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PATHOLOGY

Sialadenitis Without Sialolithiasis Treated by Sialendoscopy Christopher G. Pace, DMD,* Kyung-Gyun Hwang, DDS, PhD,y Maria E. Papadaki, MD, DMD, PhD,z and Maria J. Troulis, DDS, MScx

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Purpose:

The aim of the present study was to evaluate the effectiveness of interventional sialendoscopy in the management of non–stone obstructive sialadenitis.

Patients and Methods:

In the present retrospective study, we assessed the treatment outcomes of 51 patients (34 women and 17 men) who had been diagnosed with sialadenitis without salivary duct stones. The included patients had an obstructive duct condition coupled with recurrent episodes of swelling or pain in a major salivary gland. The patients who had a sialolith detected during the sialendoscopic procedure or by preoperative imaging were excluded from the present study. The outcomes assessment was based on successful removal of the obstructive etiology during the endoscopic procedure and the absence of clinical symptoms after 6 months. The involved glands included 16 submandibular glands and 35 parotid glands.

Results:

The sialendoscopic navigation was successful in 43 of 51 patients (84%). Of the 43 patients, 37 (86%) were free of symptoms from the obstructive gland after the endoscopic procedure. Of the 6 patients who were still symptomatic after the procedure, 5 had involvement of the parotid gland. Finally, 1 of the original 37 symptom-free patients developed recurrent symptoms and denied additional treatment, resulting in 36 of 43 patients (84%) remaining asymptomatic 6 months after endoscopic navigation.

Conclusion:

The outcomes of the present study suggest that interventional sialendoscopy can provide symptomatic improvement in most subjects. Clinicians should consider sialendoscopy as a useful and minimally invasive procedure to treat sialadenitis without sialolithiasis. Ó 2015 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg -:1-4, 2015

Chronic obstruction of the major salivary gland architecture is a common condition. The affected patients will complain of intermittent pain and swelling in the gland, which are exacerbated by prandial salivary stimulation. Sialolithiasis is the primary etiology of this obstruction; however, nonspecific sialadenitis is chronic obstruction of the duct without the presence of a stone. Some infectious or autoimmune diseases and radioiodine therapy can cause sialadenitis from stenosis of the salivary gland duct. The etiology and mechanism of this entity remain unclear.1-3

Traditionally, ‘‘conservative’’ management or surgical intervention was performed to treat obstructive salivary gland disease. However, symptomatic treatment such as antibiotics, sialogogues, and ductal irrigation with saline has had limited success in resolving the obstructive swelling.2,4,5 Surgical intervention, such as duct ligation, denervation of the parasympathetic nerve supply, and gland extirpation, risk additional dysfunction and complications.3,6,7 Balloon sialodochoplasty under fluoroscopic control has been used to enlarge the ductal stricture with some success.8,9 Currently, with the refinement of

*Instructor, Department of Oral and Maxillofacial Surgery; and

Address correspondence and reprint requests to Dr Pace: Depart-

AO/Synthes Fellow, Massachusetts General Hospital, Harvard

ment of Oral and Maxillofacial Surgery, Massachusetts General Hos-

School of Dental Medicine, Boston, MA.

pital, Harvard School of Dental Medicine, Warren Suite 1201, 55

yProfessor,

Division

of

Oral

and

Maxillofacial

Surgery,

Fruit St, Boston, MA 02114; e-mail: [email protected]

Department of Dentistry, Hanyang University College of Medicine, Seoul, South Korea.

Received July 30 2014 Accepted March 2 2015

zAttending Oral and Maxillofacial Surgeon, University Hospital of

Ó 2015 American Association of Oral and Maxillofacial Surgeons

Heraklio, Crete, Greece.

0278-2391/15/00261-X

xAssociate Professor, Harvard School of Dental Medicine; and

http://dx.doi.org/10.1016/j.joms.2015.03.012

Director, Residency Training Program, Department of Oral and Maxillofacial Surgery, Massachusetts General Hospital, Boston, MA.

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SIALADENITIS TREATED BY SIALENDOSCOPY

Table 1. DEMOGRAPHIC DATA

Variable

Total (n = 51)

Age (yr) Median Range Gender Male Female Gland Parotid Right Left Bilateral Submandibular Right Left Bilateral Imaging study CT MRI Preoperative diagnosis Sj€ ogren syndrome Radioiodine treatment Navigable ducts

48 10-78 17 34 35 17 15 3 16 8 7 1 43 16 4 3 43/51

Abbreviations: CT, computed tomography; MRI, magnetic resonance imaging. Pace et al. Sialadenitis Treated by Sialendoscopy. J Oral Maxillofac Surg 2015.

endoscopic instruments and technique in the medical field, sialendoscopy has become an effective, minimally invasive procedure for the diagnosis and treatment of salivary gland obstruction.10-12 The aim of the present retrospective study was to evaluate the results of interventional sialendoscopy for 51 patients with sialadenitis without sialolithiasis.

Patients and Methods We performed a retrospective study of 51 patients with sialadenitis but without salivary stones, who Table 2. FINDINGS AND RESULTS

Total Successful Navigations (n = 43)

Finding Obstruction observed and treated Mucous Stricture Both mucous and stricture Patients symptom free at 1 wk Patients symptom free at 6 mo

27 16 13 37 (86) 36 (84)

Data in parentheses are percentages. Pace et al. Sialadenitis Treated by Sialendoscopy. J Oral Maxillofac Surg 2015.

had been treated with sialendoscopy from 2002 to 2013. The Massachusetts General Hospital institutional review board approved the study. The study included those patients who had met the following inclusion criteria: recurrent episodes of swelling or pain in a major salivary gland associated with obstructive duct condition that was refractory to conservative therapy; intraoperative findings identifying an obstructive etiology (eg, stricture, mucous-like material, and duct wall inflammation); and obstructive sialadenitis associated with Sj€ ogren syndrome or radioiodine treatment. Excluded from the study were patients with a sialolith detected on imaging studies or during sialendoscopy. Additionally, those patients who did not continue the follow-up evaluations for at least 6 months postoperatively were excluded. The data collected from the patients’ medical records included age (range 10 to 78 years), gender (34 women and 17 men), involved gland (35 parotid, 16 submandibular), medical history, clinical symptom, procedure date, endoscopic findings, endoscopic procedure, postoperative symptoms, and complications. Favorable outcomes were assessed as resolution of the obstructive etiology (ie, stricture or mucous-like material) during the endoscopic procedure and clinically symptom free (ie, the absence of recurrent swelling and pain) at 1 week to 6 months postoperatively. Follow-up examinations were performed immediately postoperatively and at 1 week and 1, 3, and 6 months postoperatively. The sialendoscopic procedure was performed with the patient under general anesthesia as an outpatient. The duct orifice of the affected gland was dilated with irrigation containing 2% lidocaine without epinephrine to facilitate insertion of a 14-guage angiocatheter. The polyethylene angiocatheter was secured in the duct with a suture passed through the mucosa and the tube. The 1.3-mm semirigid sialendoscope (Karl Storz, Tuttlingen, Germany) was then introduced through this angiocatheter and advanced through the ductal system with the aid of normal saline irrigation under light pressure. Copious irrigation facilitated navigation through the strictures and obstructions and helped flush away any mucous-like material. The sialendoscope was advanced until it was no longer possible to gently navigate further into the duct. This is a physical limitation of a semirigid endoscope. After copious irrigation of the ductal system, intraductal prednisolone was injected. The angiocatheter ‘‘stent’’ was maintained for 1 to 2 weeks after the procedure to prevent postoperative repeat stricture or adhesion formation. The patients were prescribed a short course of antibiotics and instructed to massage the affected gland frequently, stay well hydrated, and use sugar-free lemon drop candy for salivary stimulation.

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Results From 2002 to 2013, a total of 189 patients underwent an interventional sialendoscopic procedure for obstructive salivary gland disease. Of the 189 patients, 51 had sialadenitis without sialolithiasis. The present study included 34 women and 17 men with an age range of 10 to 78 years (median 48; average 43). The involved glands were 16 submandibular glands (8 right, 7 left, and 1 bilateral) and 35 parotid glands (17 right, 15 left, and 3 bilateral). Four patients had Sj€ ogren syndrome, and three had undergone radioiodine treatment for management of thyroid cancer. In 4 of the 51 patients, the etiology of the obstruction could not be identified during endoscopy. Their ducts were found to be patent to the limits of the endoscope advancement. Interventional endoscopic navigation was accomplished in 43 of 51 patients (84%). In 8 patients, including 3 of the 4 patients with Sj€ ogren syndrome, the duct could not be dilated because of scar tissue strictures. In 43 patients, we were able to dilate the ductal stricture and flush out the mucous-like material. In 27 of those 43 patients (63%), mucous-like material was found and copiously lavaged with normal saline. In 16 of the 43 patients (37%), a stricture (duct epithelial enlargement and fibrous scar tissue in the duct) was found and dilated using hydrostatic pressure. This was followed by gentle manipulation with a basket and grasper. In 13 of the 43 patients (30%), both a ductal stricture and mucous-like material were found concurrently. In these patients, the ductal dilatation and irrigation were performed at the same time. After the endoscopic procedure, 37 of the 43 patients (86%) were asymptomatic from the obstructive gland process at their first follow-up appointment. In 6 patients (14%), who had undergone a successful sialendoscopic procedure, the symptoms had not resolved. One of these patients had a history of radioiodine treatment and 1 had Sj€ ogren syndrome. Both patients declined additional intervention. Of the 37 patients who were asymptomatic at the 1-week follow-up appointment, only 1 developed recurrent symptoms. This patient refused additional treatment. Overall, 36 of 43 patients (84%) were asymptomatic after their procedure.

Discussion In our study of 51 patients with obstructive salivary gland disease, we evaluated the effectiveness of interventional sialendoscopy in resolving symptoms. We have demonstrated that most patients (84%) who underwent a successful procedure experienced symptom resolution during the follow-up period. Obstructive salivary gland disease is manifested by swelling, tenderness, pain, and discomfort that is amplified during salivary stimulation. These symptoms will

persist for 24 hours to 1 month and can recur multiple times after initially subsiding. The symptoms are primarily caused by increased pressure in the affected duct and gland, most often because of duct blockage by a salivary stone. Autoimmune diseases (eg, Sj€ ogren syndrome) and radioiodine therapy can cause obstructive gland disease, but the mechanism of ‘‘obstructive adenitis’’ without a stone is unclear. Mandel and Witek,13 in 2001, suggested that decreased salivary production or reduced saliva flow can be considered the root etiology for ascending salivary duct infection. The reduced salivary flow cannot adequately clear the normal mucous-like production in the duct and cannot prevent the retrograde flow of oral fluid into the duct. An ascending infection of the duct can ensue, resulting in inflammation of the ductal wall and damage to the epithelial lining. The edematous ductal wall propagates additional obstruction to salivary flow and subsequent swelling from the backward pressure. The inflammation instigates multifocal irregularities within the ductal wall, promulgating stricture formation and recurring obstruction.2,13 The histopathologic study of the lavaged fluid used to treat parotid sialadenitis has demonstrated hyperemia of the ductal wall and a ductal system filled with a fiber-like substance. This substance is composed of desquamative epithelial cells and neutrophils, lymphocytes, and acidophils, indicating inflammatory and degenerative changes of the ductal wall.14 In our study, we also found mucous-like material and strictures in the ductal lumen of most patients. The precise etiology of obstructive sialadenitis without a stone remains unclear. Salivary duct disorder has traditionally been diagnosed using conventional or digital sialography and ultrasonography. However, sialography is an invasive method using contrast material and provides limited information about the disease status.16 Ultrasonography is considered a noninvasive imaging technique for the study of salivary gland disease. However, the sensitivity and specificity of the results have varied, depending on the examiner17 and provide limited information about the disease process.12 Recently, magnetic resonance sialography has been used for the noninvasive diagnosis of salivary gland disease.17,18 This diagnostic technique does not, however, provide valuable direct visual imaging of the ductal lumen and saliva consistency. When evaluating obstructive salivary gland disorders, inflammation of the ductal wall and the existence of mucous-like material and strictures are important factors for diagnosis and treatment. Current sialendoscopy allows for the direct visualization of the ductal lumen and the consistency of the salvia while concurrently treating the condition. The conventional, conservative treatments of obstructive gland disease include systemic antibiotics and irrigation with normal saline containing

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SIALADENITIS TREATED BY SIALENDOSCOPY

antibiotics.3,19,20 However, these protocols have had limited success in alleviating symptoms, and recurrence of the chief complaint is likely. Obstructive gland conditions that are refractory to a conservative approach could be indicated for surgical treatment. The surgical management of salivary gland disease includes ligation of the duct and gland extirpation. Although surgical protocols have revealed high success rates for resolving symptoms, the potential complications are many and include facial nerve damage, unsatisfactory esthetics, and xerostomia due to major salivary gland excision.2,6 Intraoral balloon sialoplasty has also been performed to treat the obstructive symptoms associated with adenitis. Although this procedure has resulted in the relief of symptoms in 50 to 80% of patients, sialoplasty cannot provide direct visualization of the ductal lumen and is performed under fluoroscopic guidance.9,21,22 Interventional sialendoscopy enables the diagnosis and treatment of salivary gland ductal disorders concurrently. The development of endoscopic equipment and surgical technique provides direct visualization and allows interventional manipulation, such as ductal irrigation and dilatation. The sialendoscopy approach is very effective and minimally invasive treatment of obstructive gland disorders; however, additional advancements in the technique and the development of high-quality imaging with smaller diameter equipment is required to improve the rate of successful symptom relief. In conclusion, minimally invasive techniques are considered the treatment of choice for obstructive salivary gland disorders without stones. The interventional sialendoscopic approach provides effective diagnosis and management of ductal obstruction. To improve the treatment results, more research is needed to understand the etiology of obstructive sialadenitis and to continue the refinement of endoscopic equipment.

Uncited Reference and Tables 15, Tables 1 and 2.

References 1. Mandel ID, Baurmash H: Sialochemistry in chronic recurrent parotitis: Electrolytes and glucose. J Oral Pathol 9:92, 1980

2. Nahlieli O, Bar T, Shacham R, et al: Management of chronic recurrent parotitis: Current therapy. J Oral Maxillofac Surg 62:1150, 2004 3. Baurmash HD: Chronic recurrent parotitis: A closer look at its origin, diagnosis, and management. J Oral Maxillofac Surg 62: 1010, 2004 4. Bowling DM, Ferry G, Rauch SD, Goodman ML: Intraductal tetracycline therapy for the treatment of chronic recurrent parotitis. Ear Nose Throat J 73:262, 1994 5. Gear KJ, Hay KD, Stumpel J: Treatment of parotid ductal stenosis and concomitant resolution of autonomic symptomatology. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 94: 632, 2002 6. Nichols RD: Surgical treatment of chronic suppurative parotitis: A critical review. Laryngoscope 87:2066, 1977 7. Mandel L, Kaynar A: Surgical bypass of submandibular duct stricture. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 88:532, 1999 8. Rontal M, Rontal E: The use of sialodochoplasty in the treatment of benign inflammatory obstructive submandibular gland disease. Laryngoscope 97:1417, 1987 9. Salerno S, Lo Casto A, Comparetto A, et al: Sialodochoplasty in the treatment of salivary-duct stricture in chronic sialoadenitis: Technique and results. Radiol Med 112:138, 2007 10. Konigsberger R, Feyh J, Goetz A, Kastenbauer E: Endoscopically-controlled electrohydraulic intracorporeal shock wave lithotripsy (EISL) of salivary stones. J Otolaryngol 22:12, 1993 11. Nahlieli O, Shacham R, Yoffe B, Eliav E: Diagnosis and treatment of strictures and kinks in salivary gland ducts. J Oral Maxillofac Surg 59:484, 2001 12. Yu C, Zheng L, Yang C, Shen N: Causes of chronic obstructive parotitis and management by sialendoscopy. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 105:365, 2008 13. Mandel L, Witek EL: Chronic parotitis: Diagnosis and treatment. J Am Dent Assoc 132:1707, 2001 14. Qi S, Liu X, Wang S: Sialendoscopic and irrigation findings in chronic obstructive parotitis. Laryngoscope 115:541, 2005 15. Yuasa K, Nakhyama E, Ban S, et al: Submandibular gland duct endoscopy: Diagnostic value for salivary duct disorders in comparison to conventional radiography, sialography, and ultrasonography. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 84:578, 1997 16. Buckenham TM, George CD, McVicar D, et al: Digital sialography: Imaging and intervention. Br J Radiol 67:524, 1994 17. Capaccio P, Cuccarini V, Ottaviani F, et al: Comparative ultrasonographic, magnetic resonance sialographic, and videoendoscopic assessment of salivary duct disorders. Ann Otol Rhinol Laryngol 117:245, 2008 18. Fischbach R, Kugel H, Ernst S, et al: MR sialography: Initial experience using a T2-weighted fast SE sequence. J Comput Assist Tomogr 21:826, 1997 19. Motamed M, Laugharne D, Bradley PJ: Management of chronic parotitis: A review. J Laryngol Otol 117:521, 2003 20. Antoniades D, Harrison JD, Epivatianos A, Papanayotou P: Treatment of chronic sialadenitis by intraductal penicillin or saline. J Oral Maxillofac Surg 62:431, 2004 21. Roberts DN, Juman S, Hall JR, Jonathan DA: Parotid duct stenosis: Interventional radiology to the rescue. Ann R Coll Surg Engl 77:444, 1995 22. Brown AL, Shepherd D, Buckenham TM: Per oral balloon sialoplasty: Results in the treatment of salivary duct stenosis. Cardiovasc Intervent Radiol 20:337, 1997

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Sialadenitis Without Sialolithiasis Treated by Sialendoscopy.

The aim of the present study was to evaluate the effectiveness of interventional sialendoscopy in the management of non-stone obstructive sialadenitis...
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