A calibre-persistent labial artery (CPLA) is a primary arterialbranch that penetrates into the submucosal tissue of the lip without arborizingor loss of caliber1. Gallard2 in 1884 was the first to describe aboutabnormally large arteries within the gastric submucosa of 3 patients who diedof gastric haemorrhage. Initially these vascular anomalies occurring in thestomach and jejunum were misdiagnosed as Arterial malformation, Crisoidaneurysm and Dieulafoys disease3.
Later in 1973 Howell and Freeman4 first described about this type of vascular anomaly in the oral cavityand 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 lowerlip being the most common site6. Few cases have also been reportedin the upper lip, palate and buccal vestibule 1,7. Most of thesepatients were between 40 and 80 years of age. CPLA can present as an ulcer, soft tissue papule or as a linearpulsating nodule with bluish or normal mucosal colour. It is usuallyasymptomatic and diagnosed during routine clinical examination. Occasionallypatient can notice an increased pulse volume at the site of the lesion thatcould be visible or palpable.
Patients with ulcers have reported with haemorrhagicepisodes too7,8. We report acase of caliber persistent artery of the upper lip.CASE REPORT A 24-year-old male patient had been aware of a pulsatile nodule onthe left side of the upper lip for about 15 days.
The nodule remained of thesame size and there wasn’t any history of trauma to the lip, bleeding orinterference in speech and mastication. His medical history was unremarkableand he does not smoke.Clinical examination revealed a slightly raised nodule measuring3x3 mm on the left side of upper labial mucosa extending from the vermilionborder. It was sessile, the surface and colour appeared normal and pulsationwas seen. The lesion was soft, non- tender and pulsatile on palpation.
Theremaining oral mucosa was normal. A provisional diagnosis of peripheral angiomatouslesion was given. (Fig 1) Colour Doppler Ultra sonographic examination wasperformed from the mucosal side of the oral cavity. The examination wasperformed using a compact linear transducer with a frequency range from 7 to 15MHz. An arterial vessel with a tortuous coursewas located on the left side of the upper labial mucosa. (Fig 2) The distancebetween the artery and the surface mucosa was 0.9mm. The diameter of the arterywas 1mm near the periphery.
(Fig 3) The peak systolic velocity was 58.06 cm/s andend diastolic velocity was 11.87 cm/s while the pulsatality and resistivityindices were 1.95 and 0.80 respectively. The spectral curve analysis showedhigh flow velocity and high resistance with forward flow in both systole anddiastole and a dicrotic notch. (Fig 4) There was no evidence of arteriovenousshunts or abnormal vascular communication or calcific deposits. Structuressurrounding the vessel appeared normal.
Immediate Surgical intervention was not recommended. DISCUSSION Caliber Persistent labial artery (CPLA) a vascular lesion in whichthe primary artery will neither lose its caliber nor arborize after penetratingthe submucosal tissue 1.In 1980 following a series of 3 cases Miko et al reported aboutthe extra gastrointestinal presentation of this lesion in the lower lip andintroduced the current terminology “caliber – persistent artery of the lower lip”. All three cases werepresented as chronic ulcers of the lower lip and were clinically misdiagnosedas malignant ulcers and treated with wedge resection. However, microscopicexamination showed ulceration without any dysplastic features. It also showedan artery with a large diameter, penetrating the orbicularis oris musclewithout division or loss of caliber representing a branch of inferior labial artery5. Marshall and Leppard 9 in 1985 reported a case ofcaliber persistent artery (CPA) in a 72-year-old male patient who presentedwith chronic ulceration of the lower lip.
A wedge lip excision was done as itmimicked squamous cell carcinoma.Miko et al5 and Marshall and Leppard 9emphasised that CPLA should be considered when chronic inflammation andulceration are evaluated. Jaspers in 1992 1 reported 2 cases of CPAwith different presentations.
The first case reported with chronic ulcerationof the hard palate with haemorrhagic episode. Following surgical removal of theulcer patient has experienced profuse haemorrhage from the surgical site. Thesecond case was an asymptomatic whitish – tan, nodular lesion involving the lowerlip that was clinically misdiagnosed as mucocele. There was “vigorous arterial haemorrhage” during surgical excision. Histopathological findings of both thelesions were consistent with CPLA. Hence Jaspers suggested that CPLA can beincluded in the differential diagnosis of soft tissue nodule as it can alsopresent without ulceration or inflammation 1.
The aetiology of CPLA remainsunclear. Different theories have been hypothesised to explain the aetiology ofthis lesion. Firstly, Miko et al 5 suggested that senile atrophy ofthe soft tissues caused senile ectasias of the blood vessels. Factors like trauma,pressure from pipe stem, sun exposure and continuous pressure exerted by thepulsating artery over the epithelium were linked as probable reasons forchronic ulcers associated with CPLA. Although theory of aging was supported by Mikoet al 5 based on his case reports in elderly patients it is unlikelythat this is the reason for the occurrence, as CPLA’s were reported in young adultstoo 8, 10. Our case was also diagnosed at the age of 24 years.
Kocyigit et al8 believed that CPLA’S are congenitalmalformation of the blood vessels that is left unrecognized for a long timeuntil it becomes prominent. The theory of sun exposure as areason for mucosal erosion or degeneration ratifies for lesions occurring onthe lip. However, the same could not be validated for lesions that occurred inbuccal vestibule and palate 1,7.Clinically CPLA can appear eitheras a soft elevated non-ulcerated pulsatile solitary lesion which could belinear or papular, located close to the mucosa having a unilateral presentationor as chronic ulcers 1,4,5,6,8. Although pulsation serves as a keyfeature in the clinical diagnosis of CPLA, non-invasive test such as colourDoppler Ultrasonography can be used to confirm the diagnosis 8,9. Vascular lesions like varicoseveins, hemangiomaand pseudoaneurysm along with nonvascular lesions such as mucocele,irritational fibroma and sclerosing sialadenitis can be considered in thedifferential diagnosis of CPLA1,8.
Misdiagnosis can lead to vigorousarterial haemorrhage when attempted with an excisional biopsy1,4,5,6,9.Location of these lesion makes it susceptible to trauma and the ensuinghaemorrhage will require a surgical procedure to achieve haemostasis 1CPLA can be diagnosed using reliable, real-time imaging methodlike colour Doppler ultrasonography with high – resolution and frequency. This non-invasivediagnostic method has several benefits over angiography as it permits to assessthe location and distance of the vessel from the mucosal surface, its diameter andflow velocity through direct visualization, thereby reducing the risk ofcomplications that might occur following a surgical procedure. Compact linertransducers that has the shape of a hockey stick enables better examination ofintraoral structures11, 12, 13, 14. Sonographic findings of thepresent case were similar to that of the previously reported CPLA’s. Commonality of CPA occurring in the stomach and lip ends withprofuse haemorrhage. Death following profuse haemorrhage has been reported inCPA’s occurring in the gastrointestinal tract 2,3 however noreported fatality following lip haemorrhage from CPLA.
Patients young age, no history of trauma or use of tobacco couldbe included as a plausible cause for lack of ulceration or bleeding 8in the present case. Treatment of choice varies with the clinical presentationof the lesion. Conservative management and reassurance can be considered forcases which are asymptomatic and surgical management for lesions that aresymptomatic and/or located at susceptible sites to trauma and /or unaestheticon patients’ demand. Confirmatory diagnosis should be made with Dopplerultrasonography in order to avoid the brisk arterial haemorrhage associatedwith excisional biopsy or surgery. 1 Jaspers MT. Oral persistent-persistent artery. Unusualpresentations of unusual lesions. Oral Surg.
Oral Med. Oral Pathol. 1992; 74:631–3. 2. Gallard T: Miliary aneurysm of the stomach giving cause tofatal hematemesis. Bull Mem Soc Med hop Paris,1884; 1: 84– 913. J.
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4. Howell JB, Freeman RG: Prominent inferior labial artery. ArchDermatol 107:386, 1973 5. Miko T, Adler P, Endes P.
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