BILATERAL MULTIPLE PAROTID CALCULI.
Bilateral multiple parotid calculi are rare entity. Calculi are found in normal individuals as well as in association with some local and systemic diseases. A case of bilateral multiple intra-parenchymal parotid calculi in and otherwise healthy patient who developed recurrent parotid swelling and had unusual cutaneous exfoliation of parotid calculi from the parotid region, is reported.
Human research ethics committee of Directorate Royal Medical Service agreed to prepare this case report, and the consent of the patient was obtained.
Key words: Sialolithiasis, parotid, bilateral, intraparenchymal, calculi
Sialolithiasis is a common disease. Its incidence is higher than it is generally realized. Postmortem studies indicate that the incidence of sialolithiasis among general population is 1.2%.1 Sialolithiasis occurs mainly in the submandibular gland (80-90%), and to a lesser degree in the parotid gland (5-20%). The sublingual and minor salivary glands are rarely affected.2
Parotid sialolithiasis usually involves one gland at a time, and the stones are usually solitary and often involves the ductal system of the gland. Simultaneous sialolithiasis of both parotid glands has a low incidence,2 and the presence of multiple stones in the parenchyma of the gland is a rare finding.3
An unusual case of bilateral multiple intra-parenchymal parotid calculi occurring in the absence of any systemic or local disease and demonstrating unusual cutaneous exfoliation of calculi in the parotid region is described in this report.
A 32-year-old woman presented to oral and maxillofacial surgery department of this hospital in 2009 with a 4-year history of recurrent pain and swelling in the right parotid gland with fluctuation in size during meals and episodes of pus discharge from right Stenson's duct. Previous investigations included ultrasonography (US) of both parotids, and fine needle aspiration cytology (FANC) and sialogram of right parotid gland, and a diagnosis of obstructive sialadinitis with sialolithiasis was made.
When the patient reported her chief complaint was the presence of a subcutaneous nodule in the right parotid region. The patient's medical history was uneventful. Physical examination of the patient revealed an infra-auricular, hard and tethered subcutaneous nodule, and the skin overlying the nodule was mildly darkened. There was absence of swellings in both parotid regions. Intra-oral examination revealed patency of orifices of Stenson's ducts with clear salivary flow on gentle manipulation of glands. A computerized tomographic (CT) scan without contrast was taken. It revealed the presence of bilateral multiple intraparenchymal parotid calculi of varying sizes and the presence of two calculi, one on each side, situated superficially in the subcutaneous tissues. The right and nearly fistulated calculus was the reason for the patient's chief complaint.
The patient underwent an extensive investigation to exclude any systemic or local diseases or the coexistence of lithiasis in other body organs. The patient's uric acid, calcium and phosphorus levels were all within normal ranges. There was no subjective sensation of dry mouth or dry eyes. Unstimulated and stimulated whole saliva collection performed at 10:00 AM when the patient was fasting and hadn't brushed her teeth before the procedure. Stimulation was induced by asking the patient to chew on a paraffin block. The saliva was then collected by asking the patient to spit out actively into a calibrated container. The values for the whole saliva collection were 2.3 ml per 15 minutes for the unstimulated and 3.2 ml per 5 minutes for the stimulated. Labial minor salivary glands biopsies revealed absence of chronic inflammatory infiltrates.
Autoimmune serology showed negative results for anti-Ro/SS-A, anti-La/SS-B and RF. ANA was 1/80. FNAC of both parotids was consistent with chronic sialadinitis. Kidney CT and gallbladder US revealed no stones.
In the face of long-standing and recurrent complaint, the decision was made to do right superficial parotidectomy. However, the patient didn't accept the possible complications and refused the surgery. The right subcutaneous stone was extirpated and the patient kept under follow-up examination. On the last recall in April 2011, both parotids were asymptomatic but the patient recalled cutaneous exfoliation of seven stones from both parotids.
Sialolithiasis may occur in normal people. Its exact pathogenesis remains unknown, and various hypotheses have been proposed.4 Lustmann et al2 in a survey on 245 patients with sialolithiasis found no correlation between sialolithiasis and systemic diseases and only 6 of the 56 patients who were available for follow-up examination were reported to suffer from nephrolithiasis. The mineral composition of salivary calculi is different from that of biliary and urinary stones,5 and contrary to nephrocalcinosis and uroliths formation, the effect of hypercalcemia on the major salivary glands seems quite weak.3 Furthermore, Sherman et al5 found no correlation between water hardness and salivary calculi in England and concluded that local factors as yet unknown are likely to be important in salivary calculogenesis.
Parotid calculi are usually single, unilateral and located within the ductal system of the gland. Simultaneous bilateral multiple intra-parenchymal calculi like the present case are rarely encountered and few case reports or case series are found in the medical literature describing such finding. Lindamn and Woolley6 described one case in a child with absence of any local or systemic disease. Wickramasinghe et al7 reported a case with MALT lymphoma of the salivary glands who also had multiple calculi in the parotid sialolithiasis in a patient who had acquired immunodeficiency syndrome and was affected by multiple myeloma. Shimizu et al8 reported three cases of Sjogren's syndrome in whom multiple sialolithiasis were observed in the parenchyma of the parotid glands. Konstantinidis et al9 reported a case of bilateral multiple calculi of the parotids in a patient with primary Sjogren's syndrome.
However, as the incidence of bilateral multiple parotid calculi, is very low their occurrence in those more common systemic diseases may be an incidental finding. Furthermore, this entity may occur in the absence of any systemic or local disease as is shown in the present case and in other case reports.6
The present case had undergone an unusual course of the disease. The cutaneous exfoliation or erosion of several calculi at different occasions without fistula formation may be attributed to the long course of the disease and the elevated intra-glandular pressure at times of recurrent obstruction. The secretory function of the parotid glands haven't suffered considerably as clear saliva was expressed from the glands, however, the salivary flow rate hadn't been measured individually for the parotid glands. Although it is commonly believed that a gland with sialolithiasis is no longer functional, Marchal et al10 in their study on submandibular glands removed because of sialolithiasis demonstrated that there was no correlation episodes and despite appropriate indication for submandibular gland removal, close to 50% of the removed glands were histopathologically normal or close to normal.
Sonography is the first-line imaging for the evaluation of many diseases affecting the salivary glands including sialolithiasis.11,12,13 Its accuracy in assessment of sialolithiasis is high.13,14 However, US is less accurate than CT in distinguishing multiple clusters of stones from single large stones.14 Due to its anatomic position, a little portion of the parotid gland may be hidden by the acoustic shadow of the mandible, and therefore not visualized in US.13 Furthermore, the deep part of the gland is sometimes difficult to visualize.14 In the present case, US did reveal the presence of a number of stones, however, there was underestimation of the true multiplicity and location of the stones, and non-enhanced CT was invaluable in detecting the true extent of the disease.
An important differential diagnosis of the present case would be phlebolithiasis. Phleboliths are calcified thrombi found within vascular channels. Although phleboliths can occur in the absence of vascular lesion, they are often associated vascular anomalies,15 and in particular anomalies of low-flow such as venous malformations and cavernous hemangiomas. In fact, the presence of phleboliths is considered by some authros to be suggestive of cavernous hemangioma.16 Vascular lesions of the adult parotid gland is considered to be rare17,18 and the cases reported in literature have been described as being vascular malformations or hemangiomas, predominantly cavernous.18 Phleboliths of head and neck vascular lesions are usually multiple, varied in size and randomly distributed,19 and appear as dense, rounded opacities in contrast to the fainty opaque, scattered shadows of salivary stones.18 However, parotid stones occurring in the parenchyma of the gland may assume similar characteristics of a phlebolith.
Nonetheless, in the present case a vascular lesion was excluded by the cumulative analysis of patient's history, physical examination and investigations.
Multiple intra-parenchymal parotid stones are considered not straightforward cases. The management of such cases is not clear-cut. In the past, such cases were treated by superficial parotidectomy. Minimally invasive techniques, especially endoscopy of the salivary glands, had reduced the number of cases in which superficial parotidectomy is indicated.20 Complicated cases have been treated by many surgeons by a combination of techniques. Nahlieli et al21 in their prospective study assessed a combined external lithotripsysialoendoscopic method for advance salivary gland sialolithiasis and the success rate for complete removal of the stones after lithotripsy sessions was easier and less complicated. In another article, Nahlieli et al20 discussed a new approach to impacted parotid stone included endoscopy and US technique to perform precise removal. They achieved complete removal of stones in 75% of cases, and multiple sialoliths were detected and removed from 4 glands.
Kulkens et al22 demonstrated a connection between the location of the parotid calculi and the success of extracorporeal shock wave lithotripsy (ESWL), in their study 10 patients had intra-parenchymal calculi, and after ESWL treatment 5 of them were free of calculi.
In the present case, the decision was to perform superficial parotidectomy for the effected gland due to the unavailability of the minimally invasive technique. The patient refused such radical treatment and so was kept under follow-up examination. As the patient remained symptom-free during the 2-year follow-up, no intervention was indicated. If symptoms supervene in the future, hopefully ESWL would be an available option as a last resort before superficial parotidectomy.
A bilateral intra-parenchymal parotid calculus is a rare entity. Its association with any systemic or local disease is uncertain. CT is invaluable in detecting the disease. Such cases may be mistaken with other local diseases that show calcifications, such as phlebolithiasis. The management of such cases is not straight-forward, and superficial parotidectomy may be inevitable.
1 Rauch S, Gorlin RJ. Diseases of the salivary glands. In: Gorlin RJ, Goldman HM, eds. Thoma's oral pathology. 6th edn., St. Louis: Mosby, 1970:997-1003
2 Lustmann J, Revag E, Melamed Y. Sialolithiasis: A survey on 245 patients and a review of the literature. Int J Oral Maxillofac Surg. 1990 Jun;19(3):135-38
3 Ottaviani F, Galli A, Lucia MB, Venture G. Bilateral parotid sialolithiasis in a patient with acquired immunodeficiency syndrome and immunoglobulin G multiple myeloma. Oral Surg Oral Med Oral Oathol Oral Radiol Endod. 1997 May;83(5):552-54
4 Marchal F, Dulguerov P. Sialolithiasis management: the state of the art. Arch Otolaryngol Head Neck Surg. 2003 Sep; 129(9):951-56
5 Sherman J.A McGurk M. Lack of association between water hardness and salivary calculi in England. Br J Oral Maxillofac Surg. 2000 Feb;38(1):50-53
6 Lindman JP, Woolley AL. Multiple inter-parenchymal parotid calculi: a case report and review of the literature. Ear Nose Throat J. 2003 Aug;82(8):615-17
7 Wickramasinghe A, Howarth A, Drage NA. Multiple bilateral parotid sialoliths in a patient with mucosa-associated lymphoid tissue lymphoma (MALT lymphoma) of the salivary glands. Oral Surg Oral Med Oral Pahol Oral Radio Endod. 2005 Apr; 99(4):496-98
8 Shimizu M, Yoshiura K, Nakayama E, Kanda S et al. Multiple sialolithiasis in the parotid gland with Sjogren's syndrome and its sonographic findings-report of 3 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2005 Jan;99(1): 85-92
9 Konstantinidis l, Paschaloudi S, Triaridis S, Fyrmpas G et al. Bilateral multiple sialolithiasis of the parotid gland in a patient with Sjogren's syndrome. Acta Othorhinolaryngol ltal. 2007 Feb;27(1):41-44
10 Marchal F, Kurt AM, Dulguero P, Becker M et al. Histopathology of submandibular glands removed for sialolithiasis. Ann Otol Rhinol Laryngol. 2001 May;110(5Pt1): 464-69
11 Howlett DC. High resolution ultrasound assessment of the parotid gland. BR J Radiol. 2004 April;76(904):271-77
12 Ottaviani F, Capaccio P, Rivolta R, Cosmacini P et al. Salivary gland stones: US evaluation in shock wave lithotripsy. Radiology 1997 Aug;204(2):437-41
13 Gritzman N, Rettenbacher T, Hollerwegar A, Macheiner P et al. Sonography of the salivary glands. Eur Radiol. 2003;13(5) 964-75
14 Yosem DM, Kraut MA, Chalian AA. Major salivary gland imaging. Radiology 2000 Jul;216(1):19-29
15 Manddel L, Surattanont F. Clinical and imaging diagnosis of intramuscular himangiomas: the wattle sign and case reports. J Oral Maxillofac Surg. 2004 Jun;62(6):754-58
16 Zachariades N, Rallis G, Papademetriou J, Konsolki E et al. phleboliths. A report of three unusual cases. Br J Oral Maxillofac Surg. 1991 Apr;29(2):117-19
17 Wong KT, Ahuja AT, King AD, Yuen EH et al. Vascular lesions of tparotid gland in adult patients: diagnosis with high-resolution ultrasound and MRI. Br J Radiol. 2004; 77:600-606
18 Saeed WR, Kolhe PS, Smith FW, Murray GL. The turkey wattle' sign revisited: diagnosing parotid vascular malformations in the adult. Br J Plast Surg. 1997;50(1):43-46
19 Mandel L, Perrino MA. Phleboliths and the vascular lesion. J Oral Maxillofac Surg. 2010 Aug;68(8):1973-76
20 Nahlieli O, London D, Zagury A, Eliav E. Combined approach to impacted parotid stones. J Oral Maxillofac Surg. 2002 Dec;60(12):1418-23
21 Nahlieli O, Shacham R, Zagui A. Combined external lithotripsy and endoscopic technique for advanced sialolithiasis cases. J Oral Maxillofac Surg. 2010 Feb;68(2):347-53
22 Kukens C, Quetz JU, Lippert BM, Folz BJ et al. Ultrasound-guided piezoelectric extracorporeal shock wave lithotripsy of parotid gland calculi. J Clin Ultrasound. 2001 Sep;29(7): 389-94
AHMAD A AL-SHARE, MAMOON M FNAISH, AHMAD M AL-ALAWNEH and MOHAMMAD JARAH
1,2,3,4 Royal Medical Services/Jordan Armed Forces Amman- Jordan, For Correspondence: Ahmad Ali Al-Share, Yarmouk University, P.O. Box 4837, lrbid-Jordan, Email: firstname.lastname@example.org
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|Author:||Al-Share, Ajmad A.; Fnaish, Mamoon M.; Al-Alawneh, Ahmad M.; Jarah, Mohammad|
|Publication:||Pakistan Oral and Dental Journal|
|Article Type:||Case study|
|Date:||Dec 31, 2011|
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