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ORIGINAL ARTICLE
Year : 2023  |  Volume : 28  |  Issue : 5  |  Page : 415-420
 

Characteristics, surgical management, and outcomes of parotid gland masses in the pediatric age group: A single tertiary institute experience


Department of Paediatric Surgery, Institute of Child Health and Hospital for Children, Madras Medical College, Chennai, Tamil Nadu, India

Date of Submission06-May-2023
Date of Decision19-Jun-2023
Date of Acceptance13-Jul-2023
Date of Web Publication05-Sep-2023

Correspondence Address:
Raghunath Sambandam Murugan
D-1, I Floor, Saravana Poiegai Apartments, 42/8, School Road, Perambur, Chennai - 600 011, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.jiaps_100_23

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   Abstract 


Context: Parotid gland lesions in children requiring surgical management are not common. Neoplastic lesions of the parotid glands are also less common. Parotid tumors in children have different characteristics from those that occur in adults. When they occur in the pediatric age group, malignancy has to be ruled out.
Subjects and Methods: This is a retrospective study of children who presented to our institute, a tertiary care referral hospital for children <12 years, with parotid swellings during the 5-year period between April 2018 and March 2023. The children who underwent surgical management for parotid lesions, in the form of parotidectomy, were included in the study. Children who were treated by nonoperative management were excluded from the study.
Results: Twelve children were included. Of the 12 children, three (25.0%) children had malignancy, four (33.33%) children had benign tumors, three (25.0%) children had vascular malformations, and the remaining two (16.67%) children had inflammatory etiology. All children underwent superficial/total parotidectomy, depending on the involvement of superficial and/or deep lobe. Of the three malignant parotid tumors, two were of mucoepidermoid carcinoma and one was myoepithelial carcinoma. One of the children with mucoepidermoid carcinoma had recurrence.
Conclusions: Facial nerve-sparing parotidectomy is the treatment for neoplastic and inflammatory lesions. Initially, lymphovascular tumors were treated aggressively with parotidectomy. Neck node dissection should be performed only in children with fine-needle aspiration cytology-confirmed nodal metastases during primary surgery. Adjuvant treatment may be required in selected cases.


Keywords: Mucoepidermoid carcinoma, pediatric parotid lesions, parotid tumors, pleomorphic adenoma, superficial parotidectomy


How to cite this article:
Murugan RS, Maniam R, Dhanasekarapandiyan V, Hariharan G. Characteristics, surgical management, and outcomes of parotid gland masses in the pediatric age group: A single tertiary institute experience. J Indian Assoc Pediatr Surg 2023;28:415-20

How to cite this URL:
Murugan RS, Maniam R, Dhanasekarapandiyan V, Hariharan G. Characteristics, surgical management, and outcomes of parotid gland masses in the pediatric age group: A single tertiary institute experience. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 Oct 2];28:415-20. Available from: https://www.jiaps.com/text.asp?2023/28/5/415/385137





   Introduction Top


Salivary glands are actively functioning structures and throughout life, may undergo neoplastic change or be subject to various inflammatory conditions.[1] Among them, parotid gland lesions are most common. Some conditions may specifically affect children more notably than others, such as vascular malformations. Parotid gland tumors are rare in the pediatric age group.[2],[3] Among the general population, 5% of salivary gland tumors and 3% of all head-and-neck tumors occur in the pediatric population.[4] Pediatric parotid tumors constitute <1% of all pediatric neoplasms. They constitute <10% of all pediatric head-and-neck tumors. Although they are rare in the pediatric age group, they are common. However, when they occur, malignancy has to be ruled out, as higher percentage of malignancy occurs in the children.[5] The proportion of malignant parotid tumors in children is between 25% and 50%, compared to 15%–25% in adults.

Nonneoplastic parotid lesions, requiring surgical management, are less frequent in children. Pleomorphic adenoma and mucoepidermoid carcinoma are the most common benign and malignant parotid tumors, respectively. Childhood parotid tumors have different characteristics compared to that in adults. Apart from their rarity and higher proportion of malignancy, radiological diagnosis is difficult as imaging changes with age, as the gland is replaced by fat.

Malignant tumors, though occur in a higher proportion in children, are mostly of low-grade tumors. Hence, they are treated with facial nerve-sparing parotidectomy of the involved lobe.[6] Simultaneous neck node dissection is done only when suspicious nodes are present and are confirmed by preoperative fine-needle aspiration cytology (FNAC),[7] as occult nodal metastases is very rare and prophylactic elective neck dissection in N0 lesions have not been studied in the pediatric population.[8] Postoperative adjuvant treatment indications are also rarer, limited only to high grade, poorly differentiated lesions, recurrent lesions, node positive children, and in children who underwent R1/R2 resection.

Nonneoplastic lesions requiring parotidectomy are chronic sialadenitis and lesions that occur due to sialolithiasis. Sialolithiasis are rare in parotid glands.

This study is aimed to review the characteristics and outcomes of the parotid gland lesions, for which surgical management, in the form of parotidectomy, was done, in the pediatric population in our tertiary care referral center for children.


   Subjects and Methods Top


This was a retrospective study of children who presented to our institute, a tertiary care referral hospital for children <12 years, with parotid swellings and were diagnosed to have parotid lesions, during the 5-year period between April 2018 and March 2023. The children who underwent surgical management, in the form of parotidectomy, were included in the study. Children who were treated by nonoperative management were excluded from the study. Parotid lesions presenting with a mass lesion, as in parotitis and as in conjunction with metabolic disorders such as Sjogren syndrome, sarcoidosis, were excluded from the study. Children who underwent incision and drainage for suppurative parotid abscesses were also excluded from the study.

Children data were collected from the case sheets and the following were obtained and analyzed: age, gender, symptoms, clinical diagnosis, radiological imaging, FNAC, medical management given, surgery performed, postoperative facial nerve paralysis, postoperative histopathological report, any adjuvant treatment given, follow-up, and outcome (recurrence and its management). Data were analyzed. Quantitative data were described as numbers and percentages.

Proper ethical clearance was obtained from the institution ethical committee. Proper informed consent was obtained from the parent of the children for using their data and their images (clinical and intraoperative).


   Results Top


Twelve children were diagnosed to have parotid masses in the study period, fulfilling the study criteria, who needed surgical management and underwent parotidectomy. Summary of the clinicopathological features are provided in [Table 1]. Summary of all the 12 individual cases is given in [Table 2] and [Table 3] as malignant and nonmalignant cases, respectively. Of them, three were diagnosed to have malignant parotid tumors. Of the remaining children, two were of inflammatory etiology, three were lymphovascular malformations, and the remaining four were benign neoplastic lesions.
Table 1: Clinicopathological features of the children with parotid masses/lesions

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Table 2: Summary of malignant cases

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Table 3: Summary of the remaining nonmalignant cases

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Age

The range of age presentation was between 1 and 12 years. The median age at presentation was 9 years. Lymphovascular tumors presented early. Two of the children presented at 4 years of age. Another child presented at 10 years of age, but the symptoms predated for 7 years before presentation. Inflammatory etiology was present in both the extremes of the age group – 1.5 and 12 years.

Benign tumors presented in the latter half of childhood; three of them at 12 years and another one at 7 years of age. Malignant parotid tumors also occurred in the second half of childhood – 6, 8 and 11 years of age.

Gender

The parotid masses requiring surgical management were more frequent in the girls (eight vs. four in boys). Malignant tumors occurred more in the boys (two vs. one in girls). Nonlymphovascular benign tumors presented equally in both the sexes (two each). All three lymphovascular tumors presented in girls.

Side

Right side involvement was more common (seven vs. five in left). Among the malignancy diagnosed children, left was more common (two vs. one in right).

Symptoms

The universal presentation of all children was swelling of the parotid region for a long duration. Average duration of presentation in our study was about 4.3 months for noninflammatory, nonvascular neoplastic lesions. The vascular malformations presented with a longer duration of complaints, almost since birth and enlarged with time slowly.[9] One of the malignant children presented with swelling of 6 months duration with a sudden increase in the size of the swelling.

Pathological type

Among the twelve children with parotid masses in our study, three were lymphovascular tumors. One was parotid hemangioma, second one was intraparotid venous malformation, and the remaining one was parotid lymphangioma. Two were limited to the superficial lobe. Hemangioma involved in both the lobes.

In the remaining nine children, three were malignant, four were benign, and the remaining two were inflammatory (chronic sialadenitis). Among the malignant cases, two were mucoepidermoid carcinoma [Figure 1]. The remaining one was a high grade/poorly differentiated myoepithelial carcinoma, which was confirmed with immune histochemistry (positive for markers pan cytokeratin, vimentin, and smooth muscle actin).
Figure 1: 4X view Histopathological examination picture of mucoepidermoid carcinoma showing solid and cystic pattern with cystic spaces filled with mucin

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Of the four benign tumors, three were of pleomorphic adenomas [Figure 2] and one was schwannoma.
Figure 2: 10X view Histopathological examination picture of pleomorphic adenoma with epithelial and myoepithelial cells admixed with stromal component in the tumor portion

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Radiological imaging

All parotid tumor children initially underwent ultrasonography (USG) of the involved parotid region and the neck, along with the Doppler USG. All children were evaluated with magnetic resonance imaging (MRI) parotid region and neck, to look into the exact extent of the lesion, involvement of the deep lobe, and the involvement of the facial nerve, if malignant.

In some children, who presented with contrast-enhanced computed tomography imaging, done outside, we proceeded with surgery without MRI. MRI is the preferred imaging modality for parotid lesions [Figure 3]. Three children had involvement of the deep lobe: two in malignancy children and one in parotid hemangioma. No neck node involvement was present in the malignancy children.
Figure 3: Magnetic resonance imaging image showing hetero-dense lesion involving the superficial lobe of left parotid gland-turned out to be malignant tumor (mucoepidermoid carcinoma)

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Fine-needle aspiration cytology

All children with nonlymphovascular tumors then underwent FNAC. FNAC correlated with the postoperative HPE, in all, except one child, who was diagnosed with pleomorphic adenoma, but later turned out to be mucoepidermoid carcinoma. Sensitivity was 87.5%. Specificity was also 87.5%.

One child had suspicion of lytic lesion with soft tissue component over ramus and condyle of mandible, underwent incision biopsy, which showed spindle cell neoplasm, but turned out to be inflammatory etiology on final histopathological examination.

Staging

All three malignant cases were localized tumors, with no nodal metastases and no facial nerve involvement. In two cases, deep lobe was involved, which necessitated total parotidectomy.

Surgical management

All children underwent facial nerve-sparing parotidectomy, superficial or total, depending on the involvement of superficial and/or deep lobes, respectively. Three children required total parotidectomy. Of the three children, two were malignant and the remaining one was hemangioma, involving both the lobes.

After placing on table, child's entire ipsilateral face was exposed for monitoring of facial nerve function during surgery. If facilities for nerve stimulation and electromyographic monitoring are available, they should be used.

A modified Blair incision was made. Skin flaps were elevated in a subcutaneous fascial plane, between the superficial muscular aponeurotic system and the parotid fascia, to the anterior aspect of the parotid gland; care was taken not to extend the dissection too far anteriorly over the masseter where distal facial nerve branches become superficial.

The greater auricular nerve was divided. The posterior belly of digastric muscle was identified. Overlying parotid parenchyma was dissected off the digastric and sternocleidomastoid muscles. The facial nerve was usually identified 1 cm inferomedial to the tragal pointer and 0.5–1 cm deep to the tympano-mastoid suture line, at the level of attachments of the posterior belly of the digastric muscle.

Once the nerve was identified, dissection proceeded over and parallel to the direction of the nerve, with division of the overlying parotid parenchyma. Once the pes anserinus was encountered, dissection then proceeded in a step-wise fashion along the branches of the facial nerve in such a way that the superficial lobe and tumor were removed en-bloc. The child's face was closely monitored for movement to avoid injury to facial nerve and its branches. Every effort was made to preserve the nerves even in the case of malignancy. If the deep parotidectomy was needed, after dissecting and mobilizing the facial nerve and its branches, the deep parotid parenchyma was then removed from between the nerve branches. The retromandibular vein and the terminal branches of external carotid artery can be ligated and divided if needed. Medially, the deep lobe is bounded by the parapharyngeal space of fat, from which it is delivered by blunt dissection.

Complications

Four children had temporary facial nerve neuropraxia, which resolved in the due course of time. Among the four children, two were malignant cases and the remaining two occurred in the lymphovascular tumors.

Two children had recurrence of the lesion following surgery. One occurred in the malignant child (mucoepidermoid carcinoma), in whom wide local excision was done, with margins negative, confirmed on histopathological examination. The hemangioma-operated child had recurrence after 1 year, which was managed with bleomycin injection. All children with pleomorphic adenoma had no facial nerve paralysis and had no recurrence.

Adjuvant treatment

Two out of the three malignant children needed adjuvant therapy. Poorly differentiated myoepithelial carcinoma was treated with adjuvant chemotherapy with six cycles of vincristine, actinomycin-D, and cyclophosphamide.

Another child with lymph node positive (who had a benign diagnosis in FNAC, but malignancy in postoperative histopathological evaluation) underwent radiotherapy.

One child with mucoepidermoid carcinoma had recurrence, who was further evaluated and underwent wide local excision and was found to have margins negative, on second surgery. As it was a low grade, no adjuvant chemotherapy was needed.

Follow-up

There was no mortality among these 12 patients. All twelve children are on follow-up. Malignant children were followed up with the following protocol: initially, monthly for first 6 months, then alternate months for second 6 months, followed by third monthly in the third 6 months and then 6 monthly thereafter.

Recurrence, disease-free survival, and overall survival

The median follow-up duration for the above parotid lesions was 2.5 years (range, 0.3–5 years). One case of mucoepidermoid carcinoma had recurrence after 6 months of follow-up. Another child, who underwent total parotidectomy for hemangioma, had recurrence after 1 year.[10] The median recurrence was 9 months (range, 6–12 months). No recurrences occurred in the pleomorphic adenoma diagnosed and operated children. The recurrence rate was 33.33% for malignant cases (one in three) and none for benign neoplastic lesions (zero of four). Disease-free survival was 83.33% at 2 years. Overall-survival was 100%, as there was no mortality.


   Discussion Top


Parotid masses per se are less common in the pediatric age group. Benign and malignant parotid tumors are rare in the pediatric age group.[11],[12] The most common benign and malignant tumors are the pleomorphic adenoma and mucoepidermoid carcinoma.[4] Lymphovascular tumors also occur in this age group.

The study results were consistent with the typical presentation; lymphovascular tumors, being congenital, presented in childhood. In our study also, 25% of children had lymphovascular tumors. 25% of children with parotid lesions were malignant (three out of twelve). Among them, 66.67 % were mucoepidermoid carcinoma (two out of three). Malignancy was more common in the second decade of life.[13] Among nonlymphovascular tumors, three were of pleomorphic adenoma. Two were of inflammatory etiology. Girls were affected more than the boys, overall. Malignancy occurred more commonly in boys.

Myoepithelial carcinoma is a rare malignant salivary gland tumor among all salivary gland malignancies. They have a high recurrence rate reaching 40%. Hence, adjuvant treatment in the form of chemotherapy is advised. Radiotherapy is advised for better local control, if there were close/positive margins, lymph node(s) were positive or facial nerve was involved.

Neck node dissection is performed in node-positive children with malignancy, confirmed by FNAC. No prophylactic neck node dissection is advised, as there are negligible reports of occult nodal metastases in parotid malignancies in children. In our study, none of the malignant children had neck node metastases at the time of presentation, and hence, node dissection was not required.

Adjuvant chemotherapy should be given in children with high grade/poorly differentiated lesions, recurrent tumors, and in rarer lesions with higher chance of systemic spread as in adenoid cystic carcinoma.[14]

Adjuvant radiotherapy is given to children diagnosed to have node positive, facial nerve involvement and who had undergone R1/R2 resection.[15] In our study, one child was treated with adjuvant chemotherapy, because of the rarer, high-grade myoepithelial carcinoma. Another child needed adjuvant radiotherapy because of positive lymph node.[16]

Preoperative cytology (FNAC) is a must before proceeding with surgery. In our study, there was only one false negative for malignancy in FNAC.[17] FNAC is also considered for clinically or radiologically suspicious lymph nodes.

In our study, adverse event occurred in two children in the form of recurrence (one–malignant, another–hemangioma).[18],[19] Four children had temporary facial nerve neuropraxia.[20] Neuropraxia settled with conservative measures.


   Conclusions Top


Parotid gland masses/lesions requiring surgical management, in the form of parotidectomy is not common in the pediatric population. Parotid tumors among children are also rare, but they are not uncommon. Hence, when they do occur, malignancy has to be definitively ruled out. Parotidectomy, either superficial, deep or total (in accordance with the involved lobe[s]) with facial nerve preservation is the mainstay of treatment for parotid lesions. Due to their age, congenital vascular malformations are more common in the pediatric age group. Vascular malformations are better treated by nonoperative methods. FNAC is a prerequisite for all nonlymphovascular lesions prior to surgery.

Acknowledgment

Great thanks and warmest gratitude to the patients, their caretakers, and all department members in our hospital who helped to treat the children and to all who provided us with all we needed to work on the study cases effectively.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Krolls SO, Trodahl JN, Boyers RC. Salivary gland lesions in children. A survey of 430 cases. Cancer 1972;30:459-69.  Back to cited text no. 1
    
2.
Kanetake H, Inaka Y, Kinoshita I, Ayani Y, Ozaki A, Omura S, et al. Characteristics and Outcomes of Parotid Gland Tumors in Adolescents. Ear Nose Throat J. 2021:1455613211064013. doi: 10.1177/01455613211064013.  Back to cited text no. 2
    
3.
Gontarz M, Wyszyńska-Pawelec G, Zapała J. Primary epithelial salivary gland tumours in children and adolescents. Int J Oral Maxillofac Surg 2018;47:11-5.  Back to cited text no. 3
    
4.
Liu B, Liu JY, Zhang WF, Jia J. Pediatric parotid tumors: Clinical review of 24 cases in a Chinese population. Int J Pediatr Otorhinolaryngol 2012;76:1007-11.  Back to cited text no. 4
    
5.
Lee DH, Yoon TM, Lee JK, Lim SC. Clinical features of pediatric parotid tumors: 10-year experience of a single institute. Acta Otolaryngol 2013;133:1213-8.  Back to cited text no. 5
    
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Sultan I, Rodriguez-Galindo C, Al-Sharabati S, Guzzo M, Casanova M, Ferrari A. Salivary gland carcinomas in children and adolescents: A population-based study, with comparison to adult cases. Head Neck 2011;33:1476-81.  Back to cited text no. 6
    
7.
Rahbar R, Grimmer JF, Vargas SO, Robson CD, Mack JW, Perez-Atayde AR, et al. Mucoepidermoid carcinoma of the parotid gland in children: A 10-year experience. Arch Otolaryngol Head Neck Surg 2006;132:375-80.  Back to cited text no. 7
    
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Dell'Aversana Orabona G, Salzano G, Abbate V, Bonavolontà P, Committeri U, Seidita F, et al. Malignant tumours of the parotid gland: Management of the neck (including the clinically negative neck) and a literature review. Br J Oral Maxillofac Surg 2021;59:665-71.  Back to cited text no. 8
    
9.
Tsui SC, Huang JL. Parotid lymphangioma. A case report. Int J Pediatr Otorhinolaryngol 1996;34:273-8.  Back to cited text no. 9
    
10.
Harris J, Phillips JD. Evaluating the clinical outcomes of parotid hemangiomas in the pediatric patient population. Ear Nose Throat J 2021;100:P242-5.  Back to cited text no. 10
    
11.
Stevens E, Andreasen S, Bjørndal K, Homøe P. Tumors in the parotid are not relatively more often malignant in children than in adults. Int J Pediatr Otorhinolaryngol 2015;79:1192-5.  Back to cited text no. 11
    
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Rebours C, Couloigner V, Galmiche L, Casiraghi O, Badoual C, Boudjemaa S, et al. Pediatric salivary gland carcinomas: Diagnostic and therapeutic management. Laryngoscope 2017;127:140-7.  Back to cited text no. 12
    
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Xu B, Aneja A, Ghossein R, Katabi N. Salivary gland epithelial neoplasms in pediatric population: A single-institute experience with a focus on the histologic spectrum and clinical outcome. Hum Pathol 2017;67:37-44.  Back to cited text no. 13
    
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Goyal G, Mehdi SA, Ganti AK. Salivary gland cancers: Biology and systemic therapy. Oncology (Williston Park) 2015;29:773-80.  Back to cited text no. 14
    
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Janz TA, Camilon PR, Nguyen SA, Levi JR, Lentsch EJ. Has the management of pediatric mucoepidermoid carcinoma of the parotid gland changed? Laryngoscope 2018;128:2408-14.  Back to cited text no. 15
    
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Ryan JT, El-Naggar AK, Huh W, Hanna EY, Weber RS, Kupferman ME. Primacy of surgery in the management of mucoepidermoid carcinoma in children. Head Neck 2011;33:1769-73.  Back to cited text no. 16
    
17.
Suzuki M, Kawata R, Higashino M, Nishikawa S, Terada T, Haginomori SI, et al. Values of fine-needle aspiration cytology of parotid gland tumors: A review of 996 cases at a single institution. Head Neck 2019;41:358-65.  Back to cited text no. 17
    
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20.
Lennon P, Silvera VM, Perez-Atayde A, Cunningham MJ, Rahbar R. Disorders and tumors of the salivary glands in children. Otolaryngol Clin North Am 2015;48:153-73.  Back to cited text no. 20
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

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