains symptom free eight months post episode. Lessons: The presented case is a very rare condition affecting the oral cavity and involvement of the pharyngeal mucosa additionally in a post COVID-19 patient is even more rare. A delayed inflammatory reaction to LA due to exposure to the SARS-COV-2 spike protein and dysregulation in immune function associated with immune thrombocytopenia (ITP) are postulated in this case of ABH-like lesions involving the oro-pharyngeal mucosa. It is very important to be clinically observant in recovered covid 19 patients too for atypical presentations.

Keywords: Coronavirus, delayed inflammatory reaction, haemorrhagic blisters, immune thrombocytopenia, oral mucosa


How to cite this article:
Kaur M, Gombra V, Rana A, Hasan S. Multiple angina bullosa haemorrhagica like oropharyngeal lesions associated with severe acute respiratory syndrome coronavirus 2-induced immune thrombocytopenia: A rare case report. Hamdan Med J 2022;15:230-2

How to cite this URL:
Kaur M, Gombra V, Rana A, Hasan S. Multiple angina bullosa haemorrhagica like oropharyngeal lesions associated with severe acute respiratory syndrome coronavirus 2-induced immune thrombocytopenia: A rare case report. Hamdan Med J [serial online] 2022 [cited 2023 Feb 1];15:230-2. Available from: http://www.hamdanjournal.org/text.asp?2022/15/4/230/364693




  Introduction Top


The COVID-19 pandemic has brought forth a slew of post-infection presentations and as in the case being reported, angina bullosa haemorrhagica (ABH) could be one of them. These lesions do not have any gender predilection and mainly occur over 50 years of age.[1],[2],[3] They can be distinguished from other mucocutaneous disorders on the basis of their isolated nature and rapid healing pattern. Cognizance of this self-limiting disorder with a potential complication for airway obstruction is important for timely intervention.[2],[3]

Many reasons in the literature have been implicated such as dental restorations, periodontal, prosthodontic treatment; minor trauma due to warm drinks, steroid inhalers, dental local anaesthesia (LA) injections, anticoagulants, etc., but the aetiopathogenesis of these haemorrhagic blisters remains unclear. ABH has been mostly linked to trauma and more often by trauma incurred during various dental treatments.[1],[3]

The aim of this case report is to describe and add an unusual case of multiple haemorrhagic blisters of the oral and pharyngeal mucosa a possible association with post-COVID-19 infection. It is discussed herewith that it could be a delayed inflammatory reaction to dental LA as the immune system probably malfunctions after exposure to severe acute respiratory syndrome coronavirus 2 (SARS CoV-2) spike protein. We also feel that these blisters could occur because of viral induced immune thrombocytopenia (ITP), to distinguish from those attributed to a local or systemic cause before establishing an association with COVID-19 disease.


  Case Report Top


A 50-year-old Indian female patient presented to the outpatient department in early August 2021 with a chief complaint of blood-filled blisters and bleeding gums since 1 week. She reported that these lesions occurred after about a week of receiving LA for root canal therapy (RCT) of her maxillary left posterior tooth. The patient had also received LA for RCT in the past without any adverse effects. Her recent history was negative for any new medications, toothpaste and mouthwashes, etc. She was on treatment since 2–3 years for hypothyroidism and hypertension. The patient contracted the SARS-COV-2 infection in April 2021, for which she had been hospitalised and discharged after recovery.

On intraoral examination, blood-filled blisters were observed bilaterally on the buccal mucosa and vestibule towards the maxillary molars and in the mandibular labial vestibule [Figure 1]. Only the lesions on the maxillary left buccal mucosa and vestibule were slightly painful. On the basis of clinical appearance and history, a provisional diagnosis of ABH as a COVID-19-related delayed inflammatory reaction to LA was given. She was advised blood investigations wherein the parameters were slightly altered. The platelet count was 120 thou/mm3 and INR was 1.11. There was a very marginal alteration in the activated partial thromboplastin time (27.90 s) and prothrombin time (12.30 s). Only reduced platelets were noted in the peripheral blood smear with their morphology being unaltered.
Figure 1: Initial presentation of ABH on the right and left buccal mucosa and lower labial vestibule. ABH: Angina bullosa haemorrhagica

Click here to view


Endoscopy was also advised since she was complaining of moderate burning sensation and difficulty swallowing. It revealed congestion of the posterior pharyngeal wall, mild oedema of the pyriform fossae and ulcerations on the left side ari-epiglottic fold. The patient was managed symptomatically. She was prescribed Benzydamine Hydrochloride 0.15% mouthwash in a 1:1 dilution with lukewarm water to be used as an oral rinse and gargle (three times a day for seven days). In addition, a 0.25% chlorhexidine mouthwash (to be used at bedtime) to prevent secondary infection was given. A soft diet was advised to ease the intake of food and she was recalled every 2–3 days for review.

On the first follow-up visit, after 2 days, the lesions had ruptured leaving behind whitish eroded areas and tissue tags from the collapsed bullae [Figure 2]. By the 7th day of follow-up, the mucosa had completely healed [Figure 3] and she was completely symptom free. This remains so at 12 months post the episode.
Figure 2: Follow-up visit showing ruptured bullae

Click here to view
Figure 3: Complete resolution of the lesions on day 7

Click here to view



  Discussion Top


The haemorrhagic blisters as described did not correspond to the site of the injection only and this widespread affliction of the oro-pharyngeal mucosa suggests an underlying pathogenesis which cannot be explained by trauma alone which is the most common postulated cause for ABH.

In COVID-19 infections, the interplay between platelets and virus infection leading to ITP has come to the forefront. Hypo-proliferative thrombocytopenia can occur at later stages of a viral infection.[4] ITP has been well documented in COVID-19,[4],[5] and in this case report, thrombocytopenia was observed 3 months after acute infection with SARS-COV-2.

Chronic post-COVID-19 syndrome has been mentioned in the literature[6] and ITP causing ABH can be one of the manifestations. The ABH-like lesion in this case report can also be arguable as a result of delayed inflammatory reaction[7] to dental LA following exposure to the coronavirus spike protein since immune dysregulation is a well-documented aspect of infection with coronavirus.

There is one possible explanation for the COVID spike protein related to a delayed inflammatory reaction, i.e. SARS Cov-2 targets the Ace-2 receptor for cell entry where ACE-2 is a ligand for the COVID spike protein and active infection can bind the available Ace-2, which further increases the response of proinflammatory markers. Moreover, there are cases reported in the literature where it has been observed, that these lesions occur several weeks after COVID exposure and sero-conversion may result in a longer rise in serum protein.[7]

Only two cases of ABH-like lesions have been reported involving the tongue and hard palate in SARS-COV-2-infected patients[8] and one of haemorrhagic bullous eruption on the skin secondary to leukocytoclastic vasculitis in a patient affected by COVID-19.[9] Various theories of vascular and thrombotic effects on oral mucosa support the manifestation of these oral alterations in COVID-19 patients. It is important to highlight that many hypotheses may occur concomitantly, and further investigation is necessary to increase evidence regarding the explanations postulated.

We are the first to describe the case of oral haemorrhagic blisters occurring as a possible long-term sequela of ITP in chronic COVID-19 syndrome. We also impress the importance of further scientific and clinical vigilance and surveillance even in recovered and asymptomatic patients of COVID-19.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Rai S, Kaur M, Goel S. Angina bullosa hemorrhagica: Report of two cases. Indian J Dermatol 2012;57:503.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Peters JP, van Kempen PM, Robijn SM, Thomeer HG. Angina bullosa hemorrhagica: Post-traumatic swelling in the oral cavity-a case report. J Adv Oral Res 2020;11:97-100.  Back to cited text no. 2
    
3.
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.  Back to cited text no. 3
    
4.
Wool GD, Miller JL. The impact of COVID-19 disease on platelets and coagulation. Pathobiology 2021;88:15-27.  Back to cited text no. 4
    
5.
Metallidis S, Gioula G, Papaioannou M, Exindari M, Chrysanthidis T, Tsachouridou O, et al. Immune thrombocytopenia in a patient recovering from COVID-19. Hemasphere 2020;4:e419.  Back to cited text no. 5
    
6.
Nalbandian A, Sehgal K, Gupta A, Madhavan MV, McGroder C, Stevens JS, et al. Post-acute COVID-19 syndrome. Nat Med 2021;27:601-15.  Back to cited text no. 6
    
7.
Munavalli GG, Guthridge R, Knutsen-Larson S, Brodsky A, Matthew E, Landau M. "COVID-19/SARS-CoV-2 virus spike protein-related delayed inflammatory reaction to hyaluronic acid dermal fillers: A challenging clinical conundrum in diagnosis and treatment". Arch Dermatol Res 2022;314:1-15.  Back to cited text no. 7
    
8.
Iranmanesh B, Khalili M, Amiri R, Zartab H, Aflatoonian M. Oral manifestations of COVID-19 disease: A review article. Dermatol Ther 2021;34:e14578.  Back to cited text no. 8
    
9.
Negrini S, Guadagno A, Greco M, Parodi A, Burlando M. An unusual case of bullous haemorrhagic vasculitis in a COVID-19 patient. J Eur Acad Dermatol Venereol 2020;34:e675-6.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
Abstract
Introduction
Case Report
Discussion
References
Article Figures

 Article Access Statistics
    Viewed216    
    Printed4    
    Emailed0    
    PDF Downloaded43    
    Comments [Add]    

Recommend this journal