10-Angina Bullosa Hemorrhaegica

Int J Dent Case Reports 2014; 4(1): 52-56
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ANGINA B ULLOSA HEMORRHAEGICA
N Rakesh 1 , Shwetha V2 , Yashoda Devi B K3 , Deepa B Pat il4
1
Reader, Depart ment of Oral Medicine, Diagnosis & Radio logy, M S Ramaiah Dental College & Hospital,
Bangalore
2
Senior Lecturer, Depart ment of Oral Medicine, Diagnosis & Radiology, M S Ramaiah Dental Co llege & Hospital,
Bangalore
3
Professor, Depart ment of Oral Medicine, Diagnosis & Radiology, M S Ramaiah Dental Co llege & Hospital,
Bangalore
4
Post Graduate Student, Department of Oral Medicine, Diagnosis & Rad iology, M S Ra maiah Dental Co llege &
Hospital, Bangalore
Address for Correspondence
Dr. Shwetha. V.
454/3, 3rd Main, 6th cross, MSR Nagar
Bangalore – 54
Telephone No. - +919620100085
E- Mail Address – [email protected] m
ABSTRACT
Introduction: ‘Angina bullosa hemo rrhaegica’(ABH) is a benign condition affecting mucosa of o ropharynx,
characterized by sudden appearance of blood filled submucosal blisters of unknown aetiology. No definitive
etiological factors have been found associated with A BH, b ut d iabetes, inhaled long-term steroids, trauma, irregular
margins of restorations, periodontal therapy, hereditary predisposition, may be associated in some instances. This
paper highlights the clinical presentation of the disease with a report of t wo cas es.
Case Reports: Two subjects presented to us, one with hemo rrhaegic bulla and another with erosion which has
formed after bursting of blood filled vesicle over the anterolateral aspect of tongue. No significant medical, family
and drug history noted in them. Biopsy and other routine investigations ruled out vesiculobullous and blood
dyscrasias. Thus arriving at diagnosis of ABH. They are still on follow up with no evidence of similar lesion after
removal of et iological factors.
Discussion: ABH is an acute and occasionally painful but benign condition of the oral mucosa characterized by the
sudden onset of blood filled vesicles and bullae. These hemorrhagic bullae rupture spontaneously after a short time
resulting in ragged, often painless superficial erosions that heal spontaneously within 1 week without scarring. These
lesions are not attributable to blood dyscrasias, vesiculobullous disorders, systemic d iseases though their clin ical
appearance may mimic them leading to misdiagnosis.
Conclusion: It is utmost important for the clinician to overcome misdiagnosis so as to provide better treatment to the
patient. Thus, Oral d iagnosticians play a vital role in d iagnosing such lesions.
KEYWORDS: Angina Bullosa Hemorrhaegica, hemorrhagic bullae, erosions
52
N Rakesh, Sh wetha V, Yashoda Devi B K, Deepa B Pat il
Angina bullosa hemorrhaegica
INTRODUCTION
blisters after a prodro me of pain and burning
Angina bullosa hemorrhaegica (ABH) is a rare
sensation. The blisters coalesced to form a large
disorder of unknown et iology. (1) Sir Badham coined
single lesion. The patient reported similar ep isodes of
the term angina bullosa hemorrhagica to describe a
blood filled blisters appearing in her oral cavity at
condition of oral mucosa which is affected by blood
varied locations for the past 6 months. Her past
filled painful blisters of abrupt onset. (2) It was first
med ical,
described and characterized as the sudden onset of
noncontributory. Intraoral examination revealed a
blisters in the oral and oropharyngeal mucosa that
single
cannot be attributed to vesiculobullous disorders
infero lateral aspect of the tongue as depicted in
(pemphigus),
Figure 1. Routine b lood examination which included
b lood
dyscrasias,
autoimmune
family
large
and
drug
hemorrhagic
the
prothrombin time, W BC count and random blood
inconclusive. Most cases described in literature seem
sugar levels were with in normal limits. Clin ical
to be associated with the long term use of inhaled
diagnosis of angina bullosa hemorrhagica was made.
steroids, others have been attributed the lesions to the
Incisional biopsy of the lesion was done to rule out
consumption of hot beverages (thermal inju ry) and
vesiculobullous
mastication related trau matic injuries. (1)
examinations revealed hemorrhagic areas with mild
The awareness about the clinical presentation of
inflammatory infilt rate. No treat ment was advised
ABH in the field of dentistry is very much necessary
and the lesion healed spontaneously without scarring
as it mimics other lesions leading to misdiagnosis.
within 3 days. Pat ient was educated and reassured
This
and was recalled in event of similar episodes.
diagnostic
criteria,
lesions.
clotting
left
Discussions about the etiology of A BH remain
the
time,
on
platelet
highlights
bleeding
bulla
were
conditions or vascular disease. (1)
paper
count,
history
time,
Histopathological
essentially based on clinical data, possible treatment
approaches of the disorder with a report of t wo cases.
Figure 2: Erosion present over the right anterolateral
Figure 1: Large b lood filled bulla present over the left
aspect of the tongue.
anterolateral aspect of tongue
CAS E 1
CAS E 2
A 49 year old wo man presented with a painful lesion
A 52 year old male patient reported to the outpatient
on her tongue since yesterday. The patient was
department with the chief comp laint of pain in the
apparently well when she developed blood filled
right lateral aspect of the tongue since a day. The
53
Int J Dent Case Reports
March 2014, Vo l.4, No. 1
N Rakesh, Sh wetha V, Yashoda Devi B K, Deepa B Pat il
Angina bullosa hemorrhaegica
patient reported that he initially developed blood
burning sensation. No definitive etio logical factors
filled blisters after a prodro me of pain and burning
have been found associated with A BH, but diabetes,
sensation. These blisters coalesced to form a large
inhaled long-term steroids, trauma, irregular margins
single bulla before rupturing. The bulla soon ruptured
of restorations, periodontal therapy, hereditary
leaving erosions. There was no history of similar
predisposition, may be associated in some instances.
blisters in other parts of the body. His past medical,
family and drug history were noncontributory. He
reported similar episodes of blood filled blisters
appearing on his cheek reg ion and lip region
previously which
ruptured leaving
ulcers. He
reported uneventful healing of the ulcers. Clin ical
examination revealed erosion on his right lateral
aspect of tongue (as shown in Figure 2) wh ich had
formed after rupturing of bulla. Tissue tags were
Literature review has shown soft palate to be the
noticed in relation to the same lesion. It was tender
most co mmon site for A BH. This is so because it is
on palpation but no bleeding was present. A sharp
covered by a thin friab le squamous epithelium of the
tooth cusp in relat ion to 46 (Shown in Figure 3) was
non keratinized type. [2, 5]
present. Absence of freshly developed vesicle or
Submucosal hemorrhage secondary to trauma can be
bulla ru led out biopsy in this case. Odontoplasty was
elicited easily in this site due to its fragile nature.
done at the time of presentation and he was reassured
Mastication causes increased blood flow to the soft
and recalled after 15 days. Healing was uneventful.
palate via parasympathetic reflex vasodilatation.
Thus overall soft palate is easily injured during
mastication of hard or crispy food and is prone to
DISCUSS ION
benign
ABH. [6] But apart fro m soft palate, lesions can also
oropharynx,
occur in other sites of the oral cavity among wh ich
characterized by sudden appearance of blood filled
tongue is most frequently involved. In our cases also
submucosal blisters of unknown etiology. It was first
ABH is present in relation to tongue. The most
‘Angina
condition
described
bullosa
hemorrhaegica’,
affecting
in
1933
mucosa
as
of
is
a
‘t raumat ic
probable etiology could be trau ma as it is bounded by
o ral
teeth.
hemophlyctenosis (TOH)’then the term ‘angina
bullosa hemorrhaegica (ABH)’ was coined by
Badham[3] for the same condition in 1967 and later it
In our first case, probable cause might be the
was renamed as ‘recurrent oral hemophlyctenosis
negative pressure. As patient is a yoga teacher, her
(ROH)’.
profession involves certain exercises which create
negative pressures within the oral cavity while
ABH is more co mmon ly diagnosed in middle-aged
performing them. And in the second case we have
and elderly population. Some patients may correlate
clearly mentioned the etiological factor as trauma
these bleeding episodes with meals or a prodro me of
fro m the sharp cusp tip adjacent to the lesion. We
54
Int J Dent Case Reports
March 2014, Vo l.4, No. 1
N Rakesh, Sh wetha V, Yashoda Devi B K, Deepa B Pat il
Angina bullosa hemorrhaegica
reassured the patients and educated them about the
As it is a benign disorder, no treat ment is required
condition and they are still on regular follow up
except for large pharyngeal bullae which may require
which revealed no recurrence of such lesions. In the
tracheostomy. So metimes NSAIDs or antimicrobials
second case no histopathological investigations were
as
carried out as at that instant it will not give much
symptomatic
informat ion regarding the lesion as there was no
infections. Identification of the etiological factor and
intact bulla and in vesiculobullous lesions clear fluid
its elimination may prove beneficial to the affected
filled bulla will be present.
individual.
On rev iew of literature we found that, the long term
CONCLUS ION
(> 5 years) use of steroid inhalers in asthamatic
ABH is a benign disorder and one should not be
patients has shown to induce mucosal atrophy and a
confused with other similar blood blistering diseases
[4,8]
as the treatment modality is entirely different. We
These sequential changes may evoke weakness and
being the oral diagnosticians play a vital role in
breakdown of capillaries, resulting in the format ion
diagnosing such lesions. As per our knowledge, in
of ABH. Similar morphologic alterat ions can also be
dental literature, there are very few cases of ABH
observed in middle aged adults which is the cause of
have been reported. This clearly demonstrates either
senile atrophy. This fact matches well with incidence
rarity of the disorder or the misdiagnosis.
decreased number of submucosal elastic fibres.
rinse
and
systemic
relief and
therapy
to
is
prevent
given
for
secondary
of ABH more widely in older ind ividuals . [4, 9]
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