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AM ER IC AN JOURNAL OF OT OLA RYNGOLOGYH E A D A N D NE CK M E D ICI N E AN D S U RGE RY 3 6 (2 0 1 5) 6 32 6 3 5

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Combined approach sialendoscopy for management


of submandibular gland sialolithiasis
Nofrat Schwartz, MD, 1 , Inbal Hazkani, MD 1 , Sivan Goshen, MD
Department of Otolaryngology, Meir Hospital, Kfar Saba, Israel
Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel

ARTI CLE I NFO

A BS TRACT

Article history:

Purpose: Sialolithiasis is the primary cause of obstructive sialadenitis, affecting the

Received 9 February 2015

submandibular gland in 8090% of cases. Sialendoscopy has dramatically changed the


diagnosis and management of salivary gland diseases. However, in cases in which
endoluminal removal via sialendoscopy is not successful, a combined approach using a
limited intraoral incision under guidance of sialendoscopy can facilitate stone removal. We
reviewed our institutions experience with combined approach sialendoscopy and
evaluated its role in managing sialolithiasis of the submandibular gland.
Materials and methods: Retrospective study of the treatment of sialolithiasis in the
submandibular gland via combined approach sialendoscopy from January 2010 through
March 2014. Demographics, clinical data, intraoperative findings and post-operative course
were reviewed.
Results: Most sialoliths (56.5%) were over 10 mm in size and were in the hilus of the gland
(56%). The success rate of the combined approach was 87%. No significant complications
were documented. Symptoms resolved in 75.7% of patients; however, this did not correlate
with placement of an intraductal stent (p=0.7) or steroid irrigation (p=0.1). An overall gland
preservation rate of 94.9% was achieved.
Conclusions: Combined approach sialendoscopy offers a minimally invasive technique for
treating refractory sialolithiasis not amenable to removal via sialendoscopy alone. The
procedure is well-tolerated, performed under local anesthesia with low morbidity and a
high success rate.
2015 Elsevier Inc. All rights reserved.

1.

Introduction

Obstructive sialadenitis is a common disease, representing


approximately one-half of benign salivary gland diseases [1].
The most common presentation is recurrent, painful, glandular swelling associated with eating, which can be complicated by bacterial superinfection and abscess formation.

Sialolithiasis is the main cause of obstructive sialadenitis,


affecting up to 1.2% of the general population [2]. The
submandibular gland is involved in 8090% of cases, followed
by the parotid (510%) and the sublingual glands (< 1%) [3].
Historically, obstructive sialadenitis was usually managed
conservatively, while surgical treatment was reserved for
refractory cases, ranging from papillotomy to sialadenectomy

Author Contributions: All authors contributed in the process of writing the article. Dr Schwartz and Dr Hazkani collected, reviewed and
analyzed the data. Dr Goshen performed all the combined approach sialendoscopy procedures and follow-up of the patients.
Corresponding author at: Department of Otolaryngology, Meir Medical Center, Kfar Saba 44281 Israel. Tel.: +972 544740811; fax: +972 3 6422051.
E-mail address: nofrat@gmail.com (N. Schwartz).
1
Co-first authors.

http://dx.doi.org/10.1016/j.amjoto.2015.04.001
0196-0709/ 2015 Elsevier Inc. All rights reserved.

AM ER IC AN JOURNAL OF OT OLARYNGOLOGYH E A D A N D NE CK M E D IC IN E A ND S U RGE RY 3 6 (2 0 1 5) 6 326 3 5

[4]. In the case of sialolithiasis, the former usually utilized for


distally located calculi and the latter for proximal or
intraglandular calculi. Although sialadenectomy is the definitive treatment for obstructive sialadenitis of any etiology, it
carries the highest rates of complications, which include
permanent nerve damage (facial, hypoglossal or lingual,
depending on the gland excised), salivary fistula, sialocele
and aesthetic sequela [5]. The introduction of sialendoscopy
has changed the diagnosis and management of salivary gland
diseases dramatically. Sialendoscopy offers a minimally
invasive approach that enables both better visualization and
diagnosis of the ductal system [6] and treatment of obstructive sialadenitis with considerably less morbidity [7]. Furthermore, a significant percentage of affected glands had a
normal histologic appearance and the gland regained its
function after stones were removed [8]. Small size, good mobility,
round or oval shape and distal location of the sialolith were
found to be positive predictive factors for sialendoscopic removal
[9]. For stones not amenable to endoluminal removal via
sialendoscopy, a combined approach using a limited intraoral
incision under guidance of sialendoscopy can facilitate stone
removal with minimal surgical morbidity. A meta-analysis
evaluating the efficacy of sialendoscopy determined that the
pooled success rate for interventional sialendoscopy alone was
86% and 93% for the combined approach, with a low incidence of
major complications [10] and recovery of secretory function [11].
There is a paucity of published data evaluating the efficacy
and safety of the combined approach for the treatment of
sialolithiasis. This study reviewed our institutions experience
with combined approach sialendoscopy and evaluated its role in
the treatment of sialolithiasis of the submandibular gland.

2.

Materials and methods

This retrospective analysis of 49 interventional, combined


approach sialendoscopies for the treatment of suspected
sialoliths in the submandibular glands was performed from
January 2010 through March 2014. The study was approved by
the Helsinki Ethics Committee of Meir Medical Center (permit
number MMC0007-14), under the supervision of the Israel
Ministry of Health. Informed consent was waived as the study
was retrospective. All procedures were performed by the senior
author (S.G.) at Meir Medical Center, which is a regional referral
center for the treatment of salivary gland diseases and
sialendoscopy. Patients who had successful sialolith removal
via sialendoscopy were excluded from the study. The presence
of comorbidities or the use of anticoagulation or antiplatelet
treatment was not considered a contraindication for the
procedure. Demographics, clinical data, intraoperative findings
and postoperative course were collected from the computerized
medical records. Demographic data included age, gender, and
systemic illness. Clinical data included obstructive symptoms,
need for antibiotic or hospitalization, imaging, glands involved
and prior procedures. Intraoperative findings, such as the
location and characteristics of the calculi were recorded. The
success rate was evaluated based on calculi removal, need for
further interventions or hospitalizations, complications, and
symptom resolution or recurrence on follow-up.

633

Diagnosis of sialolithiasis was based on symptomatology


and either a conformation with imaging (CT or US) or palpation
of the floor of the mouth. In our institution, sialendoscopy is
performed under local anesthesia. The punctum of the affected
gland is serially dilated under magnification using punctum
dilators until the sialendoscope (Polydiagnost, Pfaffenhofen,
Germany) can be introduced. The duct is visualized with the
endoscope throughout its length to assess for pathology, while
saline rinsing is performed to keep the duct patent with local
anesthetic rinsing. In cases of obstruction, sialolith removal is
first attempted without fragmentation using the basket, the
Fogarty balloon catheter to pull from behind, irrigation as a
mobilization technique or forceps. If a sialolith remains wedged
within the duct or is too large to be removed with these
techniques, a limited transoral incision is made in order to
deliver it using the sialendoscope transillumination to localize
and stabilize the sialolith. After its removal, a stent is sutured in
the duct and/or hydrocortisone rinsing is performed according
to the physicians discretion. Thereafter, the entire duct is reexplored for remnants. Postoperatively, the patients are prescribed antibiotics and followed-up after two weeks and then
serially, as needed.

2.1.

Statistical analysis

The data are presented as percentages of the population


studied. Continuous values are presented as mean SEM.
Chi-square or Fishers exact test was performed to compare
discrete categorical variables. p<0.05 was considered statistically significant. All statistical analyses were performed using
GraphPad Prism software, version 5.04 2010.

Table 1 Clinical and operative data in patients


undergoing combined approach sialendoscopy.
Patient information

Number of cases

Age (years)
Male:female
Prior to intervention
Bilateral gland involvement
Number of sialoliths >1 in imaging
Obstructive symptoms
Antibiotic treatment
Hospitalization
Intraoperative
Location of sialolith
Hilum
Major duct
Minor duct
Size >10 mm
Success of removal
Ancillary treatment
Steroid irrigation
Stent
Postoperative
Follow-up (months)
Complication
Hospitalization
Symptom resolution
Gland preservation

463
26:13
4/39
11/39
38/39
26/39
5/39

26/46
16/46
1/46
26/46
40/46
14/46
10/46
8.31.5
2/49
5/39
28/37
37/39

T1

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AM ER IC AN JOURNAL OF OT OLA RYNGOLOGYH E A D A N D NE CK M E D ICI N E AN D S U RGE RY 3 6 (2 0 1 5) 6 32 6 3 5

3.

Results

A total of 49 procedures were performed in 39 patients (Table 1).


Of these, 4 patients had bilateral submandibular gland disease
and 5 underwent more than one intervention. The average age
was 463 years and two-thirds of the patients were male (26/39).
Almost all (38/39) had obstructive symptoms prior to the
intervention and in 66.7% (26/39) received antibiotics. However,
only 12.8% (5/39) required hospitalization. As there was no
standardization and large variability in the assessment of size
of sialoliths on imaging, especially in outpatients, the results
are not shown. More than one sialolith was seen on imaging in
28% (11/39) and 18% (7/39) had more than 2.
Diagnostic sialendoscopy initially identified a sialolith in 46
of the 49 cases. A stricture was identified in 4 cases and debris
was recorded in the ductal system in addition to the sialolith in
6. Most sialoliths were identified in the hilus of the gland (26/
46). The remainder were found in the major duct (16/46) and the
minor ducts (1 case). The location was not documented in 3
cases. In 26 cases, the stone was estimated over 10 mm in size
(56.5%). Additional steroid irrigation and stent placement were
performed in 14 and 10 cases, respectively.
The success rate of the combined approach in cases where a
sialolith was identified was 87% (40/46). Failure resulted mainly
due to either the location of the sialolith or its relative size.
However, due to the low number of unsuccessful extractions this
was not statistically significant (p=0.2). Furthermore, documentation of an infection in the gland that required antibiotic
treatment prior to the procedure did not correlate with successful
sialolith extraction (p=0.16). No significant intraoperative complications were documented. Patients were followed for a mean
of 8.31.5 months (2 patients were lost to follow-up). Symptoms
resolved in 75.7% of patients (28/37). Resolution of obstructive
symptoms did not correlate to placement of an intraductal stent
(p=0.7) or steroid irrigation (p=0.1). Complications were rare and
included temporary lingual paresthesia (1/49) and a draining
sinus to the skin (1/49). Five patients required brief hospitalization after the procedure, for antibiotic treatment. Of the 9 patients
whose symptoms did not resolve, 2 underwent successful
combined approach sialendoscopy under general anesthesia
and 2 had a sialadenectomy performed. Thus, overall gland
preservation rate was 94.9% (37/39).

4.

Discussion

It is accepted that submandibular gland sialoliths larger than


4 mm are unfavorable for sialendoscopic removal, which
usually requires ancillary techniques [12,13]. Combined approach sialendoscopy offers a minimally invasive technique
for the treatment of refractory sialolithiasis not amenable for
removal via sialendoscopy alone. The present study, to our
knowledge, is one of the largest series to date specifically
addressing submandibular sialolithiasis necessitating treatment with combined approach sialendoscopy. It attempts to
characterize this population better and to provide answers
regarding the safety and effectiveness of the procedure.
Analysis of our institutions experience reveals a success rate
of 87% with the combined approach for sialoliths that were not

amenable for removal via sialendoscopy due to size or location.


This finding is in concordance with previous studies reporting
success rates ranging from 86 to 93% [13]. Most reported cases of
sialolithiasis referred for combined approach after unsuccessful
endoluminal endoscopic removal were sialoliths found in the
intraglandular hilus [11,14,15]. Accordingly, in the current study,
more than half the sialoliths were located in the hilus. Furthermore, more than half of the sialoliths were larger than 15 mm,
which is more than twice the size reported as unsuitable for
sialendoscopic removal alone [13]. These differences might
explain our rate of successful removal of the sialoliths, which
was not as high as reported in some of the studies, due to possible
selection bias of cases referred to our institution that are more
refractory to treatment.
The combined approach technique offers the advantage
of localization, stabilization and manipulation of the sialolith
in the duct, aiding in its removal, and the opportunity to
explore the ductal system for additional sialoliths or fragments. Indeed, in our experience, 28% of the patients had
more than one sialolith that required treatment during the
procedure. Despite the observation of ductal system changes
in long-standing infected glands [7], no correlation between
past events of sialadenitis and the success rate of removal of
sialoliths was found.
Resolution of symptoms was documented in 75.7% of
patients. However, removal of the gland was necessary in only
2 patients corresponding to a gland preservation rate of 94.9%.
Overall, the frequency and morbidity of the complications of
the combined approach compare favorably to the complications associated with more invasive procedures. In accordance
with previous studies, the complication rate was low and
consisted of post-operative swelling, one case of temporary
lingual paresthesia and fistula formation [10,11,14,15]. While
Wharton ductoplasty is not customary [16], there is no
consensus on the indication for or effectiveness of postoperative stenting and intraductal corticosteroid injection to prevent
stricture or stenosis formation [10]. We did not find any
correlation between either stent placement or steroid irrigation
and resolution of symptoms. Further evaluation of the efficacy
and safety of these adjuvant treatments is warranted, to aid in
determining their utility.
In conclusion, combined approach sialendoscopy offers
patients with sialolithiasis who failed all other conservative
and minimally invasive techniques, including extracorporeal
or intracorporeal fragmentation sialendoscopy, a last resort
before the need to sacrifice the gland. It is a well-tolerated
procedure performed under local anesthesia with low
morbidity and a high success rate. Functional gland recovery
after the removal of sialoliths via the combined approach
[8,11,17] emphasizes the efficacy of this modality for treating
recalcitrant sialolithiasis, sparing the need for removal of
the gland.

Acknowledgment
All authors had full access to all of the data in the study and take
responsibility for the integrity of the data and the accuracy of the
data analysis. All information and materials in the manuscript
are original and have not been published previously.

AM ER IC AN JOURNAL OF OT OLARYNGOLOGYH E A D A N D NE CK M E D IC IN E A ND S U RGE RY 3 6 (2 0 1 5) 6 326 3 5

We state that no funding was received from any organization and that there was no conflict of interest while
conducting the study or in the process of preparing the article.

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