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The Laryngoscope V C 2010 The American Laryngological, Rhinological and Otological Society, Inc. Management of Giant Sialoliths: Review of the Literature and Preliminary Experience With Interventional Sialendoscopy Eric Wallace, BSc; Marcie Tauzin, MD; Joseph Hagan, PhD; Barry Schaitkin, MD; Rohan R. Walvekar,MD Objectives/Hypothesis: To report our experi- ence with management of giant salivary stones via a combined approach technique using salivary endos- copy (CA) and results of a review of current literature related to giant salivary stones. Study Design: Retrospective case series. Methods: An institutional review board- approved chart review was performed on patients man- aged with a CA to treat giant salivary stones (15 mm). In addition, we reviewed the English literature from 1942 to 2009. Results: Table 1 consists of 54 stones; 47 of which were identified during the review of literature and 7 from our case series. Of those 47 stones, 23 were hilar in location, 23 were glandular in location, and 1 stone was missing data. The gland preservation rate in the 47 reported stones was 57% (17/30). Among patients where gland resection was reported, the majority of the patients (12/13) had hilar glandular stones. Only one patient with a ductal stone had a gland resection. In our series, CA enabled a gland preservation rate of 86% (6/7). Among these patients, stone location was hilar glandular in six (86%) and ductal in one (14%). The mean size of stones from the literature review was 35 mm and from our series was 19 mm. Conclusions: Our review reflects current treat- ment recommendations for giant stones, i.e., transoral sialolithotomy for ductal stones and gland resection for hilar glandular stones. Our preliminary experi- ence with CA for giant stones suggests improved gland preservation rates (86% vs. 57%) independent of stone location and with preservation of salivary function. Key Words: Giant stones, megalith, salivary endoscopy, interventional sialendoscopy, salivary stone. Level of Evidence: 2b Laryngoscope, 120:1974–1978, 2010 INTRODUCTION Sialolithiasis is one of the most common non-neo- plastic disorders of the major salivary glands. The reported incidence of sialolithiasis in the general popula- tion is 1.2% 1 with the submandibular gland involved in the majority of cases (80%), 2 and men affected twice as commonly as women. 3 Sialoliths can range in size from a few millimeters to a few centimeters. Eighty-eight per- cent of salivary stones are less than 10 mm with the majority ranging from 3 mm to 7 mm. 3 Stones larger than 15 mm in length are called ‘‘giant stones’’ or ‘‘mega- liths,’’ and are relatively rare in occurrence. A few cases describing their presentation and management have been reported sporadically over the past century. 4 Stones have been shown to grow at a rate of 1 mm to 1.5 mm per year. 5 Consequently, it could be hypothesized that it could take at least 10 years to obtain a stone classified as a megalith. Traditional management of large salivary stones includes surgical excision of the gland, especially for glandular or hilar stones. Transoral sialolithotomy has been described for patients with large submandibular stones that are ductal in location and easily palpable in the floor of the mouth. This procedure becomes less pre- cise when stones are not palpable or are hilar or glandular in location. In these cases, an inadvertent injury to the lingual nerve is more likely. For giant pa- rotid stones, which are extremely rare, traditional management would include gland excision when con- servative treatment fails. Surgery of the parotid gland for inflammatory conditions, such as sialolithiasis and chronic recurrent sialadenitis, is associated with an increased risk of damage to the facial nerve. Submandib- ular gland resection is associated with the potential From the Department of Otolaryngology–Head Neck Surgery (E.W., M.T., R.R.W.) Louisiana State University Health Sciences Center, New Orleans, Louisiana; the Department of Biostatistics (J.H.), Louisiana State University School of Medicine, New Orleans, Louisiana; and the Department of Otolaryngology–Head and Neck Surgery (B.S.), University of Pittsburgh, Pittsburgh, Pennsylvania, U.S.A. Editor’s Note: This Manuscript was accepted for publication April 19, 2010. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Rohan R. Walvekar, MD, Assistant Profes- sor, Department of Otolaryngology–Head and Neck Surgery, Louisiana State University Health Sciences Center, 533 Bolivar Street, Suite 566, New Orleans, Louisiana 70112. E-mail: [email protected] DOI: 10.1002/lary.21082 Laryngoscope 120: October 2010 Wallace et al.: Management of Giant Sialoliths 1974

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Page 1: Management of giant sialoliths: Review of the literature and preliminary experience with interventional sialendoscopy

The LaryngoscopeVC 2010 The American Laryngological,Rhinological and Otological Society, Inc.

Management of Giant Sialoliths: Review ofthe Literature and Preliminary ExperienceWith Interventional Sialendoscopy

Eric Wallace, BSc; Marcie Tauzin, MD; Joseph Hagan, PhD; Barry Schaitkin, MD; Rohan R. Walvekar, MD

Objectives/Hypothesis: To report our experi-ence with management of giant salivary stones via acombined approach technique using salivary endos-copy (CA) and results of a review of current literaturerelated to giant salivary stones.

Study Design: Retrospective case series.Methods: An institutional review board-

approved chart review was performed on patients man-aged with a CA to treat giant salivary stones (�15mm). In addition, we reviewed the English literaturefrom 1942 to 2009.

Results: Table 1 consists of 54 stones; 47 of whichwere identified during the review of literature and 7from our case series. Of those 47 stones, 23 were hilarin location, 23 were glandular in location, and 1 stonewas missing data. The gland preservation rate in the47 reported stones was 57% (17/30). Among patientswhere gland resection was reported, the majority ofthe patients (12/13) had hilar glandular stones. Onlyone patient with a ductal stone had a gland resection.In our series, CA enabled a gland preservation rate of86% (6/7). Among these patients, stone location washilar glandular in six (86%) and ductal in one (14%).The mean size of stones from the literature review was35 mm and from our series was 19 mm.

Conclusions: Our review reflects current treat-ment recommendations for giant stones, i.e., transoralsialolithotomy for ductal stones and gland resectionfor hilar glandular stones. Our preliminary experi-ence with CA for giant stones suggests improvedgland preservation rates (86% vs. 57%) independent

of stone location and with preservation of salivaryfunction.

Key Words: Giant stones, megalith, salivaryendoscopy, interventional sialendoscopy, salivarystone.

Level of Evidence: 2bLaryngoscope, 120:1974–1978, 2010

INTRODUCTIONSialolithiasis is one of the most common non-neo-

plastic disorders of the major salivary glands. Thereported incidence of sialolithiasis in the general popula-tion is 1.2%1 with the submandibular gland involved inthe majority of cases (80%),2 and men affected twice ascommonly as women.3 Sialoliths can range in size froma few millimeters to a few centimeters. Eighty-eight per-cent of salivary stones are less than 10 mm with themajority ranging from 3 mm to 7 mm.3 Stones largerthan 15 mm in length are called ‘‘giant stones’’ or ‘‘mega-liths,’’ and are relatively rare in occurrence. A few casesdescribing their presentation and management havebeen reported sporadically over the past century.4 Stoneshave been shown to grow at a rate of 1 mm to 1.5 mmper year.5 Consequently, it could be hypothesized that itcould take at least 10 years to obtain a stone classifiedas a megalith.

Traditional management of large salivary stonesincludes surgical excision of the gland, especially forglandular or hilar stones. Transoral sialolithotomy hasbeen described for patients with large submandibularstones that are ductal in location and easily palpable inthe floor of the mouth. This procedure becomes less pre-cise when stones are not palpable or are hilar orglandular in location. In these cases, an inadvertentinjury to the lingual nerve is more likely. For giant pa-rotid stones, which are extremely rare, traditionalmanagement would include gland excision when con-servative treatment fails. Surgery of the parotid glandfor inflammatory conditions, such as sialolithiasis andchronic recurrent sialadenitis, is associated with anincreased risk of damage to the facial nerve. Submandib-ular gland resection is associated with the potential

From the Department of Otolaryngology–Head Neck Surgery (E.W.,M.T., R.R.W.) Louisiana State University Health Sciences Center, NewOrleans, Louisiana; the Department of Biostatistics (J.H.), Louisiana StateUniversity School of Medicine, New Orleans, Louisiana; and theDepartment of Otolaryngology–Head and Neck Surgery (B.S.), Universityof Pittsburgh, Pittsburgh, Pennsylvania, U.S.A.

Editor’s Note: This Manuscript was accepted for publication April19, 2010.

The authors have no funding, financial relationships, or conflictsof interest to disclose.

Send correspondence to Rohan R. Walvekar, MD, Assistant Profes-sor, Department of Otolaryngology–Head and Neck Surgery, LouisianaState University Health Sciences Center, 533 Bolivar Street, Suite 566,New Orleans, Louisiana 70112. E-mail: [email protected]

DOI: 10.1002/lary.21082

Laryngoscope 120: October 2010 Wallace et al.: Management of Giant Sialoliths

1974

Page 2: Management of giant sialoliths: Review of the literature and preliminary experience with interventional sialendoscopy

risks of damage to the lingual, hypoglossal, and mar-ginal mandibular nerves.

Our study evaluated the results of managing giantstones with a combined approach technique using sali-vary endoscopy (CA). A review of English literature from1942 to 2009 is also presented.

MATERIALS AND METHODSThe study is comprised of two components. First, we retro-

spectively reviewed the clinical data of seven patients whounderwent a CA at the University of Pittsburgh Medical Cen-ter, Pittsburgh, Pennsylvania, and Louisiana State UniversityHealth Sciences Center, New Orleans, Louisiana, from July 1,2005 to December 1, 2009. All patients included in our studywere managed by authors (b.s., r.r.w.) at their respective insti-tutions. Appropriate institutional review board approval wasobtained per institutional requirements. Demographic and clini-cal data were collected on all patients, including age, gender,

surgical indications, imaging characteristics, operative time,complications, and patient satisfaction. We defined complica-tions as major or minor as previously reported.6 The studycohort consisted of patients who presented with obstructive sali-vary symptoms related to a giant salivary stone (i.e., �15 mmin any one measured diameter determined either by preopera-tive computerized tomography [CT] scan or on grosspathological examination) (Fig. 1) who were managed withinterventional CA for stone removal as initially described byMarchal.7 The CA technique involves localization of the salivarystone using an interventional sialendoscope (Karl Storz, Tuttlin-gen, Germany) followed by a limited transoral incision todeliver the stone in submandibular cases or by an external pa-rotid approach to deliver parotid stones.

Transillumination from the sialendoscope enables localiza-tion and extraction of the stone with limited dissection andgland preservation.2 All patients were imaged preoperativelywith a CT scan without contrast. Specific information related tothe site (parotid or submandibular), location (ductal, hilar, orintraglandular), dimensions, and number of stones wasrecorded. Dimensions in millimeters of the length (maximumsize of the stone along the duct) and width (maximum size per-pendicular to the duct) were assessed and recorded. The secondcomponent of the study included a retrospective review of Eng-lish literature for articles reporting giant salivary stonesspanning from 1942 to 2009 using the PubMed Central searchengine and key words that included giant stone, megalith, largesalivary stone, and sialolithiasis. Forty-seven stones with infor-mation on stone size, location, side affected, treatment offered,and relevant demographic data were identified in 36 papers(Table I). Relevant combined data comprising information fromthe 47 literature-reviewed stones and seven stones from our se-ries are included.

RESULTSIn our series (n ¼ 7), the sex distribution was four

males and three females, and the mean age of presenta-tion was 48 years, (range, 15–69 years). The majority ofthe stones were hilar glandular in location (6/7), withfour being hilar and two being intraglandular. One stonewas intraductal. Stone sizes ranged from 15 mm to 25mm (mean, 19 mm). Five stones were located in the sub-mandibular gland and two were located in the parotidgland.

Mean operative time was 113 minutes (range, 80–180 minutes). All procedures lasted more than 1 hour(100%). Gland preservation rate was 6/7 patients (86%).All six patients with gland preservation were satisfiedwith the surgical outcome and had restored flow of sa-liva at last follow-up determined with observed salivaryflow with glandular compression, (mean, 48 days; range,0–182 days). One patient had recurrent symptomsrelated to a stenosis subsequent to the sialolithotomythat was managed with gland resection. Other complica-tions included a partial transaction of lingual nerve inone patient. There were no facial nerve injuries, hemato-mas, salivary fistulas, or postoperative infections.

Among the data from our systematic review (n ¼47), the sex distribution was nine females and 38 males,and the mean age at presentation was 51 years, (range,10–75 years). Stones were located most commonly in thesubmandibular gland (40/47; 85%), followed by the pa-rotid gland (4/47; 9%), then sublingual gland (1/47; 2%).

Fig. 1. (A) Axial computerized tomography scan showing a hilarglandular giant stone in the right submandibular gland. (B) Grosspathological image of the stone after removal using salivaryendoscopy.

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Page 3: Management of giant sialoliths: Review of the literature and preliminary experience with interventional sialendoscopy

TABLE I.

Systematic Review of Patients With Giant Salivary Stones (1942–2009) and Authors’ Case Series.

SR No. Gland Site Largest Measurement, mm Age, y Sex Gland Status

1 SMG Duct 16 61 Male Preserved

2 SMG Gland 20 43 Male Preserved

3 SMG Duct 20 52 Female Preserved

4 Parotid Duct 20 61 Male Preserved

5 SMG Duct 20 63 Female —

6 SMG Gland 20 70 Male Resected

7 SMG Duct 22 52 Female Preserved

8 SMG Duct 22 61 Male —

9 SMG Gland 22 61 Male Preserved

10 SMG Duct 25 46 Female Preserved

11 SMG Gland 25 50 Male Resected

12 SMG Duct 25 54 Female Preserved

13 SMG Duct 26 34 Male —

14 SMG Gland 26 46 Male Resected

15 SMG Gland 28 75 Male Resected

16 SMG Duct 30 45 Male Preserved

17 Parotid Duct 30 48 Female —

18 SMG Gland 30 56 Male Resected

19 SMG Gland 30 67 Male Resected

20 SMG Gland 31 55 Male —

21 SMG Duct 32 25 Male Preserved

22 SMG Duct 32 46 Male Preserved

23 SLG Gland 32 59 Male Resected

24 — Gland 33 65 Female —

25 SMG Gland 33 70 Male Resected

26 SMG Gland 34 58 Male —

27 SMG Duct 35 10 Female Preserved

28 SMG Duct 35 27 Male —

29 SMG Duct 35 49 Male Preserved

30 SMG Duct 35 55 Male Resected

31 SMG Duct 36 48 Male Preserved

32 SMG Gland 37 22 Female Preserved

33 SMG Gland 38 55 Male Resected

34 SMG Gland 45 45 Male Resected

35 SMG Gland 45 45 Male —

36 SMG Gland 45 52 Male —

37 SMG Gland 45 53 Male Resected

38 SMG Gland 50 48 Male —

39 SMG Gland 50 50 Male —

40 SMG Duct 50 50 Male Preserved

41 Parotid Gland 50 64 Male —

42 Parotid Duct 51 66 Male —

43 — — 52 65 Male —

44 SMG Duct 55 52 Male —

45 SMG Duct 56 42 Male —

46 SMG Gland 60 28 Male Resected

47 SMG Duct 72 60 Male Preserved

48* Parotid Gland 23 69 Male Preserved

49* Parotid Gland 23 69 Male Preserved

50* SMG Duct 15 46 Female Preserved

51* SMG Gland 15 55 Male Resected

52* SMG Gland 25 33 Male Preserved

53* SMG Gland 17 15 Female Preserved

54* SMG Gland 15 50 Female Preserved

See References 3–5, 9–42.*Patients within the current series.SMG ¼ submandibular gland; SR No. ¼ serial number.

Page 4: Management of giant sialoliths: Review of the literature and preliminary experience with interventional sialendoscopy

In two cases, stone site was not reported (2/47; 4%).Twenty-three stones were ductal in location (49%), 23stones hilar glandular, and in one case, the stone loca-tion was unknown (2%). Stone sizes ranged from 16 mmto 72 mm (mean, 35 mm). The gland preservation ratewas 57% (17/30). Data regarding gland preservation orresection were not reported for 17 giant stones.

In the combined data of 54 stones (47 reportedstones plus 7 current series), the mean diameter of thegiant stones measured 31.5 mm (range, 15–72 mm).

DISCUSSIONInterventional sialendoscopy is a relatively new

therapeutic approach for the management of salivarystones. To our knowledge, this is possibly the first reportaddressing management of the giant salivary stones ormegaliths using CA. Our series using CA techniquedemonstrates successful delivery of the megaliths with agland preservation rate of 86% at last follow-up. Thereview reported a gland preservation rate of 57%. Of the47 stones reported, 13 glands were excised and of these,12 stones were hilar glandular in location (92%). Thisreflects the traditional philosophy that was based on theprinciple that gland preservation was not considered fea-sible with stones located either in the hilum or in thegland. However, recent histopathological studies suggestthat salivary glands removed for sialolithiasis have nor-mal glandular architecture.1 In addition, nuclear scansof patients who have had sialendoscopic stone removalsuggest returning gland function at one year after endo-scopic removal.8

Consequently, gland preservation should be consid-ered whenever feasible. Our results suggest that CAenables gland preservation at a higher rate in patientswith giant salivary stones. We also documented an objec-tive measure of gland function in terms of return ofnormal salivary flow on postoperative evaluations.

In addition to gland preservation, the CA offerssome advantages over traditional sialolithotomy or glandexcision. The endoscope enables visualization and local-

ization of the stone that helps to perform a focuseddissection leading to the sialolith. The identification ofthe duct via the palpation of the scope and also thetransillumination facilitates identification of the lingualnerve in the floor of the mouth in submandibular casesand helps in defining the facial nerve branches by exclu-sion in parotid cases. In particular, it has been ourobservation that giant stones tend to displace thesenerves that are in close proximity to the salivary duct innormal circumstances. Consequently, sialolithotomy todeliver the stone in the case of megaliths is safer. Theone instance of partial lingual nerve resection was aniatrogenic complication. Other advantages that the CAtechnique offers are the capability to perform a distalendoscopy to check for residual stone fragments or addi-tional stones (Fig. 2). In cases where the salivary ductneeds to be repaired, the endoscope enables irrigation tocheck the site of the repair. The CA technique must beconsidered with caution in patients who have microsto-mia, small mouth opening, or trismus, and also in obesepatients.

CONCLUSIONThe result of our literature review provides a

glimpse of current recommendations for management ofgiant stones. These recommendations are based on thetraditional principles that suggest transoral sialolithot-omy for ductal stones, and gland resection for hilarglandular stones. Our preliminary experience with CAsuggests improved gland preservation rates (86% vs.57%) can be obtained independent of stone location andwith preservation of salivary flow.

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Fig. 2. Axial computerized tomography scanshowing a megalith in the floor of the mouthand an additional hilar stone or fragment ofthe giant stone that can be retrieved endo-scopically after salivary endoscopy.

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