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 Table of Contents  
ORIGINAL ARTICLE
Year : 2021  |  Volume : 9  |  Issue : 2  |  Page : 147-152

Minor salivary gland neoplasm of the larynx: Our experience


Department of ENT-Head and Neck Surgery, Kasturba Medical College, Mahe, Manipal, Karnataka, India

Date of Submission13-Sep-2021
Date of Decision16-Oct-2021
Date of Acceptance16-Oct-2021
Date of Web Publication17-Dec-2021

Correspondence Address:
Dr. C Jalwa Ahmed
Department of ENT-Head and Neck Surgery, Kasturba Medical College, Manipal, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jhnps.jhnps_53_21

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  Abstract 


Introduction: Minor salivary gland neoplasm of larynx is a rare tumor of head and neck and account to around 1% of laryngeal cancers. This is a disease of middle age and affects subglottis mostly than other parts of the larynx. Preferred treatment is total laryngectomy (TL) due to high propensity for submucosal spread. In this case series, we discuss our experience in the management of salivary gland tumors of larynx. Methods: Retrospective analysis of five patients of salivary tumors of larynx treated at our institute for over 30 years. Results: Among the five patients, four had Adenoid cystic carcinoma (ACC) and one had clear cell carcinoma (CCC). Four ACC patients had the lesion in the subglottis and 3 underwent TL with chemoradiation and had no recurrence till latest review after 32 months of surgery. However, one patient who underwent chemoradiation alone had a persistent disease and is receiving palliative care. CCC patient had lesion in the glottis and underwent hemilaryngectomy and had no recurrence till 15 years following surgery. No locoregional metastasis identified, though one patient had lung metastasis. Discussion: ACC is the most common salivary tumor seen in the larynx. Literature had shown male predominance, however, our study had shown no sex predilection. In our study, patients who underwent TL with adjuvant chemoradiation showed no recurrence as compared to one patient who underwent chemoradiation alone and had disease progression. Conclusion: ACC is the most common salivary gland neoplasm of larynx and surgical treatment with chemoradiation seem to provide better outcome than chemoradiation alone.

Keywords: Adenoid cystic carcinoma, clear cell carcinoma, laryngeal carcinoma, minor salivary gland neoplasms of larynx


How to cite this article:
Nayak DR, Balakrishnan R, Ahmed C J. Minor salivary gland neoplasm of the larynx: Our experience. J Head Neck Physicians Surg 2021;9:147-52

How to cite this URL:
Nayak DR, Balakrishnan R, Ahmed C J. Minor salivary gland neoplasm of the larynx: Our experience. J Head Neck Physicians Surg [serial online] 2021 [cited 2022 Aug 17];9:147-52. Available from: https://www.jhnps.org/text.asp?2021/9/2/147/332723




  Introduction Top


Laryngeal malignancies fall among the 10 most common cancers in the Indian men with a wide range of regional variations. According to a review study by Bobdey et al. in 2015, incidence of laryngeal cancers in India accounts to 3%–6% of cancers among men which is equivalent to 1.26–8.18 per 1 lac population.[1] Thus, it becomes the ninth most common cancer among men in Asia and seventh in India (1).[1] However, minor salivary gland neoplasms of larynx are far rarer[2] and it accounts to only 1% of all laryngeal cancers, i.e., 0.005/100,000. The wide range distribution of the minor salivary glands from oral cavity till larynx could increase possibility of developing these neoplasms at rarer locations. Commonly these are malignant, most common being adenoid cystic carcinoma (ACC) (32%–69% of minor salivary gland neoplasm of larynx),[3],[4] followed by mucoepidermoid carcinoma (15%–35%).[3] Other varieties like clear cell carcinoma (CCC) are much rarer (1%–2% of minor salivary gland neoplasm of larynx). Along with tobacco and alcohol, indoor air pollution and spicy food habits play a significant role in laryngeal squamous cell carcinoma (>90%).[1] However, associated risk of the same in case of minor salivary gland neoplasm is rarer, up to 50%.[3] These are predominantly slow growing tumors due to the submucosal spread[2] and many a times, they have an advanced stage presentation with perineural invasion due to the slow growing pattern.[3],[4],[5] The most common location observed varies between supraglottis[4] and subglottis[3],[5] among different studies. Contrast-enhanced computerized tomography of neck and thorax stands the most common imaging modality to assess the site and extent of the disease.[3],[4],[5] These are predominantly disease of fifth to sixth decade.[3],[4] Sex predilection of these malignancies had also been found to be varying in different studies. However, majority of them agree on the female predilection.[3] Like other malignancies of larynx, the primary modality of management is surgical resection and adjuvant radiotherapy (+/-chemotherapy)[3],[4],[5] with average 5 years survival rate of 42%,[2],[3] which is more than other malignancies of larynx (28%-5 years survival rate).[1] Primary surgical management is the preferred treatment due to the perineural invasion of these malignancies.[3] When locoregional metastasis and recurrence was studied, it was found out that up to 60% of the minor salivary gland tumors of larynx cause locoregional metastasis or recurrence,[3],[5] except with ACC. The later showed higher chances of distant metastasis, which is the most common cause of failure of treatment,[2] with the most common site being lung.[3],[5] Interestingly, recurrences were higher on treatment with primary radiotherapy rather than with primary surgical management.[3] However, it must be noted that there has only been limited number of studies in the past on this subject due to its rarity and hence this case series is intended to detail our experience in diagnosing and treating minor salivary gland neoplasm of larynx at our tertiary care center.


  Methods Top


This was a retrospective study in our tertiary health care center. The aim of the study was to share our experiences and the lessons learned from treating the patients with minor salivary gland neoplasm of larynx at our institute and to compare our results with the existing literature. All patients diagnosed and treated for minor salivary gland neoplasm of larynx in our center over the last 28 years, from January 1991 to October 2019 were included in this study. In the process, we could recruit 5 subjects. The details were collected from the medical records available in the institute after ethical clearance from Institutional Ethical Committee. Collected data include the age and sex at presentation, primary site of the lesion, their clinical presentation with staging and pathological diagnosis. Also, the primary modality of the treatment of each of the subject and adjuvant therapy, recurrence or residual lesion and latest follow ups were noted. In addition, postoperative status of resected margins, lymphovascular and perineural invasion were also collected for subjects who received surgical management. Thus, the primary outcomes of the treatment including the survival or disease progression were compared with the existing literature.


  Results Top


Five subjects diagnosed with minor salivary gland neoplasm of larynx were recruited for the study. Four among the five subjects had ACC (80%) of larynx while, one subject had CCC (20%) [Table 1]. Subglottis was the subsite of the lesion for 4 subjects (80%) [Table 1] and hence the most common clinical presentation was noisy breathing i.e., 3 out of 5 subjects (60%), associated with hemoptysis or voice change depending on the extent of the lesion. It was notable that subglottis was the subsite for all 4 ACC subjects. However, CCC had the primary lesion at glottis and presented with voice change. All 4 subjects with lesion in the subglottis had an advanced stage of presentation (Stage III or IV) with extension into trachea or glottis. Although we noted equal sex predilection in cases with ACC (2 each among female and male), CCC was diagnosed in a male. Hence 3 out of 5 subjects (60%) were male. All the four ACC subjects were at the fourth to fifth decade of life while CCC presented in second decade of life. Hence 80% subjects in our study had disease diagnosed in the fourth to fifth decade of life. Imaging was done by contrast-enhanced computerized tomography of neck, extending from dura to pleura (Skull base to superior mediastinum) for all subjects [Figure 1]. Primary surgery was performed for 4 subjects (80%), 3 of them had ACC and 1 had CCC [Table 2]. Primary surgery in the form of total laryngectomy (TL) with neck dissection followed with adjuvant chemoradiation was performed for the 3 subjects with ACC [Figure 2] and [Figure 3], while the subject with CCC received only primary surgery in the form of vertical hemilaryngectomy [Figure 4]a, [Figure 4]b, [Figure 4]c and [Table 2]. No recurrence or residual lesion was seen in any of these subjects with the latest follow up of 15 years for one of these subjects. One subject with ACC (20%) underwent primary chemoradiation and was noted to have a residual lesion in the trachea at 9 months follow up [Table 2]. Interestingly, the later also had metastatic lesion in the lung at 9 months follow up whereas none of the former mentioned had any evidence of locoregional or distant metastasis. The pathological diagnosis and staging were confirmed by the resected specimen. Perineural invasion was present in 3 out 4 resected specimens, all of which were ACC. None of the specimen examined had lymphovascular invasion [Table 3].
Figure 1: Contrast-enhanced computed tomography scan of neck, lateral image showing the extension of mass

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Figure 2: Intra-operative image of total laryngectomy in subjects with adenoid cystic carcinoma of larynx

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Figure 3: Total laryngectomy specimen in case of adenoid cystic carcinoma larynx

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Figure 4: Hemilaryngectomy with sternohyoid repositioning in case of clear cell carcinoma of larynx. (a) Clear cell carcinoma of glottis. (b) Hemilaryngectomy intraoperative image. (c) Resected specimen of clear cell carcinoma of larynx

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Table 1: Clinical features of minor salivary gland neoplasm of larynx in our study

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Table 2: Management and outcomes of minor salivary gland neoplasm of larynx in our study

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Table 3: Pathologic staging of minor salivary gland neoplasm of larynx in our study

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  Discussion Top


Though laryngeal malignancies is the ninth most common carcinoma among men in Asia and seventh most common carcinoma among men in India,[1] minor salivary gland neoplasm of larynx accounts to only <1% of all laryngeal malignancies.[3],[5] Due to the rarity of the disease, it is difficult to formulate a study and hence there have been only few studies on minor salivary gland neoplasms of larynx. In addition, the existing studies also showed varying results.

Cohen et al. in 1985 studied the minor salivary gland neoplasm of larynx diagnosed and treated at their institute over a period of 38 years (1944–1982). This was one of the first studies on this subject available. They recruited 18 subjects of which 8 were ACC and 10 had adenocarcinoma. Hence according to their conclusion most common minor salivary gland neoplasms of larynx is adenocarcinoma.[2] However, this was opposed by many studies done later on. All of them concluded ACC as the most common minor salivary gland neoplasms of larynx.[3],[4],[6] This was comparable in our study also, which also showed ACC to be the most common among minor salivary gland neoplasms of larynx. However, we noted one subject had CCC, which was not noted in many study in the past[7],[8] [Table 1].

On studying the subsite of the lesion, it was noted that they can have their origin from any subsite of larynx.[2] Hence there were studies to locate the most common subsite. The results again varied between different studies. When Ian Ganly et al. in 2006 concluded subglottis as the most common subsite,[3] which was supported by other studies,[5],[6],[9],[10] our experience was also not different. Eighty percent of our subjects i.e., 4 out of 5 subjects had lesion in the subglottis. However, a study by Bishop et al. in 2016 concluded supraglottis to be the most common subsite[4] [Table 4]. Though the subsites may vary, the lesion is always submucosal in origin and hence presents as a smooth bulge or infiltrating growth.[2] This could be because of the submucosal location of seromucinous glands in the larynx, particularly in the supraglottis and subglottis.
Table 4: Comparison between similar studies in the past

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Similarly, varying results could be noted in case of sex predilection of minor salivary gland neoplasms as well. When some studies showed more female predilection,[3] some studies could not find any sex predilection.[4],[10] Our study, however, had male predilection for minor salivary gland neoplasms of larynx in general and absence of any sex predilection among patients with ACC [Table 4]. Hence there is still a necessity for further long-term studies on this subject.

Minor salivary gland neoplasms of larynx are generally noted as a disease of fifth to sixth decade of life.[6],[10] Study in 2006 by Ian Ganly noted the mean age of presentation of minor salivary gland neoplasms of larynx as 54 years.[3] Similar results were obtained by Bishop et al.[4] Although the mean age of presentation in our study was 36 years, it must be noted 4 out of 5 subjects presented in forth to fifth decade of life. Interestingly, all the four subjects had ACC. The remaining one subject had CCC and presented in the second decade of life [Table 4]. This difference in the mean age of presentation could be because of the lesser number of subjects available for the study and hence needs future studies on the subject.

It must also be noted that majority of these patients present in advanced stage. This could be because of its slow growing pattern with submucosal spread.[2],[5],[6],[11] Advanced stage of presentation include stage III or IV.[4],[10] Similar result was noted in our study also with 2 subjects each at Stage III and IVa [Table 1] and [Table 3]. In addition, we noted an earlier presentation for glottis CCC, which was similar to study in 2011 by Stephen R Hoff.[12] This could be because of earlier detected hoarseness of voice if the primary lesion is at the glottis which is not true with lesion at subglottis or supraglottis, where it presents with noisy breathing or muffled voice.[6] However, it can have other symptoms like hoarseness of voice and dyspnea depending on the extent of lesion to glottis or trachea respectively.[6],[8] Pain and laryngeal tenderness suggest perineural invasion.[10] Hemoptysis is an uncommon presentation as the lesion has submucosal spread.[10] Metastasis to cervical lymph nodes is also not a common presentation, corresponding to <15%.[6] Many a times, these symptoms develop at advanced stages only.[2],[5] The extent of the lesion, hence, is of significant importance in minor salivary gland neoplasms of larynx. This is best assessed by the contrast-enhanced computerized tomography of the neck, extending from the dura to the pleura.[3],[4],[9] This was agreed up on in our study as well. Contrast-enhanced computed tomography study helps delineating the lesion from the surrounding tissue, better due to its contrast uptake [Figure 1]. Imaging of the thorax is also performed due to the high chance of lung metastasis. Fiberoptic laryngoscopy can also be done for diagnosis and biopsy.[5] In difficult cases, multiple biopsies may be needed for diagnosis.[6] Magnetic resonance imaging of the neck is generally not preferred due to the presence of bony landmarks around and possibility of motion artifacts due to the swallowing and breathing.[12]

James Cohen, in 1985, concluded that surgery was the primary treatment modality used in most subjects.[2] Surgery performed for these lesions usually depends on the site and extent of the lesion. TL is the most common surgery performed with adjuvant therapy.[3],[4],[5],[8],[9],[10] However, supraglottic laryngectomy is also performed for lesion restricted to single site of supraglottis[3] and neck dissection is performed for clinically apparent lymphadenopathy.[10] Study in 2006 by Katherine T et al. on 22 subjects with ACC concluded surgical resection followed by radiotherapy as the best treatment for ACC, irrespective of its location (10).[10] Similar results were observed in our study as well with 4 out of 5 subjects (80%) receiving surgical management, among which 3 had ACC and 1 had CCC. Subjects with ACC underwent TL with neck dissection and subject with CCC underwent vertical hemilaryngectomy with laryngoplasty by stylohyoid repositioning[11] [Table 2]. All the resected specimen had perineural invasion as well and hence TL is recommended[3],[6],[10] [Table 3]. However, 1 subject with ACC underwent primary chemoradiation. All subjects were followed up regularly, with the latest follow up of 15 years for one of them. Subjects were assessed to look for recurrences and residual lesions at the follow up. Interestingly, only the subject who underwent primary chemoradiation was noted to have a residual lesion in the trachea at 9 months follow up [Table 2]. This was comparable with other studies as well wherein recurrences were noted high following primary radiation or primary chemoradiation[3] [Table 4]. Thus, it can be noted that early-stage salivary gland tumors of larynx can be treated successfully with trans-oral laser microsurgery.

Distant metastasis is also not uncommon in minor salivary gland neoplasms of larynx, more common in ACC. The most common site being lung, it remains the principal cause of treatment failure in these subjects[2],[3],[5],[10] [Table 4]. Distant metastasis can develop as late as 10 years and hence requires long term follow up for all these subjects.[3],[6],[10] Up to 60% of minor salivary gland neoplasm is expected to cause locoregional or distant metastasis.[3],[5],[10],[12]


  Conclusion Top


Minor salivary gland neoplasms are extremely rare, accounting to <1% of laryngeal malignancies. ACC is the most common salivary gland neoplasms of larynx, predominantly seen in subglottis than other subsites of larynx. In general, these are very slow growing lesions and are diagnosed in advanced stage due to late clinical presentation. Surgical resection with adequate margins, hence TL, with adjuvant chemoradiation seems to provide better outcome than chemoradiation alone. However, depending on the stage and number of subsite, supraglottic laryngectomy can also be performed and neck dissection is advised in the presence of cervical lymphadenopathy. Long term follow-up is suggested due to chances of recurrences or distant metastasis as late as 10 years, especially in case of ACC.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Disclosure

This material has never been published and is not currently under evaluation in any other peer reviewed publication.

Ethical approval

The permission was taken from Institutional Ethics Committee prior to starting the project. All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Informed consent

Informed consent was obtained from all individual participants included in the study.



 
  References Top

1.
Bobdey S, Jain A, Balasubramanium G. Epidemiological review of laryngeal cancer: An Indian perspective. Indian J Med Paediatr Oncol 2015;36:154-60.  Back to cited text no. 1
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2.
Cohen J, Guillamondegui OM, Batsakis JG, Medina JE. Cancer of the minor salivary glands of the larynx. Am J Surg 1985;150:513-8.  Back to cited text no. 2
    
3.
Ganly I, Patel SG, Coleman M, Ghossein R, Carlson D, Shah JP. Malignant minor salivary gland tumors of the larynx. Arch Otolaryngol Head Neck Surg 2006;132:767-70.  Back to cited text no. 3
    
4.
Karatayli-Ozgursoy S, Bishop JA, Hillel AT, Akst LM, Best SR. Malignant salivary gland tumours of the larynx: A single institution review. Acta Otorhinolaryngol Ital 2016;36:289-94.  Back to cited text no. 4
    
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Ricciardiello F, Addeo R, Di Lullo AM, Abate T, Mazzone S, Oliva F, et al. Adenoid cystic carcinoma of the larynx in a 70-year-old patient: A case report. Oncol Lett 2018;16:2783-8.  Back to cited text no. 5
    
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Wang Q, Yu XS, Liu Y, Chen H. Adenoid cystic carcinoma in ventricle of larynx: An interesting case. Medicine (Baltimore) 2018;97:e13622.  Back to cited text no. 6
    
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Kolahdouzan M, Abtahi SH, Abedini L, Jafari HR, Mostafapour E. The case report of laryngeal adenoid cystic carcinoma: A rare entity. International Journal of Cancer Management. 2017;10.  Back to cited text no. 7
    
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Raman S, Rajan R, Rajan R. Adenoid cystic carcinoma of the larynx: an unique presentation. Journal of Evolution of Medical and Dental Sciences. 2014;3:5950-4.  Back to cited text no. 8
    
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Zvrko E, Golubović M. Laryngeal adenoid cystic carcinoma. ACTA otorhinolaryngologica italica. 2009;29:279.  Back to cited text no. 9
    
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Triantafillidou K, Dimitrakopoulos J, Iordanidis F, Koufogiannis D. Management of adenoid cystic carcinoma of minor salivary glands. J Oral Maxillofac Surg 2006;64:1114-20.  Back to cited text no. 10
    
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Nayak DR, Balakrishnan R, Rao RV, Hazarika P. Clear cell carcinoma of the larynx - A case report. Int J Pediatr Otorhinolaryngol 2001;57:149-53.  Back to cited text no. 11
    
12.
Hoff SR, Apushkin M, Pytynia KB. Clear cell carcinoma of the larynx: A report of a rare case and review of the literature. Laryngoscope. 2011;121:S284.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4]



 

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