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��ࡱ�>��	WY����V��������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������������	��^9bjbj����	.V�����/3�������66yyyyy�������8�D	4�V)t==SSS...�(�(�(�(�(�(�(�.�l1�(-y.....�(yySS�)�&�&�&.jySyS�(�&.�(�&�&u(�(S������Z����!�(�(&)0V)�(�2�#��2�(�2y�(0..�&.....�(�(�&...V)....���������������������������������������������������������������������2.........6	?:	Radiotherapy is Not Exclusively Indicated  in Acinic cell carcinoma of the parotid gland, a rare neoplasm. 
Tan SN1, Primuharsa Putra SHA2, Muhaizan WM3, MS Kenali4

1Masters  of Otorhinolarygology-Head & Neck Surgery, School of Medicine, KPJ Healthcare University College, Malaysia.
2Ear, Nose and Throat-Head and Neck Consultant Clinic,KPJ Seremban Specialist Hospital, Malaysia/ KPJ Healthcare University College, Malaysia
3Consultant Pathologist, KPJ Lablink, Malaysia
4Ear, Nose and Throat-Head and Neck Consultant Clinic, KPJ Tawakkal Specialist Hospital, Malaysia/ KPJ Healthcare University College, Malaysia

Abstract
This case report highlights a rare parotid gland tumour, presenting the treatment options for this entity. It also emphasized the necessity of treatment that depends on case basis.  Acinic cell carcinoma (ACC) is a rarest form of low-grade malignant salivary neoplasm in which the predominant site is the parotid gland. The main treatment of ACC is surgical excision. Radiotherapy may be indicated in some cases. However, it has not been proven to have an impact on the survival rate.  We present a case of 46 years lady who presents with slowly enlarging right parotid mass which was surgically removed. The mass was confirmed to be acinic cell carcinoma by pathology. No adjuvant radiotherapy was given. No evidence recurrence after 2 years of post operative follow up.

Keywords : radiotherapy, acinic cell carcinoma, rare malignat parotid neoplasm




Introduction
Salivary gland carcinoma is not common, make up to less than 1 percent of the cancers in the United States[1]. In parotid glands, 20-25% of the tumors are malignant compared to another salivary gland cancer[2]. Acinic cell carcinoma being fourth most common malignant parotid gland tumor after adenoid cystic carcinoma (AdCC)  mucoepidermoid carcinoma(MEC), and carcinoma ex pleomorphic adenoma[3]. Approximately 10% of salivary gland malignant tumors are acinic(or Acinar) cell carcinoma and most commonly occurs in the parotid gland[4]. 

Besides parotid gland, this tumor can be seen in the lacrimal glands, minor salivary glands of the oral cavity, throat, submandibular gland,   and larynx. In comparison to other salivary gland malignancies, ACC malignancy is quite low[5,6].
ACC happens at an earlier age than other salivary gland tumors.  ACC tumor is seen in person between 5th and 6th decade of life[7]. Most studies have noticed that women are affected more frequently than men[ 7] with female to male ratio of 1.5:1. 
Salivary gland cancers etiological agents still remain unclear. Although most head and neck carcinoma are strongly associated with smoking and alcohol drinking, however, this is not so for salivary gland [8,9].

The cause of acinic cell carcinoma is unknown as little or no research has been done in this area10. It has to do with multiple factors, probably the environmental or genetic. A familial predisposition in parotid cancer, development of minor salivary gland adenocarcinomas of the nasal and paranasal sinuses due to chronic wood dust inhalation in certain individuals are other causes that have been cited in the literature. Second-hand smoke is another possible carcinogen related cause. The potential causes listed are theoretical and anecdotal only. Theses causes have been documented in other cancers, but not ACC specifically[10].
ACC typically appears as a solitary, painless encapsulated,soft and slow-growing, mass that gradually enlarges usually over a period of several years. This might result in facial asymmetry although not all the cases present so. There are approximately 40 years of ACC progression leading to a huge tumour documented in a few literature[3]. Cushing syndrome sometimes may be associated with ACC coexists with pituitary adenoma or releases ACTH[11]. 


















Case report
My patient, 46 years, Malay lady with no known medical illness presented to head and neck surgery clinic with right parotid swelling of a year duration, insidious onset and progressive in nature. 
    The patient had a firm to hard multilobulated swelling over right parotid gland which was tender measuring 3cm x 3cm. Facial nerve function was intact. Other head and neck examination was normal. A pre-operative CT scan of right parotid mass was suggested right intra parotid mass involving the superficial lobe measured 5cm x 4cm x 1.5cm. She then underwent superficial parotidectomy with preservation of the facial nerve. The multilobulated mass measures 4cm x 3cm x 2cm and was well encapsulated at the superficial lobe of the right parotid gland. Deep lobe appeared normal. 
    The postoperative histopathological report revealed Acinic Cell Carcinoma with margin of the cells clear. 
    She is then follow-up by the oncology specialist and no radiotherapy was done in view of complete surgical excision. No recurrence on follow-up of three years. 








               






Discussion
We present a case report where incidental result detected by histopathology report with diagnosis of acinic cell carcinoma of the parotid gland. Initially, was thought to be benign disease entity. Foote and Frazell [3,11] first coined it as a carcinoma in the early 1950s [3,7,11].
The presented case of such a parotid gland with histology of acinic cell carcinoma is unusual in the literature[3,4].

The possible differential diagnosis can be divided into nonneoplastic and neoplastic conditions which include neoplastic lesions, congenital, vascular malformations, acute and chronic cervical lymphadenopathies and cystic lesions[12,13].

Radiological imaging such as ultrasonography, CT, and MRI are the ultimate requirement for evaluation of tumour size and adjacent anatomical structures involvement. These are helpful in the preoperative planning of the surgical procedure [3,14].
The basis of tumour biopsy determined the diagnosis[3].
Tumour diameter greater than 3cm with presence of invasion of tissue are said to be other unfavourable prognostic factors[5].
Acinic cell carcinomas are low-grade and have good survival rates (about 70% at 10 years)[1,4].

Surgery is often the primary treatment which is the preferred method of treatment for ACC of the parotid gland. However, it has not been established if removal of the entire gland is preferred or if it is better to attempt to remove only the cancerous tissue. 

Removal of tumour within the limits of unaffected tissues was thought to be the most effective way of treatment according to the literature of Goodwin et al.  A similar opinion  by Kim and Mathog, where they suggest a superficial parotidectomy for tumors in the superficial lobe of the parotid gland. They also suggest  total parotidectomy and facial nerve sparing for neoplasms in the deep lobe of the parotid gland[15].

The role of radiotherapy in acinic cell salivary tumours, however, remains controversial. It is said that adjuvant radiotherapy does not have an effect in a significant survival advantage for early stage or lower grade parotid ACC[16].  Primary radiotherapy is limited to patients not fit for surgery or refusing surgery. However, it is not favoured as a primary mode of treatment, as ACC has regarded as being not radiosensitive. Nevertheless, there still appeared to be a part for adjuvant postoperative radiotherapy although this is ill-defined and controversial. 
Post-operative radiation therapy is usually recommended as adjuvant treatment, usually in cases of positive surgical margins or tumour recurrence. Tumour of the deep lobe tumour in the facial nerve, diameter of the tumour greater than 4cm, extra glandular tissue spread, and regional lymph node metastases are all other possible indications of radiotherapy[17].

Debate is still ongoing regarding the role for neck dissection, adjuvant radiotherapy  and extent of resection needed as there is no impact on survival rate[10].
In this case, a superficial parotidectomy was done. Due to no lymph node metastases in the neck, clear surgical margins, and only involvement of the superficial lobe of the parotid gland � the patient was not subjected for postoperative radiotherapy even though recommended by some authors in case of this tumour[13,18]. 
Nevertheless, follow up of the patient did not show any mass, recurrence or metastasis.
A further case studies and long term follow up are indicated for further documentation.








Conclusion
��Acinic cell carcinoma is a low-grade malignant salivary neoplasm rarely diagnosed in salivary gland that may be overlooked. The overall prognosis after the surgical resection depends on the extent of lesion and adequacy of initial resection. Careful long term follow up protocol is advised in view of its malignant potential. One should have a high index of clinical suspicion, prompt diagnosis and definitive surgical treatment. If the condition is dealt appropriately from the beginning, the morbidity is extremely low and permanent cure is highly probable. The proper identification of this ACC is of paramount importance for decision of radiotherapy while also allowing one to avoid the irradiation adverse effects in patients who do not require such treatment.


References
Anthony C. Nichols, Michelle Chan-Seng-Yue, John Yoo, et al. (2013). A Case Report and Genetic Characterization of a Massive Acinic Cell Carcinoma of the Parotid with Delayed Distant Metastases. Oncological Medicine, Article ID 270362, 7 pages
Victor Shing Howe To, Jimmy Yu Wai Chan, Raymond K.Y. Tsang et al. (2012). Review of Salivary Gland Neoplasm. International Scholarly Research Network ISRN Otolaryngology Article ID 872982 6 pages
JarosBaw Dbrowski, Tomasz Pitka, Jan Przybysz et al. (2011). Acinic cell carcinoma of parotid gland. A case report.  Contemporary Oncology vol. 15; 1 (47 50)
Lucinei Roberto Oliveira; Danilo Figueiredo Soave; Jo�o Paulo Oliveira da Costa et al. (2010). Acinic Cell Carcinoma of Parotid Gland:Report of Three Cases and Literature Review. Revista Portuguesa de Estomatologia, Medicina Dent�ria e Cirurgia Maxilofacial Volume 51.
Gnepp DR, Henley JD, Simpson R, Eveson J. Salivary and Lacrimal Glands. (2009). Diagnostic surgical pathology of the head and neck Gnepp DR. Saunders 2nd edition; 476-81.
Goldman NC, Lee J, Tolley B. (2007). Acinic cell carcinoma of the parotid gland. Otolaryngol Head Neck Surg ; 137: 828-9

Wonjae Cha, MD�Min-Su Kim, MD�Jae-Chul Ahn et al. (2011). Clinical Analysis of Acinic Cell Carcinoma in Parotid Gland. Clinical and Experimental Otorhinolaryngology Vol. 4, No. 4: 188-192, Korea.
American Joint Committee of Cancer (2010). Major Salivary Gland. AJCC Cancer Staging Manual. 7th ed. New York, Springer; 2010: 79-82.
John Han-Chih Chang, Kenneth Blank(2001). Acinic Cell Carcinoma of the Salivary Gland. Abramson Cancer Center of University of Pennsylvania November 1, 2001
Alexander Delides, George Velegrakis, George Kontogeorgos, et al. (2005). Familial bilateral acinic cell carcinoma of the parotid synchronous with pituitary adenoma : case report. Wiley InterScience (www.interscience.wiley.com). DOI: 10.1002/hed.20219
Foote FW Jr, Frazell EL (1953).Tumors of the major salivary glands. Cancer. 6(6):1065-133.
Chiara Bianchini, Andrea Ciorba, Francesco Stomeo et al.(2011). A case of parotid acinic cell carcinoma in a young boy. Braz J Otorhinolaryngol. 77(3):402.
Harrison W. Lin, MD, Neil Bhattacharyya(2010). Parotid mass. ACS Surgery Principle and Practice Section 2 Head and Neck.
Kim SA, Mathog RH(2005). Acinic cell carcinoma of the parotid gland: a 15year review limited to a single surgeon at a single institution. Ear Nose Throat J 84: 597-602
Michael T. Andreoli, Steven M. Andreoli, Mark G. Shrime et al.(2012) Radiotherapy in Parotid Acinic Cell CarcinomaDoes It Have an Impact on Survival? JAMA otolaryngology Arch Otolaryngol Head Neck Surg.;138(5):463-466. doi:10.1001/archoto.2012.226. )Cited in PubMed
Sang-il S, Hae Young S, Tai-kun K, et al.(2005) Acinic cell carcinoma of the head and neck : radiologic � pathologic correlations. J Comput Assist Tomogr 29: 121-6
Samuel R. Greig, John M. Chaplin, Nicholas P. Mcivor, et al.(2008) Acinic cell carcinoma of the parotid gland: Auckland Experience and Literature review� ANZ J. Surg. 78: 754�758










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