Removal of Synchronous Bilateral Warthin’s Tumor in a Single sitting: A Case Report

Abstract

Warthin’s tumor constitute a minority of salivary gland neoplasms. It is a monomorphic adenoma commonly involving the parotid gland and considered to be unique because of its histological appearance, unknown origin and pathogenesis. Large, bilateral Warthin’s tumor of the parotid gland is clinically very rare. We are reporting the case of a patient having synchronous bilateral Warthin’s tumor with co –morbidities and operated for bilateral parotidectomy in a single sitting; which is very exceptional.

Introduction

Warthin's tumor, also known as papillary cystadenoma lymphomatosum or adenolymphoma of the parotid gland; is the second most common benign tumor of the parotid gland representing 2–6% of all tumors of the salivary glands [1]. Usually asymptomatic or may present as slowly growing tumor. It predominantly affects men (10:1) in the 5th and 6th decade [2]. Although the pathogenesis is unknown but there is a strong association with cigarette smoking [2]. Warthin's tumor is often multicentric and bilateral in 10 to 15% of cases. It comprises 70% of all bilateral salivary gland neoplasms [3]. Here we discuss a case of bilateral Warthin’s tumor removal via parotidectomy in a single sitting without any post-operative complication.

Case Report

A 68-years-old, male patient presented in Department of ENT, SBKS MIRC, with the history of bilateral swelling over the parotid region, gradually increasing in size over the past one year. On clinical examination, mobile, non-tender, firm swellings were palpated which measured approximately 2 × 3 cm and 3 × 4 cm on right and left side respectively. Patient is diabetic and hypertensive with history of chronic bidi smoking since last 40 years. The MRI scan showed approximately 28 × 29 x 33 mm and 32 × 31 x 45 mm sized well-defined heterogeneous altered signal intensity lesion in the superficial lobe of right and left parotid gland respectively. FNAC suggestive of salivary gland lesion with many mature lymphocytes along with amorphous and granular debris in the background. The MRI Scan and FNAC were in coherence with the diagnosis of Warthin’s tumor.

The patient underwent bilateral superficial parotidectomy under GA. A cervico-fascio-mastoid incision was kept over right parotid region and a superficial musculo-aponeurotic system (SMAS) advancement flap was prepared. Bilateral superficial parotidectomy encompassing the tumor was done with preservation of the integrity of all facial nerve branches and sent for histopathological examination. No immediate post-operative facial nerve palsy was observed. Delayed onset left sided grade 2 facial nerve palsy was recorded that eventually resolved within a week. The HPE confirmed the pre-operative diagnosis of Warthin’s tumor of bilateral parotid gland.

The patient was followed up one month and fifth month post-operatively and there weren’t any delayed onset complications seen (Figs. 1, 2).

Fig. 1.

Post contrast MRI scan showing bilateral parotid swelling

Fig. 2.

Facial nerve function immediate post-operatively and on follow up (after 1 month)

Discussion

Warthin’s tumor is the second most common tumor after pleomorphic adenoma which has the prevalence of 50% among all salivary gland tumors [4]. Smoking is a major risk factor for the developmentof Warthin’s tumor and in our case, the patient had been smoking 20 bidis a day for last 40 years.

Warthin’s tumor can occur bilaterally and such findings are a rare entity. As per the studies, it has been shown that this tumor was previously more common in men but recently no gender predilection is seen [5]. There are multiple theories to explain the origin and development of the tumor, but most accepted theory describes entrapment of salivary ductal cells in embryonic lymphocytic-rich tissues.

Patient usually presents with the history of a nodular lesion that is painless or a bit painful, growing slowly over the parotid region in front of the auricle. Due to their non-painful nature, such tumors are usually shun away by the patients. Macroscopically, the tumor is cystic and filled with a brown gelatinous liquid while microscopically findings are suggestive of epithelial parenchyma and lymphoid stroma, with amorphous cell groups and a mixture of lymphocytes, epithelial cells, oncocytes, and mast cells [6].

Treatment of Warthin’s tumor includes parotidectomy depending upon the involvement of the lobes. Postsurgical complications like facial nerve paresis/palsy and seroma are common while Frey’s syndrome, wound infections, dehiscence and hematomas are unusual. To salvage the facial nerves bilaterally in a single sitting parotidectomy requires utmost skill of the surgeon. Ethically, facial nerve on both sides of face are not exposed at once due to risk of palsy but surgeons should be prudent to protect the facial nerve and minimize its damage while performing any surgical intervention over the parotid gland bearing the landmarks and anatomy in mind. Synchronously operating bilateral parotids also reduced the risk of repeated anesthesia exposure in an old, morbid male patient.

As per Dong Hoon Lee et al. ten patients were studied retrospectively and thirteen parotidectomies were performed during the course of it. Patients with bilateral Warthin tumors in the parotid gland were divided into three groups according to the surgical methods used to treat these individuals. In group 1, the patients were closely observed without undergoing contralateral parotidectomy after unilateral parotidectomy. In group 2, the patients underwent concurrent bilateral parotidectomies. In group 3, the patient underwent contralateral parotidectomy 2 months after unilateral parotidectomy was performed. The overall rate of transient facial nerve dysfunction was 31%. Their findings suggested that concurrent superficial parotidectomy may be an appropriate method for treating bilateral Warthin tumors in the parotid gland, at least for desired patients. The symptoms of this type of tumor and physical examination findings were frequently non-specific and presented in the unilateral parotid gland. Therefore, a high degree of discernment was needed and imaging techniques were essential for the determining the correct pre-operative diagnosis [7].

Conclusion

Bilateral presence of Warthin’s tumor is a rare entity. They are non-painful, slowly growing and usually appears in large size. Bilateral tumors can be removed in a single sitting according to the prowess of the surgeon which avoids the additional risk of general anaesthesia of second stage surgery as well as preservation of bilateral facial nerve is of utmost importance in such cases.