Indian Journal of Dermatology
  Publication of IADVL, WB
  Official organ of AADV
Indexed with Science Citation Index (E) , Web of Science and PubMed
 
Users online: 2202  
Home About  Editorial Board  Current Issue Archives Online Early Coming Soon Guidelines Subscriptions  e-Alerts    Login  
    Small font sizeDefault font sizeIncrease font size Print this page Email this page


 
Table of Contents 
CORRESPONDENCE
Year : 2021  |  Volume : 66  |  Issue : 1  |  Page : 103-105
Progesterone-induced angina bullosa hemorrhagica


Department of Dermatology, Venereology and Leprosy, IMS and SUM Hospital, Bhubaneswar, Odisha, India

Date of Web Publication1-Feb-2021

Correspondence Address:
Annie Raizada
Department of Dermatology, Venereology and Leprosy, IMS and SUM Hospital, Bhubaneswar, Odisha
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_274_19

Rights and Permissions



How to cite this article:
Raizada A, Behera D, Panda M, Debata I. Progesterone-induced angina bullosa hemorrhagica. Indian J Dermatol 2021;66:103-5

How to cite this URL:
Raizada A, Behera D, Panda M, Debata I. Progesterone-induced angina bullosa hemorrhagica. Indian J Dermatol [serial online] 2021 [cited 2021 Feb 28];66:103-5. Available from: https://www.e-ijd.org/text.asp?2021/66/1/103/308494




Sir,

Angina Bullosa Hemorrhagica is a benign, relatively rare condition of uncertain etiology, characterized by acute onset of painless, blood filled blisters of oral and oropharayngeal mucosa, which is unrelated to any systemic disease, vesiculo bullous condition, blood dyscrasia, or any autoimmune pathologies.[1] This entity was first described as traumatic oral hemophlyctenosis in 1933 and in 1967 Badham used the currently accepted term Angina Bullosa Hemorrhagica.[1] Clinically, it manifests as solitary or rarely multiple lesions,[2] often asymptomatic (painful in few cases) with sudden onset, color ranging from dark red to purple, expands quickly and spontaneously ruptures, within 24-48 h, leaving a shallow ulcer which heals without scarring, pain, or discomfort. Most common region affected is the soft palate. It affects ages between 50 and 70 years commonly with no gender predilection[2] Exact etiopathogenesis is unclear and is considered a multifactorial entity with majority of cases resulting due to minor trauma to oral mucosa. This condition is self-limiting and no treatment is required in majority of cases.

A 36-year-old female, presented with sudden onset of a single, non-tender, tense, dark red, round blister of size 3 cm * 2 cm on floor of the mouth for the past 2 days. Roof of the blister was covered with translucent mucosa and the content was hemorrhagic fluid. Surface of blister was smooth, with no secondary changes and visible pulsations [Figure 1]. Patient gave a history of recurrent episodes of such lesion, 7-8 days prior to the onset of periods, and resolved spontaneously on its own with the start of cycles every month. We advised her to re-visit once her lesion gets subsided. Seven days later, with onset of her cycles she revisited us, and size of lesion had reduced to size <0.5 cm [Figure 2].
Figure 1: A single tense hemorrhagic bulla of size 3 cm*2 cm on floor of mouth on the first visit, 7-8 days before onset of cycles in a patient of 36 years old female

Click here to view
Figure 2: A single tense hemorrhagic bulla of size <0.5 cm on floor of mouth on the second visit, at the onset of cycle in a patient of 36 years old female

Click here to view


There were no petechiae, ecchymosis, other mucosal, or skin changes. The margins of the lesion appeared normal. Regional lymph nodes were not enlarged and systemic examination did not reveal any abnormality. Lesion was not associated with fever or other constitutional symptoms, no medical history of blood dyscrasias, anti-coagulant therapy, no significant family or personal history. There was no history of trauma to oral cavity, no history of menstrual irregularities, or any drug intake. Patient was on no other drugs. Subsequently on further visits, we estimated her serum progesterone levels coinciding with the day of appearance and subsidence of the bulla. The serum progesterone was 28 ng/ml and 0.75 ng/ml, respectively. (Normal values of progesterone: In luteal phase 2 ng/ml–30 ng/ml, peaking during progesterone surge i.e., during 21st day of menstrual cycle and in menstrual phase <1 ng/ml).

Routine blood picture which included Bleeding Time, Clotting Time, Platelet count, Prothrombin Time, WBC count, and random Blood sugar levels were within normal limits. As patient was aware of self-resolution of the lesion in past, she refused to undergo biopsy and immunofluorescence testing. Patient was counseled and no active treatment was given and regular follow up was advised. Based on history, clinical examination, and serum progesterone levels, a diagnosis of Angina Bullosa Hemorrhagica was made, course of which directly co-related with her menstrual cycles.

Angina Bullosa Hemorrhagica, is defined as acute, usually subepithelial, oral or oropharyngealmucosal, blood filled blisters, which are not attributed to any systemic or hemostatic disorders. Although, Angina Bullosa Hemorrhagica (ABH) is considered as a benign condition, choking or gagging sensation can be seen associated with lesions in the posterior pharynx or in epiglottis.[3] Acute upper airway obstruction has been also reported as a rare complication.[3] The etiology is unclear and the cause remains multifactorial; however, minor traumatic insult to mucosa due to intake of solid, crispy, hot food items, and sometimes endoscopic trauma or dental procedures may be the causative factors.[4] However, steroid hormone has been a predisposing factor, i.e., chronic use of inhaled steroids in asthmatics.[5] Systemic diseases like chronic renal failure, hypertension, diabetes mellitus have been attributed to the cause.[4] The predisposing factors can cause weakened junction between epithelial and connective tissues, especially in the nonkeratinized mucosa, which results in the formation of blood filled sub-epithelial oral blisters. Most common site of involvement is soft palate. Other areas that can be involved are lateral and ventral aspect of tongue, lips, buccal mucosa, floor of mouth. Gingiva and hard palate may rarely be involved.[6] After thorough literature review, we could not find any evidence indicating progesterone hormone or menstrual cycle association with ABH and the possible reason and mechanism of association between ABH and hormonal variations during menstrual cycle could not be elicited, as we could not get adequate related literature about it and hence, possible explanation requires further research.

Differential diagnosis includes linear immunoglobulin A disease, benign mucous membrane pemphigoid, oral bullous lichen planus, amyloidosis, epidermolysis bullosa, erythema multiforme, bullous fixed drug eruption, and pemphigus vulgaris.

Diagnosis is generally clinical; however, blood picture, punch biopsy, and Direct immunofluorescence (DIF) can be done to rule out other conditions. ABH carries a good prognosis and treatment modalities includes diet counseling and avoiding dried, crispy, coarse food. A total of 0.2% chlorhexidine gluconate oral rinse to prevent infections[5] or benzydamine hydrochloride oral rinse provides symptomatic relief.

To conclude, our patient is an unusual case of ABH with cyclical onset and subsidence associated with menstrual cycle. Hormonal induced ABH has not been described in literature to the best of our knowledge.

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.
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.
Patigaroo SA, Dar NH, Thinles T, Ul-Islam M. Multiple angina bullosa hemorrhagica: A case report. Int J Pediatr Otorhinolaryngol 2014;9:125-7.  Back to cited text no. 2
    
3.
Cinar SL, Kartal D, Canöz Ö, Borlu M, Ferahbas A. Case Report: A rare cause oforal bullae: Angina bullosa hemorrhagica. F1000Res 2017;6:1974.  Back to cited text no. 3
    
4.
Supekar BB, Sawatkar G, Wankhade VH. Angina bullosa hemorrhagica. Indian Dermatol Online J 2019;10:89.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Magesh KT, Malathi N, Anandan S. 'Angina Bullosa Hemorrhagica - A case report'. JIAOMR 2006;18:103-5.  Back to cited text no. 5
    
6.
Asokan GS, Anand V, Balaji N, Aswini S, Balachander N. Angina Bullosa Hemorrhagica: A rare case report. Biomed Pharmacol J 2015;8:105-7.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2]



 

Top
Print this article  Email this article
 
 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
    Article in PDF (999 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    References
    Article Figures

 Article Access Statistics
    Viewed577    
    Printed2    
    Emailed0    
    PDF Downloaded11    
    Comments [Add]    

Recommend this journal