Revealing Enigma in Parotid Gland: Venolymphoma—A Rare Case Report

Abstract

Venolymphoma (VL) is a benign condition of oral cavity, but it is misdiagnosed as pleomorphic adenoma. Exact diagnosis can be made only after histopathological examination. In this report the author has presented a case of a 45-year-old male patient with VL of right parotid which was operated. The treatment indicated was complete excision of the lesion, which showed an excellent prognosis with low recurrence rate.

Introduction

Venolymphoma (VL) is a benign condition of oral cavity, but it is rarely present in the parotid gland. It manifests since childhood occurs in early age as congenital soft painless presentation. There are very few cases available that are proved to have VL with oral pathology as patients directly visit the ENT surgeon [1]. Most of the time this condition can be misdiagnosed as pleomorphic adenoma. Exact diagnosis can be made only after histopathological examination [2]. Swelling may be present at birth; it enlarges with as the patient grows and never regresses. Surgery on parotid is challenging because of the presence of facial nerve. In this case, VL was presented with well-defined borders and it became necessary to preserve the surrounding structure as it had a tendency to infiltrate the surrounding tissue making complete resection of the lesion a necessity in order to prevent its recurrence [3].

Case Report

A 45-year-old male patient reported to the department of oral surgery complaining of swelling on the right side of his face since childhood. On inspection, extraoral examination showed asymmetry of the face on the right side with obliteration of the nasolabial fold and elevation of the right nasal alar region. The swelling was measuring about 10 × 8 cm in size extending from right external ear pinna to 1 cm from the inferior border of the mandible (supero-inferiorly) and from the angle of the mandible till 6 cm from the midline of the chin (antero-posteriorly) and had a similar coloration to the adjacent mucosa (Fig. 1). On palpation, the lesion was soft with fluctuating consistency and non-tender with no local rise in temperature. The clinical hypothesis was made as pleomorphic adenoma. Ultrasonography (USG) of local swelling was suggestive of pleomorphic adenoma. In magnetic resonance imaging finding, the lesion was seen compressing the right internal jugular vein. In addition to this, fine-needle aspiration cytology was performed suggestive of no epithelial component with no diagnostic pathology.

Fig. 1.

The soft and fluctuating swelling on right side of face

Patient was taken for surgery under general anaesthesia. Modified Blair incision was marked on the right side. Exploration around parotid gland was completed, and methylene blue dye was injected in the lobe of parotid. Pulsation around the facial artery was felt, and internal jugular vein was appreciated. The submandibular component of swelling was removed. The larger portion of the swelling over parotid gland was removed. With the help of nerve stimulator, care was taken to avoid any injury to facial nerve branches (Fig. 2).

Fig. 2.

The use of nerve stimulator over facial nerve

Histopathology

Venolymphoma was confirmed by the histopathological examination (Fig. 3). The section stained with the help of hematoxylin and eosin (H&E) staining technique showed salivary gland acini with lipomatic and oncocytic changes in parenchyma. Many lymph nodes were evident in the surrounding capsule. It also showed the presence of dilated venules lined by flat endothelial layer and lymphatic vessels along with patchy smooth muscle in the wall making dilated duct and blood vessels evident. The vessels filled with eosinophilic proteinaceous material were also seen indicating lymphatic vessels. Overall features were suggestive of lipomatosis of salivary gland.

Fig. 3.

The H and E stained section of Venolymphoma

Discussion

Venolymphoma lesions are rare in oral cavity and are even rarer in parotid gland. These are slow-flow and slow-growing lesions made up of lymphatic and venous components and are present since birth caused by a disturbance in the late stages of angiogenesis. Imaging techniques, such as ultrasonic colour Doppler, can aid in diagnosis. Vascular malformations cause abnormalities of the surrounding soft tissue, leading to functional as well as aesthetic impairments. The size, location, and proximity to the vital structures are all contributory factors for the treatment of VL [4]. Surgical treatment, i.e. total resection of lesion while preserving the facial nerve, is the most opted management approach [5]. Recently, 70 cases were analysed by Starkman et al. [5–7] which accounted to be the largest case series till date, showing 90% of lipomatous tumour of parotid gland as lipomas, and the remainder as variants and malignant tumours. Achache [6] found 10 cases of parotid VMs in 614 parotidectomies. Beahrs et al. and Byars et al. [4, 5, 8] reported incidence only 0.5% and 0.6%, respectively, in their study of a large number of parotidectomy specimens with a female predilection. An excisional biopsy is usually carried out as the lesion can be easily removed during the procedure, and despite the vascular component, there is no excessive bleeding during the procedure [7–10]. Depending on the nature of the lesion, the treatment planned by the author for this patient was surgical resection of the malformation. Patient was kept on follow-up for 1 year post-operatively, and there was no sign of any recurrence of swelling.

Conclusion

The clinical features of oral VL show similarity to other commonly seen lesions, which makes it difficult to diagnose pre-operatively. The treatment indicated was complete excision of the lesion, which showed an excellent prognosis with low recurrence rate.

Declarations

Conflict of interest

The authors declare that they have no conflict of interest.