A calibre-persistent labial artery (CPLA) is a primary arterial
branch that penetrates into the submucosal tissue of the lip without arborizing
or loss of caliber1. Gallard2 in 1884 was the first to describe about
abnormally large arteries within the gastric submucosa of 3 patients who died
of gastric haemorrhage. Initially these vascular anomalies occurring in the
stomach and jejunum were misdiagnosed as Arterial malformation, Crisoid
aneurysm and Dieulafoys disease3. Later in 1973 Howell and Freeman4 first described about this type of vascular anomaly in the oral cavity
and it was termed as “prominent inferior labial artery”. The current terminology calibre-persistent labial artery (CPLA)
was given by Miko et al in 19805.
The incidence of CPLA is approximately 3%4 with lower
lip being the most common site6. Few cases have also been reported
in the upper lip, palate and buccal vestibule 1,7. Most of these
patients were between 40 and 80 years of age.
CPLA can present as an ulcer, soft tissue papule or as a linear
pulsating nodule with bluish or normal mucosal colour. It is usually
asymptomatic and diagnosed during routine clinical examination. Occasionally
patient can notice an increased pulse volume at the site of the lesion that
could be visible or palpable. Patients with ulcers have reported with haemorrhagic
episodes too7,8. We report a
case of caliber persistent artery of the upper lip.
A 24-year-old male patient had been aware of a pulsatile nodule on
the left side of the upper lip for about 15 days. The nodule remained of the
same size and there wasn’t any history of trauma to the lip, bleeding or
interference in speech and mastication. His medical history was unremarkable
and he does not smoke.
Clinical examination revealed a slightly raised nodule measuring
3×3 mm on the left side of upper labial mucosa extending from the vermilion
border. It was sessile, the surface and colour appeared normal and pulsation
was seen. The lesion was soft, non- tender and pulsatile on palpation. The
remaining oral mucosa was normal. A provisional diagnosis of peripheral angiomatous
lesion was given. (Fig 1)
Colour Doppler Ultra sonographic examination was
performed from the mucosal side of the oral cavity. The examination was
performed using a compact linear transducer with a frequency range from 7 to 15
MHz. An arterial vessel with a tortuous course
was located on the left side of the upper labial mucosa. (Fig 2) The distance
between the artery and the surface mucosa was 0.9mm. The diameter of the artery
was 1mm near the periphery. (Fig 3) The peak systolic velocity was 58.06 cm/s and
end diastolic velocity was 11.87 cm/s while the pulsatality and resistivity
indices were 1.95 and 0.80 respectively. The spectral curve analysis showed
high flow velocity and high resistance with forward flow in both systole and
diastole and a dicrotic notch. (Fig 4) There was no evidence of arteriovenous
shunts or abnormal vascular communication or calcific deposits. Structures
surrounding the vessel appeared normal.
Immediate Surgical intervention was not recommended.
Caliber Persistent labial artery (CPLA) a vascular lesion in which
the primary artery will neither lose its caliber nor arborize after penetrating
the submucosal tissue 1.
In 1980 following a series of 3 cases Miko et al reported about
the extra gastrointestinal presentation of this lesion in the lower lip and
introduced the current terminology “caliber – persistent artery of the lower lip”. All three cases were
presented as chronic ulcers of the lower lip and were clinically misdiagnosed
as malignant ulcers and treated with wedge resection. However, microscopic
examination showed ulceration without any dysplastic features. It also showed
an artery with a large diameter, penetrating the orbicularis oris muscle
without division or loss of caliber representing a branch of inferior labial artery
Marshall and Leppard 9 in 1985 reported a case of
caliber persistent artery (CPA) in a 72-year-old male patient who presented
with chronic ulceration of the lower lip. A wedge lip excision was done as it
mimicked squamous cell carcinoma.
Miko et al5 and Marshall and Leppard 9
emphasised that CPLA should be considered when chronic inflammation and
ulceration are evaluated. Jaspers in 1992 1 reported 2 cases of CPA
with different presentations. The first case reported with chronic ulceration
of the hard palate with haemorrhagic episode. Following surgical removal of the
ulcer patient has experienced profuse haemorrhage from the surgical site. The
second case was an asymptomatic whitish – tan, nodular lesion involving the lower
lip that was clinically misdiagnosed as mucocele. There was “vigorous arterial haemorrhage” during surgical excision. Histopathological findings of both the
lesions were consistent with CPLA. Hence Jaspers suggested that CPLA can be
included in the differential diagnosis of soft tissue nodule as it can also
present without ulceration or inflammation 1.
The aetiology of CPLA remains
unclear. Different theories have been hypothesised to explain the aetiology of
this lesion. Firstly, Miko et al 5 suggested that senile atrophy of
the soft tissues caused senile ectasias of the blood vessels. Factors like trauma,
pressure from pipe stem, sun exposure and continuous pressure exerted by the
pulsating artery over the epithelium were linked as probable reasons for
chronic ulcers associated with CPLA. Although theory of aging was supported by Miko
et al 5 based on his case reports in elderly patients it is unlikely
that this is the reason for the occurrence, as CPLA’s were reported in young adults
too 8, 10. Our case was also diagnosed at the age of 24 years.
Kocyigit et al8 believed that CPLA’S are congenital
malformation of the blood vessels that is left unrecognized for a long time
until it becomes prominent. The theory of sun exposure as a
reason for mucosal erosion or degeneration ratifies for lesions occurring on
the lip. However, the same could not be validated for lesions that occurred in
buccal vestibule and palate 1,7.
Clinically CPLA can appear either
as a soft elevated non-ulcerated pulsatile solitary lesion which could be
linear or papular, located close to the mucosa having a unilateral presentation
or as chronic ulcers 1,4,5,6,8. Although pulsation serves as a key
feature in the clinical diagnosis of CPLA, non-invasive test such as colour
Doppler Ultrasonography can be used to confirm the diagnosis 8,9.
Vascular lesions like varicose
and pseudoaneurysm along with nonvascular lesions such as mucocele,
irritational fibroma and sclerosing sialadenitis can be considered in the
differential diagnosis of CPLA1,8.
Misdiagnosis can lead to vigorous
arterial haemorrhage when attempted with an excisional biopsy1,4,5,6,9.
Location of these lesion makes it susceptible to trauma and the ensuing
haemorrhage will require a surgical procedure to achieve haemostasis 1
CPLA can be diagnosed using reliable, real-time imaging method
like colour Doppler ultrasonography with high – resolution and frequency. This non-invasive
diagnostic method has several benefits over angiography as it permits to assess
the location and distance of the vessel from the mucosal surface, its diameter and
flow velocity through direct visualization, thereby reducing the risk of
complications that might occur following a surgical procedure. Compact liner
transducers that has the shape of a hockey stick enables better examination of
intraoral structures11, 12, 13, 14. Sonographic findings of the
present case were similar to that of the previously reported CPLA’s.
Commonality of CPA occurring in the stomach and lip ends with
profuse haemorrhage. Death following profuse haemorrhage has been reported in
CPA’s occurring in the gastrointestinal tract 2,3 however no
reported fatality following lip haemorrhage from CPLA.
Patients young age, no history of trauma or use of tobacco could
be included as a plausible cause for lack of ulceration or bleeding 8
in the present case. Treatment of choice varies with the clinical presentation
of the lesion. Conservative management and reassurance can be considered for
cases which are asymptomatic and surgical management for lesions that are
symptomatic and/or located at susceptible sites to trauma and /or unaesthetic
on patients’ demand. Confirmatory diagnosis should be made with Doppler
ultrasonography in order to avoid the brisk arterial haemorrhage associated
with excisional biopsy or surgery.
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lesion or caliber-persistent artery,” Archives of Pathology and Laboratory Medicine, vol. 130, no. 2,
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