Acquired venous vascular malformation of the lip: A rare Case report.

Omar N. Elprince^1*, Noora . Alshammari¹, Saud F. Alhazzaa² and Faleh A. Alshahrani²

^*Correspondence: Omar N. Elprince, Department of Oral and Maxillofacial Surgery, Vision Colleges of Dentistry and Nursing, Saudi Arabia, Email:

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Introduction

Vascular malformations constitute a broad spectrum of pathologies involving the vascular structures of the human body. The diagnostic complexity and the pathological management accordingly have been varied largely due to its varied spectrum of presentation. Management for vascular malformation has been reported to be varied from medical to minimally interventional and finally radical modalities [1]. In 2011, Nair et al [2]. Introduced a comprehensive classification of vascular malformation. The vascular malformations were categorized into five categories according to their anatomy and depth of location in the head and neck region. Type I is superficial lesions requiring excision of skin or mucosa, local or regional flaps have been used in defect reconstruction. Type II contains the sub mucosal lesions which require complete excision after elevation of skin flaps. Type III is the lympho-venous malformations or venous malformations involving glands of the head and neck are excised along with the affected gland. Type IV which contains intraosseous lesions requires excision with affected bone and reconstruction when required. Type V lesions involving deep visceral spaces, such as the Para pharyngeal or infratemporal fossa, which require skeletal osteotomy for appropriate exposure and excision.

According to Forbes et al. vascular malformations can also be classified into slow/low flow and high/ fast flow lesions based on hemodynamic and contrast angiographic appearance. Lymphatic and venous malformations belong to the first group, while arterovascular malformation belong to second group [3].

The diagnostic modalities for vascular malformation start with the clinical diagnosis which contains a thoroughly examination of shape, consistency and character delineation and possible invasive lesion, with subsequent deterioration of vital functions [1]. Imaging modalities such as Color Doppler UltraSound (CDUS), MRI, CECT scan, Digital Subtraction Angiography (DSA) (catheterizing the entire vertebral or vascular tree) and MR venography for low-flow lesions has been reported to aid in diagnosis. MRI sectional images can avoid signal overlap and thereby demonstrate the relationship between the lesion and the deep structure. MRI is therefore superior to CT in demonstrating the relationship between the extent of the lesion and normal tissue [4,5].

During histopathological interpretation of vascular malformations, it is of paramount importance to distinguish between the vascular malformation and hemangiomas, this can be attributed to the fact that management of hemangioma differs from treatment of vascular malformation [6]. In distinction to hemangiomas, vascular malformations result from abnormal vascular or lymphatic morphogenesis, not due to abnormal endothelial proliferation. Despite some clinical similarities in appearance between vascular malformations and hemangiomas, the histopathologic features are quite different. Usually, the vascular malformations have the following: no endothelial cell proliferation contains large vascular channels lined by endothelium, unilamellar basement membrane, not incorporating tritiated thymidine in endothelial cells, and normal mast cell counts [7].

Case Report

A 33 years old female came to the hospital with swelling related to left area of upper lip that developed 3 years ago after an accident (Figure 1). The cause of trauma was reported by the patient to be due motor vehicle accident. The patient had not received any treatment at that time and the injury healed spontaneously. The patient was asymptomatic & lesion was painless with increasing in its size during the past 3 years. Upon clinical examination, the lesion was found to be mobile, rubbery, and non-tender. Additionally, the volume of lower lip was slightly enlarged by the Valsalva maneuver on physical examination. This finding has directed us to suspect vascular anomalies. An MRI has been done for further investigations. The MRI images showed an increased vascular activity in the affected area (Figure 2).

Figure 1. A clinical image showing swelling related to left side of the upper lip.

Figure 2. MRI investigations. (A) contrast-enhanced MRI T1 image shows intensely enhancing localized lesion involving the left upper lip area ,(B) Contrast-enhanced MRI T1 axial image shows intensely enhancing localized lesion involving the left upper lip area showing voids suggested low blood flow in blood vessels and phleboliths.,(C) A T2-weighted, fat-saturated sagittal magnetic resonance image shows a hyper-intense lesion with flow voids within upper lip in left area, (D) Three dimensional MRI reconstructive images shows the lesion does not derive its vascular supply through superior labial artery (arrow).

Diode laser (sirolaser machine, wavelength 970 nm) was done as minimally invasive surgery. The power was 3 W, Continoues wave mode, interstitial tip inside a lesion for five visits. The swelling was shrinking but stil laser cannot reach to base of swelling because the maximum depth of penetration is 3-4 mm. surgical excision was done by scalpel as excisional biopsy and employment of hemostatic measures such as corset suturing has been made. Afterwards, we send the excisional biopsy for pathological investigations (Figure 3). Upon pathological examination, photomicrographs showed numerous blood vessels within the muscular layer with normal endothelial lining in conjunction with dilated venules and arterioles. The final pathological diagnosis commensurate with a vascular malformation (Figure 4). The follow up phase (A) after five visits of laser, (B) after surgical excision, and (C) three months postoperatively (Figure 5).

Figure 3. After surgical removal, the specimen kept in labeled

Figure 4. Hematoxylin and eosin-stained sections showing numerous blood vessels within the thickened muscular layer with flat endothelial lining in conjunction with dilated venules.

Figure 5. Follow up phase. (A) After five visits of laser, (B) surgical excision postoperatively, (C) 3 months postoperatively.

Discussion

The lip is the most common site for the occurrence of many types of vascular lesions, this can be attributed to the fact that lip has relatively good blood supply owing to the presence of large blood vessels approximating the surface at this location than the other parts, and that the lip is often subjected to traumatic insults [8]. Vascular anomalies of the head and neck region are challenging in diagnosis and treatment. From the distinction between vascular malformation and hemangioma to the management and treatment is quiet confusing [1]. Vascular malformation might be congenital or acquired. Acquired vascular malformations are quite rare in head and neck region and most of the reported cases are related to a traumatic etiological factor [9].

In the present case, the history of trauma as well as the enlargement of the lesion with Valsalva maneuver during clinical examination, and absence of lesion during birth has raised speculations regarding the development of vascular malformation. The decision for MRI was made to continue our investigation. As according to Manjunath et al [3]. MRI was found to be superior diagnostic tool when compared to other diagnostic radiography as it precisely depicts the extent of lesion as well as differentiate between types of vascular malformations.

Upon MRI investigation, the contrast enhanced MRI has showed intensely enhancing localized lesion involving the left upper lip area contains voids suggested low blood flow in blood vessels and phleboliths. This finding commensurate with a law flow venous vascular malformation. Additionally, the communication between the superior labial artery and the lesion has not been identified. The lack of communication between a feeding artery and the vascular malformation as well as the appearance of phleboliths has guided us towards confirming the diagnosis of venous malformation and excluding the diagnosis with arteriovenous malformation.

Many theories have been proposed regarding the etiology of venous malformation. Sun exposure, smoking, genetics or traumas are all among the etiological factors to develop this lesion [10]. In our present case report, the cause of this lesion is highly likely to be from the lip trauma that caused this vascular malformation.

Considering the small size of the lesion and its slow flaw nature, a decision for a surgical excisional biopsy has been made without embolization. This was in accordance with Jeong et al [11], who demonstrated in their retrospective study an algorithm to approach vascular malformations of the lip. During the surgical procedure the hemostatic measures were made employing corset sutures. Nair, et al [12]. have showed that corseting technique in management of hemorrhage during surgical procedure for law flow of vascular malformation is an effective method. Furthermore, the use of Diode laser was chosen due to its reported superior efficacy on treatment of small oral vascular malformations [13].

The histopathological findings have confirmed the diagnosis of vascular malformation with an inclination towards suggesting of a venous type. The lab finding showed numerous blood vessels within the thickened muscular layer with flat endothelial lining in conjunction with dilated venules and arterioles. As stated by Miller et al, our findings came along with their description of a venous vascular malformation [14, 15].

Conclusion

In conclusion, traumatic events can lead to venous vascular malformation of the lip. Additionally, MRI along with a thoroughly clinical examination and patient’s history recording is mandatory to reach a proper diagnosis of vascular malformation types.

Financial Support and Sponsorship

Nil

Conflicts of Interest

There are no conflicts of interests.

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