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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 31  |  Issue : 1  |  Page : 79-83

Diagnosis of bilateral parotid enlargement (Sialosis) by sonography: A case report and literature review


1 Department of Oral Medicine and Radiology, Vyas Dental College and Hospital, Jodhpur, Rajasthan, India
2 Department of Oral and Maxillofacial Surgeon, Sardana Hospital and Trauma Centre, Panipat, India
3 Department of Oral and Maxillofacial Surgery, SGT University, Gurugram, India
4 Department of Oral and Maxillofacial Surgeon, Ambala, Haryana, India

Date of Submission01-Oct-2018
Date of Acceptance11-Feb-2019
Date of Web Publication23-Apr-2019

Correspondence Address:
Dr. Sugandha Arya
Department of Oral Medicine and Radiology, Vyas Dental College and Hospital, Jodhpur - 342 001, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_168_18

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   Abstract 


Alcoholism is a common cause of sialosis. We describe a case of sialosiswith diffuse painless bilateral parotid gland enlargement associated with alcoholism. The diagnosis was based on clinical appearance, biochemical investigations and ultrasonography. The parotid swelling resulting from sialadenosisis asymptomatic, which concerns the patient because of cosmetic reasons. The dental practitioner needs to be able to differentiate sialosisfrom other pathological states that demonstrate bilateral parotid swelling to avoid unnecessary treatment.

Keywords: Alcoholic sialosis, sialoadenosis, sialosis


How to cite this article:
Arya S, Pilania A, Kumar J, Talnia S. Diagnosis of bilateral parotid enlargement (Sialosis) by sonography: A case report and literature review. J Indian Acad Oral Med Radiol 2019;31:79-83

How to cite this URL:
Arya S, Pilania A, Kumar J, Talnia S. Diagnosis of bilateral parotid enlargement (Sialosis) by sonography: A case report and literature review. J Indian Acad Oral Med Radiol [serial online] 2019 [cited 2019 Dec 14];31:79-83. Available from: http://www.jiaomr.in/text.asp?2019/31/1/79/256890




   Introduction Top


Sialadenosis is a unique form of non-inflammatory, non- neoplastic bilateral salivaryglandd is order characterized by recurrent, perisistent, bilateral parotid enlargement that is asymptomatic and does not affect salivary gland function.[1],[2],[3] For these unique salivary gland swellings, sialosis has been recommended as the correct diagnostic term by the WHO rather than sialoadenosis.[4] This entity has been found to be associated with alcoholism, chronic malnutrition, eating disorders, obesity, endocrinopathies, drugs mainly anti-hypertensives along with many idiopathic causes.[5],[6],[7],[8] Incidence of sialosis has been estimated to be 30–86%in patients with alcoholism and alcoholic cirrhosis.[9]

The disorder is found to be affecting people in the fourth and seventh decades of life without any sex predilection and can persist for more than 20 years.[10],[11],[12] The parotid swellings are soft in tone, noninflammatory, non-neoplastic, and usually symmetrical. They are slow growing and may be associated with pain.[12],[13],[14] These swellings fill the depression in the auricular area between the mandibular ramus and the superior segment of the sternocleidomastoid muscle.[14] This article presents a case of sialosis with diffuse painless bilateral parotid gland enlargement associated with alcoholism.


   Case Report Top


A 35-year-old patient reported to the Department of Oral medicine Radiology in Vyas dental college and Hospital complaining of painless swellings present on both sides of the face belowthe preauricular region from past 1.5 year. The medical history was noncontributory and the patient was not on any medication. The patient reported that food did not cause the swellings to change in size, and he was unaware of any recent increase in their size. During the past 1.5 year, swellings had remained persistent without any pain, fever, purulent discharge or discomfort. The patient did not observe any submandibular salivary gland swelling nor reported any history of any eating disorder or dry mouth. Patient was moderately nourished without any weakness or numbness reported and indicated that his weight was static from many years. Questioning of the patient indicated that for the past 7–8 years he had imbibed 4 ounces of hard liquor each night. Patient was examined after obtaining consent. On examination, patient had diffuse bilateral enlargement of the parotid gland, supero-inferiorlyit was roughly 6 × 3 cm and 5 × 4 cm on both right and left side respectively and antero-posteriorly from the line joining the tragus till the angle of mandible with slight lifting of ear lobes seem bilaterally. No erythema was observed [Figure 1], [Figure 2], [Figure 3]. On palpation of parotid gland, supero-inferior extension of swelling was 5 × 2 cm and 5 × 3cm on both right and left side, respectively. Both the parotid glands were nontender on palpation, normal in tone and soft torubbery inconsistency with no palpable masses or any local rise in temperature. No other salivary glands were swollen, and cervical lymphadenopathy was not evident. Intraoral findings were noncontributory except the patient's mucosa was found to be moist. Both parotid ducts were patent and on milking of parotid gland clear fluid was readily expressed from Stensen's duct. On probing the parotid ducts, we did not encounter any impediments. Tongue blade test for dryness of mouth was negative. Biochemical investigations such as Random blood glucose and Hb were observed in normal range. A liver profile study was requested to ascertain hepatic function. Liver enzymes, bilirubin, protein, and albumin levels were all found to be within normal range. Sonography of the neck region was advised, and it revealed bilateral enlarged parotid glands, echotexture was normal with no masses seen and without evidence of lymphadenopathy. Bilateral submandibular glands were found to be normal [Figure 4]. A diagnosis of alcohol induced sialosis was then made by integrating history, clinical appearance, chair side, and biochemical investigations and ultrasonography. Absolute alcohol abstinence and habit counseling along with dietary supplements was advised and patient was kept under a regular follow up.
Figure 1: Showing bilateral parotid enlargement in a 35 year old male

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Figure 2: Showing right parotid enlargement (6 × 3 cm supero-inferiorly)

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Figure 3: Showing left parotid enlargement (5 × 4 cm supero-inferiorly)

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Figure 4: (a) Showing sonography of neck region. (b) Showing sonography of neck region

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   Discussion Top


Sialosis is a chronic, bilateral, diffuse, noninflammatory, non-neoplastic swelling of the major salivary glands that primarily affects the parotid glands, but occasionally involves the submandibular glands and rarely the minor salivary glands (Scully 2008).[15] Clinically, it presents as soft, often bilateral, usually painless, and recurrent swelling of the parotid gland. Few cases with unilateral Sialoadenosishas been reported as well.[7] The highest rate of incidence has been reported between 30 and 69 years of age[9] which is in accordance in our case where the 35-year-old reported with painless diffuse bilateral parotid enlargement with a positive history of alcoholism which was later observed to be the cause of sialosis in our case. L Mandel et al.[9] reported chronic alcoholismasone of the primary causes of sialosisalong with obesity, malnutrition, endocrinopathy (diabetes, hypothyroidism), medications, heavy metal sandotheridiopathic causes.[16] The pathophysiology of sialosis is obscure and hence, elucidation of the physiological mechanisms of salivary gland secretion is required. The parotid gland has both a parasympathetic and a sympathetic supply. The parasympathetic innervation is related with fluid and electrolyte secretion. The sympathetic supply is concerned with intracellular protein synthesis and protein secretion. With a disturbance in the autonomic sympathetic innervation, dysregulation of protein synthesis and/or its secretion occurs leading to development of cytoplasmic swelling from engorgement by intracytoplasmic zymogen granules hence, resulting in increase in size of the parotid's acini, which normally measure 40 μm in diameter to as much as 100 μm. This enlargement causes the clinically visible glandular hypertrophy.[17] Alcoholism tends to initiate an autonomic neuropathy that manifests itself as a demyelinating polyneuropathy. There by, its chronic consumption has been implicated in the etiology of sialosis.[9],[17] Only 10% of patients with noncirrhotic alcohol damaged livers demonstrate the classic parotid swelling, whereas rest 30–80% of patients represent with cirrhosis.[2],[9],[18],[19] Our case reported was amongst 10% wherein alcoholic sialosis occurred without any evidence of liver disease. A firm distinction has also not always been possible between the effects of alcoholism itself and those of hepatic cirrhosis and it is still debatable.[2] It has been documented that EtOH consumption has been associated with triggering damage to salivary glands, which can cause morphological and functional changes resulting in disturbances in the metabolism and excretion of the salivary glands. The salivary glands, notably parotid glands, may become swollen in long term alcohol drinkers[20] as observed in our case. Different histopathology pictures in alcoholic sialosis have been identified by many authors[21],[22],[23] who reported a reduction in the proportion of fatty tissue of stroma with an enlargement in ductal epithelium felt to contribute to an increased caliber of the striated ducts along with accumulation of secretory granules in the acinar cells' cytoplasm and enlarged excretory ducts. Imaging is rarely performed to diagnose sialosis, and is usually not helpful as the enlarged gland varies in echotexture, attenuation, and signal intensity depending on the dominant histological change. On sialography, the parotid glands appear enlarged and the ducts are usually normal in appearance but splayed by the increased gland volume.[8] Hence, it can play an important role in the diagnosis of sialosis, particularly in the later stages of the disease, when changes are caused by acinar swelling with compression of the proximal ducts resulting in a characteristic 'leaflesstree pattern.'[12] In the early stages of the disease, there may be no sialographic changes, as occurred in our case. Diagnosis of sialosis is facilitated by its clinical manifestations. The bilateral painless parotid swellings have been present for a prolonged time, they do not fluctuate in size in association with meals, and patent parotid ducts with a free and clear salivary flow can be observed. Blood chemistry studies are a significant aid in determining the existence of liver dysfunction, diabetes, or abnormalities associated with malnutrition.[1] In our case, diagnostic criteria for sialosis given by A Gadodia et al.[8] on the basis history and physical findings and chair-side investigations like milking of gland from which clear fluid was obtained along with normal blood chemistry investigations such as random blood glucose, liver function tests helped in establishing the accurate diagnosis. Additional sonography was advised to rule out pseudo lesions as well as other pathologies associated with bilateral parotid enlargement [Figure 4]. Bilateral parotid swellings are not an unusual occurrence. Because these swellings are not localized to 1 gland, it must be assumed that systemic factors are usually in play. A wide variety of conditions may be responsible for enlargement of the parotid salivary gland.[1]

Chen et al.[23] reported sialosisto be the most commonest reason for bilateral involvement in parotid enlargement along with iodine mumps. History and physical exam findings such as disease onset, pain, and nodularity are key elements in the algorithm to quickly narrow the differential diagnosis of bilateral parotid enlargement.[8],[23]

When confronted with a patient who has parotid enlargement, the initial step is to decide whether this represents a discrete tumor mass or a diffuse swelling of the gland. If a discrete tumor mass is present, treatment is relatively straight forward and usually involves surgical excision. When a diffuse or bilateral enlargement exists, there are many diagnostic possibilities. The differential diagnosis of such a disorder is necessary to prevent unnecessary treatment as dentists must be able to differentiate sialosis from other pathological states that demonstrate bilateral parotid swelling.[8],[9],[14],[23],[24],[25] Benign masseteric hypertrophy is the commonly encountered entity which can be mistaken for parotid enlargement and has been considered as the pseudolesion[8] because of the fact that the bulk of the superficial lobe of parotid gland rests over posterior aspect of the masseter muscle. Clinically, the face takes a rectangular configuration. Radiographically there is bony hyperplasia at the site of muscle mandibular insertion with flaring of the mandibular angle. The other pathological states which shows parotid enlargement and could be a possible differential for sialosis include: Diabetic parotid sialosis causing bilateral parotid enlargementit can be differentiated with the evaluation of serum glucose levels which in our case was found to be in normal range. The persistent bilateral parotid swelling seen with bulimia that represents a work hypertrophy which is secondary to the parotid stimulation associated with the chronic emesis; epidemic parotitis, or mumps, which is a communicable viral disease that usually occurs in children as a single episode of painfuland firm parotid swelling. None was contributory in our case.

Recurrent parotid swelling with or without associated pain suggests a chronic inflammatory disease, which can be due to recurrent bacterial infections, granulomatous gland diseases, an autoimmune process, or can be idiopathic. Recurrent parotitis in children that has an unknown etiology and is characterized by intermittent inflammatory episodes ofparotid swelling with remission usually occurring at puberty; Chronic recurrent sialadenitis is clinically characterized by recurrent diffuse painful swelling of the salivary gland accompanied by incomplete ductal obstruction. Patients usually have mildly painful parotid swelling aggravated by eating. Massage of the gland typically yields scant saliva from Stensen's duct. Our patient's swelling was nontender, regardless of eating activity. Furthermore, massage of the gland produced clear, abundant saliva. Sarcoidosis is a granulomatous disease that can involve the salivary gland in 6% of cases. It manifests as bilateral parotid swelling, lung infiltrates and hilar lymphadenopathy that can be detected radiographically. Sjogren's syndrome with bilateral parotid swelling that is identifiable by the pathognomonic hallmarks of xerostomia, xerophthalmia, classic serum antibody findings and an association with a connective-tissue disease[8],[9],[14],[23],[24],[25] Other conditions such as the bilateral parotid swelling seen in HIV that usually reflects the presence of lymphoepithelial cysts that can be imaged by a CT, scan and HIV that can be substantiated by blood studies; the bilateral parotid swellings caused by a non-Hodgkin's lymphoma that can be diagnosed by the presence of widespread organ involvement, lymphadenopathies and a histologic examination. Radiation sialadenitis which occurs following the radioactive iodine (I131) treatment of thyroid carcinoma causing transient bilateral parotid swelling occurring usually within 24 h of iodine ingestion, which resolves spontaneously within a week[8],[9],[14],[23],[24],[25] which in our case were lacking.

Thereby, integrating the findings on the basis of history and physical finding diagnostic criteria[8] and algorithm approaches[23] in obtaining the diagnosis of bilateral parotid enlargement along with biochemical investigations and ultrasonography, the diagnosis of alcoholism induced sialosis was confirmed in our case.

The importance of diagnosing sialosis rests in the fact that it signifies a hidden underlying systemic disease. Thereby, recognizing mandates a medical referral and the initiation of the requisite care.[1] Since, the parotid swellings associated with alcoholic sialosis cause no patient discomfort but cosmetic concerns. Since, no treatment can be offered other than surgical reduction, andsuch a procedure is rarely warranted. Furthermore, Alcoholic sialosis needs to be directed at the more serious aspect as it commonly indicates liver damage. Medical therapy results in some decrease of liver disease and parotid gland size, but the long-term prognosis varies.[9] Our case was alcoholic sialosis without any liver disease thereby, in our case absolute alcohol abstinence and psychological counseling along with dietary supplements were given and patient was kept under regular follow up.


   Conclusion Top


Various pathologies that affect the parotid gland mostly result in enlargement and xerostomia. Such limited response of the salivary glands to a multitude of insults makes diagnosis difficult. Thereby, differentiating sialosis from other pathologic states that cause bilateral parotid swellings is important to avoid unnecessary treatment. Furthermore, such diagnosis can indicate the existence of occult hepatic disease, which demands early intervention so that progression to more serious liver pathology can be avoided.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

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Mandel L, Surattanont F. Bilateral parotid swelling: A review. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;93:221-37.  Back to cited text no. 1
    
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Guggenheimer J, Close JM, Eghtesad B. Sialadenosis in patients with advanced liver Disease. Head and Neck Pathol 2009;3:100-5.  Back to cited text no. 2
    
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Yu YH, Park YS, Kim SH, Son BK, Jun DW, Jo YJ, et al. Sialadenosis in a patient with alcoholic fatty liver developing after heavy alcohol drinking. Korean J Gastroenterol 2009;54:50-4.  Back to cited text no. 3
    
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Jagtap SV, Aramani SS, Mane A, Bonde V. Sialosis: Cytomorphological significance in the diagnosis of an uncommon entity. J Cytol 2017;34:51-2.  Back to cited text no. 7
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Werning JT. Infectious and systemic diseases. In: Ellis GL, Auclair PL, Gnepp DR, editors. Surgical Pathology of the Salivary Glands. Philadelphia: W.B. Saunders Company; 1991. p. 39-59.  Back to cited text no. 10
    
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Scully C, Bagán JV, Eveson JW, Barnard N, Turner FM. Sialosis: 35 cases of persistent parotid swelling from two countries. Br J Oral and Maxillofac Surg 2008;46:468-72.  Back to cited text no. 15
    
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Riad MA, Salma HT, Sabra RMI, Hamdy TA. Parotid sialometry: A standard technique in the diagnosis of parotid sialadenosis. Egypt J Otolaryngol 2014;30:138-41.  Back to cited text no. 19
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    Figures

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



 

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