Combined Approach Sialendoscopy for Management of Submandibular Gland Sialolthiasis Nofrat Schwartz MD, Inbal Hazkani MD, Sivan Goshen MD PII: DOI: Reference:
S0196-0709(15)00087-3 doi: 10.1016/j.amjoto.2015.04.001 YAJOT 1565
To appear in:
American Journal of Otolaryngology–Head and Neck Medicine and Surgery
Received date: Accepted date:
9 February 2015 3 April 2015
Please cite this article as: Schwartz Nofrat, Hazkani Inbal, Goshen Sivan, Combined Approach Sialendoscopy for Management of Submandibular Gland Sialolthiasis, American Journal of Otolaryngology–Head and Neck Medicine and Surgery (2015), doi: 10.1016/j.amjoto.2015.04.001
This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Combined Approach Sialendoscopy for Management of
T
Submandibular Gland Sialolthiasis
RI P
Nofrat Schwartz MD, Inbal Hazkani MD, Sivan Goshen MD Department of Otolaryngology, Meir Hospital, Kfar Saba, Israel and
MA NU
SC
Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel
Correspondence: Nofrat Schwartz MD
ED
Department of Otolaryngology Meir Medical Center
PT
Kfar Saba 44281 Israel
CE
Tel: 972-544740811 Fax: 972-3-6422051
AC
Email: [email protected]
Key Words: Sialolithiasis, Sialadenitis, Sialendoscopy, Combined approach Word Count: 1715
ACCEPTED MANUSCRIPT Abstract Purpose: Sialolithiasis is the primary cause of obstructive sialadenitis, affecting the
T
submandibular gland in 80-90% of cases. Sialendoscopy has dramatically changed
RI P
the diagnosis and management of salivary gland diseases. However, in cases in which endoluminal removal via sialendoscopy is not successful, a combined approach using
SC
a limited intraoral incision under guidance of sialendoscopy can facilitate stone removal. We reviewed our institution's experience with combined approach
MA NU
sialendoscopy and evaluated its role in managing sialolithiasis of the submandibular gland.
Materials and Methods: Retrospective study of the treatment of sialolithiasis in the
ED
submandibular gland via combined approach sialendoscopy from January 2010 through March 2014. Demographics, clinical data, intraoperative findings and post-
PT
operative course were reviewed.
Results: Most sialoliths (56.5%) were over 10 mm in size and were in the hilus of the
CE
gland (56%). The success rate of the combined approach was 87%. No significant
AC
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 sialoendoscopy alone. The procedure is well-tolerated, performed under local anesthesia with low morbidity and a high success rate.
ACCEPTED MANUSCRIPT 1.1 Introduction Obstructive sialadenitis is a common disease, representing approximately one-half of
T
benign salivary gland diseases.[1] The most common presentation is recurrent,
RI P
painful, glandular swelling associated with eating, which can be complicated by bacterial superinfection and abscess formation. Sialolithiasis is the main cause of
SC
obstructive sialadenitis, affecting up to 1.2% of the general population.[2] The submandibular gland is involved in 80-90% of cases, followed by the parotid (5-10%)
MA NU
and the sublingual glands (< 1%).[3]
Historically, obstructive sialadenitis was usually managed conservatively, while surgical treatment was reserved for refractory cases, ranging from papilotomy to
ED
sialadenectomy.[4] In the case of sialolithiasis, the former usually utilized for distally located calculi and the latter for proximal or intraglandular calculi. Although
PT
sialadenectomy is the definitive treatment for obstructive sialadenitis of any etiology, it carries the highest rates of complications, which include permanent nerve damage
CE
(facial, hypoglossal or lingual, depending on the gland excised), salivary fistula,
AC
sialocele and aesthetic sequela.[5] The introduction of sialoendoscopy 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
ACCEPTED MANUSCRIPT can facilitate stone removal with minimal surgical morbidity. A meta-analysis evaluating the efficacy of sialendoscopy determined that the pooled success rate for
T
interventional sialendoscopy alone was 86% and 93% for the combined approach,
RI P
with a low incidence of major complications[10] and recovery of secretory function.[11]
SC
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
MA NU
institution's experience with combined approach sialendoscopy and evaluated its role in the treatment of sialolithiasis of the submandibular gland.
ED
1.2 Materials and Methods
This retrospective analysis of 49 interventional, combined approach sialendoscopies
PT
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
CE
Ethics Committee of Meir Medical Center (permit number MMC0007-14), under the
AC
supervision of the Israel Ministry of Health. Informed consent was waived as the study was retrospective. All procedures were performed by the senior author (G,S.) 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
ACCEPTED MANUSCRIPT procedures. Intraoperative findings, such as the location and characteristics of the calculi were recorded. The success rate was evaluated based on calculi removal, need
T
for further interventions or hospitalizations, complications, and symptom resolution or
RI P
recurrence on follow-up.
Diagnosis of sialolithiasis was based on symptomatology and either a conformation
SC
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
MA NU
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
ED
is performed to keep the duct patent with local anesthetic rinsing. In cases of obstruction, sialolith removal is first attempted without fragmentation using the
PT
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
CE
removed with these techniques, a limited transoral incision is made in order to deliver
AC
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 physician’s 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. 1.2.1 Statistical analysis The data are presented as percentages of the population studied. Continuous values are presented as mean ± SEM. Chi-square or Fisher’s exact test were 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.
ACCEPTED MANUSCRIPT
1.3 Results
T
A total of 49 procedures were performed in 39 patients (Table 1). Of these, 4 patients
RI P
had bilateral submandibular gland disease and 5 underwent more than one intervention. The average age was 46 ±3 years and two-thirds of the patients were
SC
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
MA NU
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%
ED
(7/39) had more than 2.
Diagnostic sialendoscopy initially identified a sialolith in 46 of the 49 cases. A
PT
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
CE
(26/46). The remainder were found in the major duct (16/46) and the minor ducts (1
AC
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 8.3±1.5 months (2 patients
ACCEPTED MANUSCRIPT 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)
T
or steroid irrigation (p=0.1). Complications were rare and included temporary lingual
RI P
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
SC
symptoms did not resolve, 2 underwent successful combined approach sialendoscopy under general anesthesia and 2 had a sialadenectomy performed. Thus, overall gland
MA NU
preservation rate was 94.9% (37/39).
1.4 Discussion
ED
It is accepted that submandibular gland sialoliths larger than 4 mm are unfavorable for sialendoscopic removal, which usually requires ancillary techniques.[12,13]
PT
Combined approach sialoendoscopy offers a minimally invasive technique for the treatment of refractory sialolithiasis not amenable for removal via sialendoscopy
CE
alone. The present study, to our knowledge, is one of the largest series to date
AC
specifically addressing submandibular sialolithiasis necessitating treatment with combined approach sialoendoscopy. It attempts to characterize this population better and to provide answers regarding the safety and effectiveness of the procedure. Analysis of our institution’s experience reveals a success rate of 87% with the combined approach for sialoliths that were not amenable for removal via sialoendoscopy 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
ACCEPTED MANUSCRIPT 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]
T
These differences might explain our rate of successful removal of the sialoliths, which
RI P
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.
SC
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
MA NU
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-
ED
standing infected glands,[7] no correlation between past events of sialadenitis and the success rate of removal of sialoliths was found.
PT
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
CE
of 94.9%. Overall, the frequency and morbidity of the complications of the combined
AC
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.
ACCEPTED MANUSCRIPT In conclusion, combined approach sialendoscopy offers patients with sialolithiasis who failed all other conservative and minimally invasive techniques, including
T
extracorporeal or intracorporeal fragmentation sialendoscopy, a last resort before the
RI P
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
SC
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
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the gland.
1.5 Acknowledgment
ED
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
PT
performed all the combined approach sialendoscopy procedures and follow up of the patients.
CE
All authors had full access to all of the data in the study and take responsibility for the
AC
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. 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.
ACCEPTED MANUSCRIPT
References
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1. Harrison JD. Causes, natural history, and incidence of salivary stones and
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obstructions. Otolaryngol Clin North Am. 2009. 42(6):927-947. 2. Rauch S, Gorlin RJ. Diseases of the salivary glands. Oral Pathology, 6th ed.
SC
St. Louis, MO: Mosby. 1970. 997-1003.
3. Bodner L. Salivary gland calculi: diagnostic imaging and surgical
MA NU
management. Compendium. 1993; 14: 572-586.
4. Marchal F, Kurt AM, Dulguerov P, Lehmann W. Retrograde theory in sialolithiasis formation. Arch Otolaryngol Head Neck Surg. 2001;127(1): 66-
ED
68.
5. Capaccio P, Torretta S, Pignataro L. The role of adenectomy for salivary gland
PT
obstructions in the era of sialoendoscopy and lithotripsy. Otolaryngol Clin North Am. 2009;42(6):1161-1171.
CE
6. Hasson O. Sialoendoscopy and sialography: Strategies for assessment and
AC
treatment of salivary gland obstructions. J Oral Maxillofac Surg. 2007; 65(2):300-304.
7. Nahlieli O, Nakar LH, Nazarian Y, Turner MD. Sialoendoscopy: A new approach to salivary gland obstructive pathology. J Am Dent Assoc. 2006; 137(10):1394-1400. 8. Makdissi J, Escudiern MP, Brown JE, Osailan S, Drage N, McGurk M. Glandular function after intraoral removal of salivary calculi from the hilum of the submandibular gland. Br J Oral Maxillofac Surg. 2004;42(6):538-541.
ACCEPTED MANUSCRIPT 9. Luers JC, Grosheva M, Stenner M, Beutner D. Sialoendoscopy: prognostic factors for endoscopic removal of salivary stones. Arch Otolaryngol Head
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Neck Surg. 2011;137(4): 325-329.
RI P
10. Strychowsky JE, Sommer DD, Gupta MK, Cohen N, Nahlieli O. Sialendoscopy for the management of obstructive salivary gland disease: a
SC
systematic review and meta-analysis. Arch Otolaryngol Head Neck Surg. 2012;138(6): 541-547.
MA NU
11. Liu DG, Jiang L, Xie XY, Zhang ZY, Zhang L, Yu GY. Sialoendocopy assisted sialolithectomy for submandibular Hilar Calculi. J Oral Maxillofacial surgery. 2013;71:295-301.
ED
12. Marchal F, Dulguerov P. Sialolithiasis management: the state of the art. Arch Otolaryngol Head Neck Surg. 2003;129(9): 951-956.
PT
13. Marchal F.A combined endoscopic and external approach for extraction of large stones with preservation of parotid and submandibular glands.
CE
Laryngoscope. 2007;117(2):373-377.
AC
14. Walvekar RR, Bomeli SR, Carrau RL, Schaitkin B. Combined approach technique for the management of large salivary stones. Laryngoscope. 2009;119(6):1125-1129.
15. Wallace E, Tauzin M, Hagan J, Schaitkin B, Walvekar RR. Management of giant sialoliths: review of the literature and preliminary experience with interventional sialoendoscopy. Laryngoscope. 2010;120(10):1974-198. 16. Pagliuca G, Martellucci S, de Vincentiis M, Greco A, Fusconi M, De Virgilio A, et al. Wharton's duct repair after combined sialolithectomy: is ductoplasty necessary? Otolaryngol Head Neck Surg. 2013;148(5):775-777.
ACCEPTED MANUSCRIPT 17. Su YX, Xu JH, Liao GQ, Zheng GS, Cheng MH, Han L et al. Salivary gland
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functional recovery after sialendoscopy. Laryngoscope. 2009;119(4):646-652.
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Table 1. Clinical and operative data in patients undergoing combined approach sialendoscopy
Number of cases
Age (years)
46±3
Male: Female
26:13
4/39
Number of sialoliths › 1 in imaging
11/39
ED
Bilateral gland involvement
MA NU
Prior to intervention
38/39
Obstructive symptoms
26/39 5/39
CE
Hospitalization
PT
Antibiotic treatment
Intraoperative
Location of sialolith
AC
SC
Patient information
Hilum
26/46
Major duct
16/46
Minor duct
1/46
Size › 10 mm
26/46
Success of removal
40/46
Ancillary treatment Steroid irrigation
14/46
Stent
10/46
ACCEPTED MANUSCRIPT Postoperative 8.3±1.5
Complication
2/49
Hospitalization
5/39
Symptom resolution
28/37
Gland preservation
37/39
AC
CE
PT
ED
MA NU
SC
RI P
T
Follow up (months)
S0196-0709(15)00087-3 doi: 10.1016/j.amjoto.2015.04.001 YAJOT 1565
To appear in:
American Journal of Otolaryngology–Head and Neck Medicine and Surgery
Received date: Accepted date:
9 February 2015 3 April 2015
Please cite this article as: Schwartz Nofrat, Hazkani Inbal, Goshen Sivan, Combined Approach Sialendoscopy for Management of Submandibular Gland Sialolthiasis, American Journal of Otolaryngology–Head and Neck Medicine and Surgery (2015), doi: 10.1016/j.amjoto.2015.04.001
This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Combined Approach Sialendoscopy for Management of
T
Submandibular Gland Sialolthiasis
RI P
Nofrat Schwartz MD, Inbal Hazkani MD, Sivan Goshen MD Department of Otolaryngology, Meir Hospital, Kfar Saba, Israel and
MA NU
SC
Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel
Correspondence: Nofrat Schwartz MD
ED
Department of Otolaryngology Meir Medical Center
PT
Kfar Saba 44281 Israel
CE
Tel: 972-544740811 Fax: 972-3-6422051
AC
Email: [email protected]
Key Words: Sialolithiasis, Sialadenitis, Sialendoscopy, Combined approach Word Count: 1715
ACCEPTED MANUSCRIPT Abstract Purpose: Sialolithiasis is the primary cause of obstructive sialadenitis, affecting the
T
submandibular gland in 80-90% of cases. Sialendoscopy has dramatically changed
RI P
the diagnosis and management of salivary gland diseases. However, in cases in which endoluminal removal via sialendoscopy is not successful, a combined approach using
SC
a limited intraoral incision under guidance of sialendoscopy can facilitate stone removal. We reviewed our institution's experience with combined approach
MA NU
sialendoscopy and evaluated its role in managing sialolithiasis of the submandibular gland.
Materials and Methods: Retrospective study of the treatment of sialolithiasis in the
ED
submandibular gland via combined approach sialendoscopy from January 2010 through March 2014. Demographics, clinical data, intraoperative findings and post-
PT
operative course were reviewed.
Results: Most sialoliths (56.5%) were over 10 mm in size and were in the hilus of the
CE
gland (56%). The success rate of the combined approach was 87%. No significant
AC
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 sialoendoscopy alone. The procedure is well-tolerated, performed under local anesthesia with low morbidity and a high success rate.
ACCEPTED MANUSCRIPT 1.1 Introduction Obstructive sialadenitis is a common disease, representing approximately one-half of
T
benign salivary gland diseases.[1] The most common presentation is recurrent,
RI P
painful, glandular swelling associated with eating, which can be complicated by bacterial superinfection and abscess formation. Sialolithiasis is the main cause of
SC
obstructive sialadenitis, affecting up to 1.2% of the general population.[2] The submandibular gland is involved in 80-90% of cases, followed by the parotid (5-10%)
MA NU
and the sublingual glands (< 1%).[3]
Historically, obstructive sialadenitis was usually managed conservatively, while surgical treatment was reserved for refractory cases, ranging from papilotomy to
ED
sialadenectomy.[4] In the case of sialolithiasis, the former usually utilized for distally located calculi and the latter for proximal or intraglandular calculi. Although
PT
sialadenectomy is the definitive treatment for obstructive sialadenitis of any etiology, it carries the highest rates of complications, which include permanent nerve damage
CE
(facial, hypoglossal or lingual, depending on the gland excised), salivary fistula,
AC
sialocele and aesthetic sequela.[5] The introduction of sialoendoscopy 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
ACCEPTED MANUSCRIPT can facilitate stone removal with minimal surgical morbidity. A meta-analysis evaluating the efficacy of sialendoscopy determined that the pooled success rate for
T
interventional sialendoscopy alone was 86% and 93% for the combined approach,
RI P
with a low incidence of major complications[10] and recovery of secretory function.[11]
SC
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
MA NU
institution's experience with combined approach sialendoscopy and evaluated its role in the treatment of sialolithiasis of the submandibular gland.
ED
1.2 Materials and Methods
This retrospective analysis of 49 interventional, combined approach sialendoscopies
PT
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
CE
Ethics Committee of Meir Medical Center (permit number MMC0007-14), under the
AC
supervision of the Israel Ministry of Health. Informed consent was waived as the study was retrospective. All procedures were performed by the senior author (G,S.) 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
ACCEPTED MANUSCRIPT procedures. Intraoperative findings, such as the location and characteristics of the calculi were recorded. The success rate was evaluated based on calculi removal, need
T
for further interventions or hospitalizations, complications, and symptom resolution or
RI P
recurrence on follow-up.
Diagnosis of sialolithiasis was based on symptomatology and either a conformation
SC
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
MA NU
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
ED
is performed to keep the duct patent with local anesthetic rinsing. In cases of obstruction, sialolith removal is first attempted without fragmentation using the
PT
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
CE
removed with these techniques, a limited transoral incision is made in order to deliver
AC
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 physician’s 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. 1.2.1 Statistical analysis The data are presented as percentages of the population studied. Continuous values are presented as mean ± SEM. Chi-square or Fisher’s exact test were 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.
ACCEPTED MANUSCRIPT
1.3 Results
T
A total of 49 procedures were performed in 39 patients (Table 1). Of these, 4 patients
RI P
had bilateral submandibular gland disease and 5 underwent more than one intervention. The average age was 46 ±3 years and two-thirds of the patients were
SC
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
MA NU
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%
ED
(7/39) had more than 2.
Diagnostic sialendoscopy initially identified a sialolith in 46 of the 49 cases. A
PT
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
CE
(26/46). The remainder were found in the major duct (16/46) and the minor ducts (1
AC
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 8.3±1.5 months (2 patients
ACCEPTED MANUSCRIPT 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)
T
or steroid irrigation (p=0.1). Complications were rare and included temporary lingual
RI P
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
SC
symptoms did not resolve, 2 underwent successful combined approach sialendoscopy under general anesthesia and 2 had a sialadenectomy performed. Thus, overall gland
MA NU
preservation rate was 94.9% (37/39).
1.4 Discussion
ED
It is accepted that submandibular gland sialoliths larger than 4 mm are unfavorable for sialendoscopic removal, which usually requires ancillary techniques.[12,13]
PT
Combined approach sialoendoscopy offers a minimally invasive technique for the treatment of refractory sialolithiasis not amenable for removal via sialendoscopy
CE
alone. The present study, to our knowledge, is one of the largest series to date
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specifically addressing submandibular sialolithiasis necessitating treatment with combined approach sialoendoscopy. It attempts to characterize this population better and to provide answers regarding the safety and effectiveness of the procedure. Analysis of our institution’s experience reveals a success rate of 87% with the combined approach for sialoliths that were not amenable for removal via sialoendoscopy 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
ACCEPTED MANUSCRIPT 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]
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These differences might explain our rate of successful removal of the sialoliths, which
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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.
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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
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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-
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standing infected glands,[7] no correlation between past events of sialadenitis and the success rate of removal of sialoliths was found.
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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
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of 94.9%. Overall, the frequency and morbidity of the complications of the combined
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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.
ACCEPTED MANUSCRIPT In conclusion, combined approach sialendoscopy offers patients with sialolithiasis who failed all other conservative and minimally invasive techniques, including
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extracorporeal or intracorporeal fragmentation sialendoscopy, a last resort before the
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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
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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
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the gland.
1.5 Acknowledgment
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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
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performed all the combined approach sialendoscopy procedures and follow up of the patients.
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All authors had full access to all of the data in the study and take responsibility for the
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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. 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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References
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1. Harrison JD. Causes, natural history, and incidence of salivary stones and
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obstructions. Otolaryngol Clin North Am. 2009. 42(6):927-947. 2. Rauch S, Gorlin RJ. Diseases of the salivary glands. Oral Pathology, 6th ed.
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St. Louis, MO: Mosby. 1970. 997-1003.
3. Bodner L. Salivary gland calculi: diagnostic imaging and surgical
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management. Compendium. 1993; 14: 572-586.
4. Marchal F, Kurt AM, Dulguerov P, Lehmann W. Retrograde theory in sialolithiasis formation. Arch Otolaryngol Head Neck Surg. 2001;127(1): 66-
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5. Capaccio P, Torretta S, Pignataro L. The role of adenectomy for salivary gland
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obstructions in the era of sialoendoscopy and lithotripsy. Otolaryngol Clin North Am. 2009;42(6):1161-1171.
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6. Hasson O. Sialoendoscopy and sialography: Strategies for assessment and
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treatment of salivary gland obstructions. J Oral Maxillofac Surg. 2007; 65(2):300-304.
7. Nahlieli O, Nakar LH, Nazarian Y, Turner MD. Sialoendoscopy: A new approach to salivary gland obstructive pathology. J Am Dent Assoc. 2006; 137(10):1394-1400. 8. Makdissi J, Escudiern MP, Brown JE, Osailan S, Drage N, McGurk M. Glandular function after intraoral removal of salivary calculi from the hilum of the submandibular gland. Br J Oral Maxillofac Surg. 2004;42(6):538-541.
ACCEPTED MANUSCRIPT 9. Luers JC, Grosheva M, Stenner M, Beutner D. Sialoendoscopy: prognostic factors for endoscopic removal of salivary stones. Arch Otolaryngol Head
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Neck Surg. 2011;137(4): 325-329.
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10. Strychowsky JE, Sommer DD, Gupta MK, Cohen N, Nahlieli O. Sialendoscopy for the management of obstructive salivary gland disease: a
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11. Liu DG, Jiang L, Xie XY, Zhang ZY, Zhang L, Yu GY. Sialoendocopy assisted sialolithectomy for submandibular Hilar Calculi. J Oral Maxillofacial surgery. 2013;71:295-301.
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12. Marchal F, Dulguerov P. Sialolithiasis management: the state of the art. Arch Otolaryngol Head Neck Surg. 2003;129(9): 951-956.
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13. Marchal F.A combined endoscopic and external approach for extraction of large stones with preservation of parotid and submandibular glands.
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Laryngoscope. 2007;117(2):373-377.
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14. Walvekar RR, Bomeli SR, Carrau RL, Schaitkin B. Combined approach technique for the management of large salivary stones. Laryngoscope. 2009;119(6):1125-1129.
15. Wallace E, Tauzin M, Hagan J, Schaitkin B, Walvekar RR. Management of giant sialoliths: review of the literature and preliminary experience with interventional sialoendoscopy. Laryngoscope. 2010;120(10):1974-198. 16. Pagliuca G, Martellucci S, de Vincentiis M, Greco A, Fusconi M, De Virgilio A, et al. Wharton's duct repair after combined sialolithectomy: is ductoplasty necessary? Otolaryngol Head Neck Surg. 2013;148(5):775-777.
ACCEPTED MANUSCRIPT 17. Su YX, Xu JH, Liao GQ, Zheng GS, Cheng MH, Han L et al. Salivary gland
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functional recovery after sialendoscopy. Laryngoscope. 2009;119(4):646-652.
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Table 1. Clinical and operative data in patients undergoing combined approach sialendoscopy
Number of cases
Age (years)
46±3
Male: Female
26:13
4/39
Number of sialoliths › 1 in imaging
11/39
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Bilateral gland involvement
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Prior to intervention
38/39
Obstructive symptoms
26/39 5/39
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Hospitalization
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Antibiotic treatment
Intraoperative
Location of sialolith
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Patient information
Hilum
26/46
Major duct
16/46
Minor duct
1/46
Size › 10 mm
26/46
Success of removal
40/46
Ancillary treatment Steroid irrigation
14/46
Stent
10/46
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Complication
2/49
Hospitalization
5/39
Symptom resolution
28/37
Gland preservation
37/39
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Follow up (months)
