The Laryngoscope C 2014 The American Laryngological, V
Rhinological and Otological Society, Inc.
Pediatric Sialendoscopy: Initial Experience in a Pediatric Otolaryngology Group Practice Ryan Semensohn, BS; Zorik Spektor, MD; David J. Kay, MD, MPH; Alfredo S. Archilla, MD; David L. Mandell, MD Objectives/Hypothesis: To describe the initial results of sialendoscopy as a diagnostic and therapeutic tool in pediatric inflammatory salivary gland disease. Study Design: Retrospective review of patient medical records from a private practice consisting of three fellowshiptrained pediatric otolaryngologists. Methods: Consecutive pediatric patients with either recurrent or chronic sialadenitis underwent diagnostic and therapeutic sialendoscopy as an alternative to continued antibiotic therapy or surgical gland excision. Data collected included age, gender, indications for surgery, intraoperative findings, complications, recurrences, follow-up intervals, and need for additional procedures. Results: Twelve pediatric patients underwent sialendsocopy (9 cases of juvenile recurrent parotitis, 3 cases of chronic submandibular sialadenitis. Intraoperative findings included ductal stricture (n 5 8), thick intraductal mucus (n 5 6), and ductal calculus (n 5 1). The only postoperative complication was one case of a submandibular gland, which remained enlarged for 1 month postoperatively before resolving. Average follow-up was 16.5 months (range: 1–49 months), during which time two patients had recurrence (17%). One patient had repeated recurrences that only resolved after salvage parotidectomy. Another patient had one isolated recurrence that resolved with antibiotics. To date, 92% of patients have not required any further surgical intervention after a sialendoscopy procedure. Conclusions: Sialendoscopy was successfully implemented as a safe and effective technique for management of recurrent and chronic parotid and submandibular sialadenitis in a pediatric otolaryngology practice. Key Words: Sialendoscopy, pediatric, juvenile recurrent parotitis, sialadenitis. Level of Evidence: 4. Laryngoscope, 125:480–484, 2015
INTRODUCTION Inflammation is a common presentation of pediatric major salivary-gland disorders. Juvenile recurrent parotitis is defined as recurrent swelling and inflammation of either one or both parotid glands during childhood, occurring at least twice per year. This disorder is felt to be multifactorial in nature, with contributions from allergies, immune deficiency, and genetic factors that include congenital ductal abnormalities.1,2 Chronic and recurrent inflammation involve the submandibular gland less commonly in children than the parotid gland, but when the submandibular gland is involved, it is more likely than the parotid gland to have sialolithiasis as the underlying etiology. From the NOVA Southeastern University College of Osteopathic Medicine, Department of Surgery, Division of Otolaryngology (R.S., D.J.K., D.L.M.), Ft. Lauderdale-Davie; and the Center for Pediatric ENT (Z.S., D.J.K., A.A., D.L.M.), Boynton Beach, Florida, U.S.A. Editor’s Note: This Manuscript was accepted for publication July 15, 2014. Presented at the Triological Society Meeting, Las Vegas, Nevada, U.S.A., May 14–15, 2014. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to David L. Mandell, MD, Center for Pediatric ENT, 10301 Hagen Ranch Road, Suite B-900, Boynton Beach, FL 33437. E-mail: [email protected] DOI: 10.1002/lary.24868
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During an acute episode of acute pediatric sialadenitis, the mainstay of treatment is with oral antibiotics and analgesics. Concurrent conservative measures can also be added, such as hydration, sialagogues, warm compresses, and parotid massage.1,2 In those patients for whom the acute episodes are recurrent, it is challenging to find a minimally invasive way to prevent these acute recurrences. A variety of management options have been attempted, including prophylactic antibiotics, transoral ductal ligation, and tympanic neurectomy, but these techniques have generally been felt to be ineffective or untenable.1,2 Open excision of the offending gland is considered a definitive treatment option, which is not widely embraced or performed for this indication due to procedural morbidity, particularly with respect to facial paralysis after pediatric superficial parotidectomy.1 In recent years, the technique of sialendoscopy has emerged as a minimally invasive alternative to gland excision in cases of recurrent sialadenitis and sialolithiasis. The procedure utilizes a small endoscope to transorally navigate the major salivary gland ductal system for both diagnostic and therapeutic purposes, which allows for ductal dilatation, removal of stones, and irrigation with medications that include corticosteroids.3 Initially described for use in adults, the development of smaller instrumentation has allowed a broadening of the Semensohn et al.: Pediatric Sialendsocopy
Fig. 1. Sialendoscopic view from within Stensen’s duct, showing the first level of normal ductal branching pattern. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
application to pediatric patients, with several recent series describing their experience with this technique in children who have disorders of the major salivary glands.1–7 The purpose of this study is to describe the experience and results of a pediatric otolaryngology practice that has incorporated the emerging technique of pediatric sialendsocopy as a diagnostic and therapeutic management option for cases of recurrent and chronic pediatric sialadenitis.
All procedures were performed under general anesthesia with orotracheal intubation. Patients were placed in a supine position with an appropriate-sized bite block placed on the contralateral side of the oral cavity from the side of the gland undergoing sialendoscopy. The ducts (either Stensen’s or Wharton’s, depending on the involved gland) were serially dilated using a set of six ductal dilators that are included in the sialendoscope kit (Karl Storz Endoscopy-America, Inc., El Segundo, CA). Next, a 1.1-mm outer-diameter, 10-cm length, semirigid sialendoscope attached to a camera and a television screen was introduced into the duct via the dilated orifice. The sialendoscope has a 0.25-mm inner-diameter irrigating channel with a 0.45-mm inner-diameter working channel through which instrumentation can be passed if needed. As the scope is introduced into the duct, the duct is gently dilated open with hydrostatic pressure via sterile normal saline irrigation. The scope is then advanced to the first branching point of the main duct, and if possible each branch is examined as far as the scope can comfortably pass (Fig. 1). If needed, there is a pneumatic balloon available for ductal stenoses and a hand drill and basket for sialoliths. With our initial patients, saline irrigation was administered by an assistant via a 10-mL syringe attached to the sialendoscope side port. The syringe often had to be replaced because all the saline was injected. Later, we began irrigating saline through the sialendoscope using IV tubing attached to an Endo-Scrub device (Medtronic, Inc., Minneapolis, MN) on a low setting (500 rpm). Use of this technique allowed the operator to hold the scope in one hand, retract the patient’s buccal mucosa for countertension against the scope with the other hand, and irrigate saline with a foot pedal, thus making this a procedure that can be performed by one operator. Stenoses were diagnosed based on a white color without the natural proliferation of blood vessels seen in normal ducts,3 along with a narrowing of the duct that led to difficulty passing and advancing the scope (Fig. 2). Strictures and stenosis were dilated via hydrostatic pressure. In our patients, medications were not injected via the duct, although all patients received an intravenous dose of dexamethasone (0.5 mg/kg, maximum of 12 mg).
MATERIALS AND METHODS A retrospective study from January 2010 to February 2014 was conducted at the Center for Pediatric ENT (Boynton Beach, FL), a private practice consisting of three fellowshiptrained pediatric otolaryngologists. This study was found to be exempt under 45CFR56.101(b)4 from the NOVA Southeastern University College of Osteopathic Medicine, Fort Lauderdale, Florida. An electronic medical records search was performed for all patients in the practice who underwent sialendoscopy (using the CPT code 42505). These patients were all referred to the practice with complaints of either recurrent acute major salivary gland swelling that had occurred at least two times per year, or chronic major salivary gland swelling that had lasted at least 3 months. Information collected included patient age at the time of procedure; gender; and duration of salivary gland inflammatory disease prior to the procedure—such as number of acute inflammatory episodes, intraoperative findings, surgical complications, length of follow-up, and any recurrences—as well as how recurrences were managed. Most patients received some type of imaging prior to surgery, either CT or ultrasound, although imaging was not considered a prerequisite for a patient with the appropriate history to be considered for sialendoscopy. Patients were offered sialendoscopy after a careful discussion of the risks, benefits, and alternatives to treatment. After informed consent was obtained, the patients were then scheduled for the procedure.
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Fig. 2. Sialendoscopic view of stenosis of Stensen’s duct, with white avascular appearance of ductal mucosa (5-year-old girl with juvenile recurrent parotitis). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
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Fig. 3. Sialendoscopic view of thick mucoid secretions from within Stensen’s duct (11-year-old girl with juvenile recurrent parotitis). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
RESULTS Twelve patients underwent sialendoscopy during the study period. The mean age at the time of the procedure was 9 years old (range: 3 to 18 years old). There were five males and seven females. The procedure was performed for juvenile recurrent parotitis in nine of the 12 patients (male to female ratio of 4:5; 4 bilateral cases). The procedure was performed for submandibular sialadenitis in three patients, including one case of sialolithiasis (male to female ratio of 1:2, 1 bilateral case). The mean duration of signs and symptoms of sialadenitis prior to sialendoscopy was 29 months (range: 3 months–8 years); and for those with juvenile recurrent parotitis, the mean number of acute inflammatory episodes experienced prior to sialendoscopy was 11 (range: 3–28). Due to some cases undergoing the procedure bilaterally, a total of 17 glands underwent sialendoscopy among the 12 patients. The total amount of sterile saline irrigation used to dilate the salivary ductal system was typically between 20 to 30 mL per gland. More saline was actually utilized during the procedure, but some of it would be “wasted” when the scope was primed and when on occasion the sialendoscope would slip out of the duct, especially when the scope was first being introduced into the duct at the onset of the procedure. It became more difficult to precisely measure the amount of saline administered after a switch from hand-held 10mL syringes to the Endo-Scrub (Medtronic) powered irrigator. Imaging studies had been obtained in 11 of the 12 patients, including nine computed tomography (CT) scans with intravenous contrast and two ultrasounds. Most of these studies had been ordered by the referring practitioners. None of the CT scans required general anesthesia to be performed. Computed tomography Laryngoscope 125: February 2015
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typically either showed normal-appearing major salivary glands, if taken between acute inflammatory episodes, or homogenous salivary gland enlargement, if taken during an acute infection. Sialolithiasis was found on CT imaging in one patient (the patient presented with recurrent submandibular sialadenitis), and two unilateral submandibular sialoliths were demonstrated on the scan. One ultrasound study in a patient with juvenile recurrent parotitis showed dilated parotid ducts bilaterally, and the other ultrasound obtained showed diffuse enlargement of the affected parotid gland. Among the 17 major salivary gland ducts that were explored endoscopically in our patients, intraoperative findings included the following: ductal stenosis (n 5 13) (Fig. 2), thick intraductal mucus and debris (n 5 10) (Fig. 3), and sialolithiasis (n 5 1) (Fig. 4). There were no intraoperative complications. Due to the intraductal saline irrigation, patients typically awoke from anesthesia with noticeable swelling of the affected gland, followed by spontaneous resolution within several hours. The only postoperative complication noted in our patients was one case of persistent submandibular gland swelling, which persisted for 1 month after submandibular sialendoscopy before spontaneously resolving. The mean duration of postoperative follow-up was 16.5 months (range: 1–49 months). Of the 12 patients in the study, there were two for whom recurrences were noted (17%). In one patient who underwent unilateral parotid sialendoscopy for juvenile recurrent parotitis, there was an acute inflammatory recurrence in the same gland 15 months after the procedure. This episode resolved with a course of oral antibiotics, with no further recurrences to date 9 months after the recurrence. In another patient, who had a history of 8 years of recurrent unilateral parotitis that included a history of undergoing incision and drainage for a parotid abscess, two
Fig. 4. Sialendoscopic view from within Wharton’s duct, showing a sialolith within a ductal branch. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
Semensohn et al.: Pediatric Sialendsocopy
inflammatory recurrences occurred within the same gland within the first 6 months after sialendoscopy. We were suspicious that there may have been an underlying first branchial cleft cyst but could not demonstrate a discrete lesion with imaging. Regardless, the patient subsequently underwent superficial parotidectomy, followed by no further recurrences on the affected side and one isolated episode of the contralateral parotid that was treated successfully with oral antibiotics (follow-up over 2 years subsequent to parotidectomy). Overall, 83% of patients had no postoperative recurrences, and 92% of patients did well enough after sialendoscopy to be considered candidates for any further surgical intervention.
DISCUSSION The technique of sialendoscopy was first described by Katz in 19908 and subsequently popularized in the 1990s, mainly in adult patients.9,10 In adults, a common indication for sialendoscopy is sialolithiasis, whereas in children the most common indication is juvenile recurrent parotitis, which usually occurs in the absence of sialolithiasis.5 There does not appear to be an obvious consensus on the exact number of episodes of acute parotitis needed to make the diagnosis of juvenile recurrent parotitis, but recent series generally have applied this diagnosis if episodes occur between two times per year5 to two times in 6 months.4 Severity is determined by the frequency of infections, not necessarily by the features of each individual infection.5 Associated signs and symptoms of JRP include pain, erythema of the skin overlying the gland, and fever. The peak incidence of JRP is between 3 to 6 years of age, and there is an approximately 2:1 male predominance reported in the literature.1,3 A male predominance was not seen in our study, possibly due to the small number of patients. The etiology of JRP is unknown. It has been theorized that the cause is multifactorial, with likely contributions from allergies, immune deficiency, and genetic factors including congenital ductal abnormalities.1,2 In the literature, 33% of patients with juvenile recurrent parotitis who undergo sialendoscopy have bilateral disease1; the rate in our study was 44%. Awareness of sialendoscopy appears to be increasing for both diagnosis and treatment of inflammatory disorders of the major salivary glands in children.1 The procedure allows direct visualization of the duct for diagnostic purposes and offers treatment by breaking the cycle of inflammation via dilatation of ductal stenoses (with hydrostatic pressure, balloons, or the scope itself), removal of ductal debris with irrigation, removal of stones (if present), and instillation of medications.2 In our patients, we found that ductal dilatation with hydrostatic pressure from saline irrigation was sufficient in most cases to achieve the desired clinical result. We did not use balloons in our study or inject any medication (steroids or antibiotics) intraductally, and only one patient required extra instrumentation (a micro hand drill and basket for removal of a sialolith). We did modify our technique to use a powered Endo-Scrub machine Laryngoscope 125: February 2015
(Medtronic) to deliver the saline irrigation via intravenous tubing connected to the sialendoscope, thus allowing the procedure to be performed without an assistant injecting saline via a syringe. When we initially tried injecting saline with an assistant surgeon, we found that the assistant required a subjectively large amount of manual pressure to inject the saline, and the syringe had to be repeatedly replaced when the saline ran out, causing a temporary loss of endoscopic visualization. It is known that one risk of pediatric sialendoscopy, when performed bilaterally, is deep parotid lobe enlargement with temporary airway obstruction due to excessive ductal irrigation, and we are certainly cognizant of this.3 However, thus far this particular complication was not observed in the patients, even with use of the powered saline pump that was set at a low pressure. In the literature, intraoperative sialendoscopic findings include fibrinous, purulent, or mucoid debris within the ducts—as well as ductal stenosis, which is typified by a white ductal layer without the natural proliferation of blood vessels and is often found in juvenile recurrent parotitis.2,3,5 Similar intraoperative sialendoscopic findings were noted in our study as well. When sialoliths are found, they are usually within the submandibular gland3,5; and when ductal stenoses are found, they are mostly in the parotid duct.3 One of the disadvantages of pediatric sialendoscopy is that general anesthesia is typically needed in children in order to perform the procedure. However, one recent report described pediatric sialendoscopy being successfully performed under local anesthesia in seven of nine children for whom this approach was attempted.4 Another disadvantage of pediatric sialendoscopy is that during the first few hours after the procedure, swelling of the gland that underwent endoscopy is common, although it is usually asymptomatic and resolves within a few hours.1 The reported complication rate in several recent case series has ranged from 0% to 8% to 17%.2–5,7 When complications have been reported, they have been able to be resolved. Complications have included temporary postoperative airway obstruction (presumably from deep lobe parotid swelling in bilateral parotid sialendoscopy with copious ductal irrigation),3 pain and swelling 1 week postop requiring oral antibiotics,5 and Stensen’s duct perforation (2 cases in the recent literature, one associated with use of a laser3 and the other with use of a balloon,5 with both cases repaired surgically). The mean age of patients undergoing the procedure in the literature has been 6 years to 9 years,1,5 which is similar to our mean patient age (9 years old). The mean number of episodes before treatment has ranged from 4.7 to 6,1,5 whereas in our study, patients had a mean number of 11 infections prior to sialendoscopy, perhaps reflecting the lack of access to, and awareness of, sialendoscopy in our practice area . The most recent pediatric sialendoscopy case series in the English-language literature have included 6, 9, 9, 18, 38, and 70 patients.1–5,7 In studies that have looked at all cases of pediatric sialendoscopy (parotid and submandibular), the parotid gland has been the primary target in 61%, 72%, and 83% of cases.2,3,5 We found that Semensohn et al.: Pediatric Sialendsocopy
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the parotid gland was involved in 75% of our sialendoscopy patients. The most common sialendoscopic finding in cases of submandibular sialadenitis is sialolithiasis7 and ductal stenosis in cases of juvenile recurrent parotitis.1 Regarding pediatric patients, the success rate at either eliminating or reducing the severity and frequency of recurrences has been reported in recent series to range between 83% to 100%.1–3,5,7 Our results (83% success if success is defined as no recurrences; 92% success if success defined as no more than 1 recurrence), are similar to the results reported in the literature. However, it should be cautioned that follow-up is still relatively limited in our own study as well as in the literature, and as more time passes there will be more understanding regarding the long-term benefit (or lack thereof) of the procedure. In addition, some evidence exists that repeat sialendoscopy can be effective in eliminating recurrences if the gland fails to respond to an initial sialendoscopy procedure.4,5 In the literature, the ductal system has been irrigated or rinsed with medications, such as 50% xylocaine (2%) and 50% normal saline (0.9% NaCl) with 120-mg prednisolone,3 half xylocaine 2% and half NaCl 0.9%,4 or 100 mg of hydrocortisone via the sialendoscope into the gland at the completion of the procedure.1 Our group did not irrigate with any medication, in part because there is no evidence-based research that this approach is effective. In the literature, sialoliths have been broken up with a laser (2.1-um continuous YAG-thulium laser), and severe stenoses ( 3 mm) have also been opened with a laser (2.1-um continuous YAG-thulium laser), although laser use may increase the risk of ductal perforation.3 Small stones and stones that have been broken up with a laser can be removed with a wire basket. A 300-um microdrill can also be used for challenging strictures and sialoliths.1 In most series, preoperative imaging is performed (usually via ultrasonography).1,4 One drawback to sonography is that sialoliths smaller than 2 mm, which can be seen with sialendoscopy, are difficult to visualize on sonography.3 Classic sonographic findings in patients
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with parotid sialadenitis include multiple, small round/ oval hypoechoic areas throughout the gland that represent dilated ducts.1 However, when ductal stenosis is found on sialendoscopy, the presumed sonographic corresponding finding of ductal dilatation is not always seen on sonography.3 In our study, we felt that the most relevant contribution of preoperative sonography was the benefit of ruling out other potential etiologies (such as masses or tumors).
CONCLUSION Pediatric sialendoscopy is emerging as a safe, minimally invasive procedure that the authors have found to have a fast learning curve and a satisfactory rate of success at resolving cases of recurrent pediatric sialadenitis, particularly concerning the parotid gland. Because of these benefits, sialendoscopy has now become fully integrated as the preferred treatment modality in our pediatric otolaryngology practice for children with recurrent sialadenitis, prior to consideration of more invasive procedures such as parotidectomy.
BIBLIOGRAPHY 1. Shacham R, Bar Droma E, London D, Bar T, Nahlieli O. Long-term experience with endoscopic diagnosis and treatment of juvenile recurrent Parotitis. J Oral Maxillofac Surg 2009;67:162–167. 2. Jabbour N, Tibesar R, Lander T, Sidman J. Sialendoscopy in children. Int J Pediatr Otolaryngology 2010;74:347–350. 3. Martins-Cavalho C, Plouin-Gaudon I, Quenin S, et al. Pediatric sialendoscopy. A 5-year experience at a single institution. Arch Otolaryngol Head Neck Surg 2010;136:33–36. 4. Konstantinidis I, Chatziavramidis E, Tsakiropoulou E, Malliari H, Constantinidis J. Pediatric sialendoscopy under local anesthesia: limitations and potentials. Int J Pediatr Otorhinolaryngol 2011;75:245–249. 5. Hackett AM, Baranano CF, Reed M, Duvvuri U, Smith RJ, Mehta D. Sialoendoscopy for the treatment of pediatric salivary gland disorders. Arch Otolaryngol Head Neck Surg 2012;138:912–915. 6. Nguyen AM, Francis CL, Larsen CG. Salivary endoscopy in a pediatric patient with HLA-B27 seropositivity and recurrent submandibular sialadenitis. Int J Pediatr Otorhinolaryngol 2013;77:1045–1047. 7. Mikolajczak S, Meyer MF, Beutner D, Luers JC. Treatment of chronic recurrent juvenile parotitis using sialendoscopy. Acta Otolaryngol 2014; 134:531–535. 8. Katz P. New method of examination of the salivary glands: the fiberscope. Inf Dent 1990;72:785–786. 9. Nahlieli O, Baruchin AM. Sialendoscopy: three years’ experience as a diagnostic and treatment modality. J Oral Maxillofac Surg 1997;55:912–918. 10. Marchal F, Dulguerov P, Lehmann W. Interventional sialendoscopy. N Engl J Med 1999;341:1242–1243.
Semensohn et al.: Pediatric Sialendsocopy
Rhinological and Otological Society, Inc.
Pediatric Sialendoscopy: Initial Experience in a Pediatric Otolaryngology Group Practice Ryan Semensohn, BS; Zorik Spektor, MD; David J. Kay, MD, MPH; Alfredo S. Archilla, MD; David L. Mandell, MD Objectives/Hypothesis: To describe the initial results of sialendoscopy as a diagnostic and therapeutic tool in pediatric inflammatory salivary gland disease. Study Design: Retrospective review of patient medical records from a private practice consisting of three fellowshiptrained pediatric otolaryngologists. Methods: Consecutive pediatric patients with either recurrent or chronic sialadenitis underwent diagnostic and therapeutic sialendoscopy as an alternative to continued antibiotic therapy or surgical gland excision. Data collected included age, gender, indications for surgery, intraoperative findings, complications, recurrences, follow-up intervals, and need for additional procedures. Results: Twelve pediatric patients underwent sialendsocopy (9 cases of juvenile recurrent parotitis, 3 cases of chronic submandibular sialadenitis. Intraoperative findings included ductal stricture (n 5 8), thick intraductal mucus (n 5 6), and ductal calculus (n 5 1). The only postoperative complication was one case of a submandibular gland, which remained enlarged for 1 month postoperatively before resolving. Average follow-up was 16.5 months (range: 1–49 months), during which time two patients had recurrence (17%). One patient had repeated recurrences that only resolved after salvage parotidectomy. Another patient had one isolated recurrence that resolved with antibiotics. To date, 92% of patients have not required any further surgical intervention after a sialendoscopy procedure. Conclusions: Sialendoscopy was successfully implemented as a safe and effective technique for management of recurrent and chronic parotid and submandibular sialadenitis in a pediatric otolaryngology practice. Key Words: Sialendoscopy, pediatric, juvenile recurrent parotitis, sialadenitis. Level of Evidence: 4. Laryngoscope, 125:480–484, 2015
INTRODUCTION Inflammation is a common presentation of pediatric major salivary-gland disorders. Juvenile recurrent parotitis is defined as recurrent swelling and inflammation of either one or both parotid glands during childhood, occurring at least twice per year. This disorder is felt to be multifactorial in nature, with contributions from allergies, immune deficiency, and genetic factors that include congenital ductal abnormalities.1,2 Chronic and recurrent inflammation involve the submandibular gland less commonly in children than the parotid gland, but when the submandibular gland is involved, it is more likely than the parotid gland to have sialolithiasis as the underlying etiology. From the NOVA Southeastern University College of Osteopathic Medicine, Department of Surgery, Division of Otolaryngology (R.S., D.J.K., D.L.M.), Ft. Lauderdale-Davie; and the Center for Pediatric ENT (Z.S., D.J.K., A.A., D.L.M.), Boynton Beach, Florida, U.S.A. Editor’s Note: This Manuscript was accepted for publication July 15, 2014. Presented at the Triological Society Meeting, Las Vegas, Nevada, U.S.A., May 14–15, 2014. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to David L. Mandell, MD, Center for Pediatric ENT, 10301 Hagen Ranch Road, Suite B-900, Boynton Beach, FL 33437. E-mail: [email protected] DOI: 10.1002/lary.24868
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During an acute episode of acute pediatric sialadenitis, the mainstay of treatment is with oral antibiotics and analgesics. Concurrent conservative measures can also be added, such as hydration, sialagogues, warm compresses, and parotid massage.1,2 In those patients for whom the acute episodes are recurrent, it is challenging to find a minimally invasive way to prevent these acute recurrences. A variety of management options have been attempted, including prophylactic antibiotics, transoral ductal ligation, and tympanic neurectomy, but these techniques have generally been felt to be ineffective or untenable.1,2 Open excision of the offending gland is considered a definitive treatment option, which is not widely embraced or performed for this indication due to procedural morbidity, particularly with respect to facial paralysis after pediatric superficial parotidectomy.1 In recent years, the technique of sialendoscopy has emerged as a minimally invasive alternative to gland excision in cases of recurrent sialadenitis and sialolithiasis. The procedure utilizes a small endoscope to transorally navigate the major salivary gland ductal system for both diagnostic and therapeutic purposes, which allows for ductal dilatation, removal of stones, and irrigation with medications that include corticosteroids.3 Initially described for use in adults, the development of smaller instrumentation has allowed a broadening of the Semensohn et al.: Pediatric Sialendsocopy
Fig. 1. Sialendoscopic view from within Stensen’s duct, showing the first level of normal ductal branching pattern. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
application to pediatric patients, with several recent series describing their experience with this technique in children who have disorders of the major salivary glands.1–7 The purpose of this study is to describe the experience and results of a pediatric otolaryngology practice that has incorporated the emerging technique of pediatric sialendsocopy as a diagnostic and therapeutic management option for cases of recurrent and chronic pediatric sialadenitis.
All procedures were performed under general anesthesia with orotracheal intubation. Patients were placed in a supine position with an appropriate-sized bite block placed on the contralateral side of the oral cavity from the side of the gland undergoing sialendoscopy. The ducts (either Stensen’s or Wharton’s, depending on the involved gland) were serially dilated using a set of six ductal dilators that are included in the sialendoscope kit (Karl Storz Endoscopy-America, Inc., El Segundo, CA). Next, a 1.1-mm outer-diameter, 10-cm length, semirigid sialendoscope attached to a camera and a television screen was introduced into the duct via the dilated orifice. The sialendoscope has a 0.25-mm inner-diameter irrigating channel with a 0.45-mm inner-diameter working channel through which instrumentation can be passed if needed. As the scope is introduced into the duct, the duct is gently dilated open with hydrostatic pressure via sterile normal saline irrigation. The scope is then advanced to the first branching point of the main duct, and if possible each branch is examined as far as the scope can comfortably pass (Fig. 1). If needed, there is a pneumatic balloon available for ductal stenoses and a hand drill and basket for sialoliths. With our initial patients, saline irrigation was administered by an assistant via a 10-mL syringe attached to the sialendoscope side port. The syringe often had to be replaced because all the saline was injected. Later, we began irrigating saline through the sialendoscope using IV tubing attached to an Endo-Scrub device (Medtronic, Inc., Minneapolis, MN) on a low setting (500 rpm). Use of this technique allowed the operator to hold the scope in one hand, retract the patient’s buccal mucosa for countertension against the scope with the other hand, and irrigate saline with a foot pedal, thus making this a procedure that can be performed by one operator. Stenoses were diagnosed based on a white color without the natural proliferation of blood vessels seen in normal ducts,3 along with a narrowing of the duct that led to difficulty passing and advancing the scope (Fig. 2). Strictures and stenosis were dilated via hydrostatic pressure. In our patients, medications were not injected via the duct, although all patients received an intravenous dose of dexamethasone (0.5 mg/kg, maximum of 12 mg).
MATERIALS AND METHODS A retrospective study from January 2010 to February 2014 was conducted at the Center for Pediatric ENT (Boynton Beach, FL), a private practice consisting of three fellowshiptrained pediatric otolaryngologists. This study was found to be exempt under 45CFR56.101(b)4 from the NOVA Southeastern University College of Osteopathic Medicine, Fort Lauderdale, Florida. An electronic medical records search was performed for all patients in the practice who underwent sialendoscopy (using the CPT code 42505). These patients were all referred to the practice with complaints of either recurrent acute major salivary gland swelling that had occurred at least two times per year, or chronic major salivary gland swelling that had lasted at least 3 months. Information collected included patient age at the time of procedure; gender; and duration of salivary gland inflammatory disease prior to the procedure—such as number of acute inflammatory episodes, intraoperative findings, surgical complications, length of follow-up, and any recurrences—as well as how recurrences were managed. Most patients received some type of imaging prior to surgery, either CT or ultrasound, although imaging was not considered a prerequisite for a patient with the appropriate history to be considered for sialendoscopy. Patients were offered sialendoscopy after a careful discussion of the risks, benefits, and alternatives to treatment. After informed consent was obtained, the patients were then scheduled for the procedure.
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Fig. 2. Sialendoscopic view of stenosis of Stensen’s duct, with white avascular appearance of ductal mucosa (5-year-old girl with juvenile recurrent parotitis). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
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Fig. 3. Sialendoscopic view of thick mucoid secretions from within Stensen’s duct (11-year-old girl with juvenile recurrent parotitis). [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
RESULTS Twelve patients underwent sialendoscopy during the study period. The mean age at the time of the procedure was 9 years old (range: 3 to 18 years old). There were five males and seven females. The procedure was performed for juvenile recurrent parotitis in nine of the 12 patients (male to female ratio of 4:5; 4 bilateral cases). The procedure was performed for submandibular sialadenitis in three patients, including one case of sialolithiasis (male to female ratio of 1:2, 1 bilateral case). The mean duration of signs and symptoms of sialadenitis prior to sialendoscopy was 29 months (range: 3 months–8 years); and for those with juvenile recurrent parotitis, the mean number of acute inflammatory episodes experienced prior to sialendoscopy was 11 (range: 3–28). Due to some cases undergoing the procedure bilaterally, a total of 17 glands underwent sialendoscopy among the 12 patients. The total amount of sterile saline irrigation used to dilate the salivary ductal system was typically between 20 to 30 mL per gland. More saline was actually utilized during the procedure, but some of it would be “wasted” when the scope was primed and when on occasion the sialendoscope would slip out of the duct, especially when the scope was first being introduced into the duct at the onset of the procedure. It became more difficult to precisely measure the amount of saline administered after a switch from hand-held 10mL syringes to the Endo-Scrub (Medtronic) powered irrigator. Imaging studies had been obtained in 11 of the 12 patients, including nine computed tomography (CT) scans with intravenous contrast and two ultrasounds. Most of these studies had been ordered by the referring practitioners. None of the CT scans required general anesthesia to be performed. Computed tomography Laryngoscope 125: February 2015
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typically either showed normal-appearing major salivary glands, if taken between acute inflammatory episodes, or homogenous salivary gland enlargement, if taken during an acute infection. Sialolithiasis was found on CT imaging in one patient (the patient presented with recurrent submandibular sialadenitis), and two unilateral submandibular sialoliths were demonstrated on the scan. One ultrasound study in a patient with juvenile recurrent parotitis showed dilated parotid ducts bilaterally, and the other ultrasound obtained showed diffuse enlargement of the affected parotid gland. Among the 17 major salivary gland ducts that were explored endoscopically in our patients, intraoperative findings included the following: ductal stenosis (n 5 13) (Fig. 2), thick intraductal mucus and debris (n 5 10) (Fig. 3), and sialolithiasis (n 5 1) (Fig. 4). There were no intraoperative complications. Due to the intraductal saline irrigation, patients typically awoke from anesthesia with noticeable swelling of the affected gland, followed by spontaneous resolution within several hours. The only postoperative complication noted in our patients was one case of persistent submandibular gland swelling, which persisted for 1 month after submandibular sialendoscopy before spontaneously resolving. The mean duration of postoperative follow-up was 16.5 months (range: 1–49 months). Of the 12 patients in the study, there were two for whom recurrences were noted (17%). In one patient who underwent unilateral parotid sialendoscopy for juvenile recurrent parotitis, there was an acute inflammatory recurrence in the same gland 15 months after the procedure. This episode resolved with a course of oral antibiotics, with no further recurrences to date 9 months after the recurrence. In another patient, who had a history of 8 years of recurrent unilateral parotitis that included a history of undergoing incision and drainage for a parotid abscess, two
Fig. 4. Sialendoscopic view from within Wharton’s duct, showing a sialolith within a ductal branch. [Color figure can be viewed in the online issue, which is available at www.laryngoscope.com.]
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inflammatory recurrences occurred within the same gland within the first 6 months after sialendoscopy. We were suspicious that there may have been an underlying first branchial cleft cyst but could not demonstrate a discrete lesion with imaging. Regardless, the patient subsequently underwent superficial parotidectomy, followed by no further recurrences on the affected side and one isolated episode of the contralateral parotid that was treated successfully with oral antibiotics (follow-up over 2 years subsequent to parotidectomy). Overall, 83% of patients had no postoperative recurrences, and 92% of patients did well enough after sialendoscopy to be considered candidates for any further surgical intervention.
DISCUSSION The technique of sialendoscopy was first described by Katz in 19908 and subsequently popularized in the 1990s, mainly in adult patients.9,10 In adults, a common indication for sialendoscopy is sialolithiasis, whereas in children the most common indication is juvenile recurrent parotitis, which usually occurs in the absence of sialolithiasis.5 There does not appear to be an obvious consensus on the exact number of episodes of acute parotitis needed to make the diagnosis of juvenile recurrent parotitis, but recent series generally have applied this diagnosis if episodes occur between two times per year5 to two times in 6 months.4 Severity is determined by the frequency of infections, not necessarily by the features of each individual infection.5 Associated signs and symptoms of JRP include pain, erythema of the skin overlying the gland, and fever. The peak incidence of JRP is between 3 to 6 years of age, and there is an approximately 2:1 male predominance reported in the literature.1,3 A male predominance was not seen in our study, possibly due to the small number of patients. The etiology of JRP is unknown. It has been theorized that the cause is multifactorial, with likely contributions from allergies, immune deficiency, and genetic factors including congenital ductal abnormalities.1,2 In the literature, 33% of patients with juvenile recurrent parotitis who undergo sialendoscopy have bilateral disease1; the rate in our study was 44%. Awareness of sialendoscopy appears to be increasing for both diagnosis and treatment of inflammatory disorders of the major salivary glands in children.1 The procedure allows direct visualization of the duct for diagnostic purposes and offers treatment by breaking the cycle of inflammation via dilatation of ductal stenoses (with hydrostatic pressure, balloons, or the scope itself), removal of ductal debris with irrigation, removal of stones (if present), and instillation of medications.2 In our patients, we found that ductal dilatation with hydrostatic pressure from saline irrigation was sufficient in most cases to achieve the desired clinical result. We did not use balloons in our study or inject any medication (steroids or antibiotics) intraductally, and only one patient required extra instrumentation (a micro hand drill and basket for removal of a sialolith). We did modify our technique to use a powered Endo-Scrub machine Laryngoscope 125: February 2015
(Medtronic) to deliver the saline irrigation via intravenous tubing connected to the sialendoscope, thus allowing the procedure to be performed without an assistant injecting saline via a syringe. When we initially tried injecting saline with an assistant surgeon, we found that the assistant required a subjectively large amount of manual pressure to inject the saline, and the syringe had to be repeatedly replaced when the saline ran out, causing a temporary loss of endoscopic visualization. It is known that one risk of pediatric sialendoscopy, when performed bilaterally, is deep parotid lobe enlargement with temporary airway obstruction due to excessive ductal irrigation, and we are certainly cognizant of this.3 However, thus far this particular complication was not observed in the patients, even with use of the powered saline pump that was set at a low pressure. In the literature, intraoperative sialendoscopic findings include fibrinous, purulent, or mucoid debris within the ducts—as well as ductal stenosis, which is typified by a white ductal layer without the natural proliferation of blood vessels and is often found in juvenile recurrent parotitis.2,3,5 Similar intraoperative sialendoscopic findings were noted in our study as well. When sialoliths are found, they are usually within the submandibular gland3,5; and when ductal stenoses are found, they are mostly in the parotid duct.3 One of the disadvantages of pediatric sialendoscopy is that general anesthesia is typically needed in children in order to perform the procedure. However, one recent report described pediatric sialendoscopy being successfully performed under local anesthesia in seven of nine children for whom this approach was attempted.4 Another disadvantage of pediatric sialendoscopy is that during the first few hours after the procedure, swelling of the gland that underwent endoscopy is common, although it is usually asymptomatic and resolves within a few hours.1 The reported complication rate in several recent case series has ranged from 0% to 8% to 17%.2–5,7 When complications have been reported, they have been able to be resolved. Complications have included temporary postoperative airway obstruction (presumably from deep lobe parotid swelling in bilateral parotid sialendoscopy with copious ductal irrigation),3 pain and swelling 1 week postop requiring oral antibiotics,5 and Stensen’s duct perforation (2 cases in the recent literature, one associated with use of a laser3 and the other with use of a balloon,5 with both cases repaired surgically). The mean age of patients undergoing the procedure in the literature has been 6 years to 9 years,1,5 which is similar to our mean patient age (9 years old). The mean number of episodes before treatment has ranged from 4.7 to 6,1,5 whereas in our study, patients had a mean number of 11 infections prior to sialendoscopy, perhaps reflecting the lack of access to, and awareness of, sialendoscopy in our practice area . The most recent pediatric sialendoscopy case series in the English-language literature have included 6, 9, 9, 18, 38, and 70 patients.1–5,7 In studies that have looked at all cases of pediatric sialendoscopy (parotid and submandibular), the parotid gland has been the primary target in 61%, 72%, and 83% of cases.2,3,5 We found that Semensohn et al.: Pediatric Sialendsocopy
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the parotid gland was involved in 75% of our sialendoscopy patients. The most common sialendoscopic finding in cases of submandibular sialadenitis is sialolithiasis7 and ductal stenosis in cases of juvenile recurrent parotitis.1 Regarding pediatric patients, the success rate at either eliminating or reducing the severity and frequency of recurrences has been reported in recent series to range between 83% to 100%.1–3,5,7 Our results (83% success if success is defined as no recurrences; 92% success if success defined as no more than 1 recurrence), are similar to the results reported in the literature. However, it should be cautioned that follow-up is still relatively limited in our own study as well as in the literature, and as more time passes there will be more understanding regarding the long-term benefit (or lack thereof) of the procedure. In addition, some evidence exists that repeat sialendoscopy can be effective in eliminating recurrences if the gland fails to respond to an initial sialendoscopy procedure.4,5 In the literature, the ductal system has been irrigated or rinsed with medications, such as 50% xylocaine (2%) and 50% normal saline (0.9% NaCl) with 120-mg prednisolone,3 half xylocaine 2% and half NaCl 0.9%,4 or 100 mg of hydrocortisone via the sialendoscope into the gland at the completion of the procedure.1 Our group did not irrigate with any medication, in part because there is no evidence-based research that this approach is effective. In the literature, sialoliths have been broken up with a laser (2.1-um continuous YAG-thulium laser), and severe stenoses ( 3 mm) have also been opened with a laser (2.1-um continuous YAG-thulium laser), although laser use may increase the risk of ductal perforation.3 Small stones and stones that have been broken up with a laser can be removed with a wire basket. A 300-um microdrill can also be used for challenging strictures and sialoliths.1 In most series, preoperative imaging is performed (usually via ultrasonography).1,4 One drawback to sonography is that sialoliths smaller than 2 mm, which can be seen with sialendoscopy, are difficult to visualize on sonography.3 Classic sonographic findings in patients
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with parotid sialadenitis include multiple, small round/ oval hypoechoic areas throughout the gland that represent dilated ducts.1 However, when ductal stenosis is found on sialendoscopy, the presumed sonographic corresponding finding of ductal dilatation is not always seen on sonography.3 In our study, we felt that the most relevant contribution of preoperative sonography was the benefit of ruling out other potential etiologies (such as masses or tumors).
CONCLUSION Pediatric sialendoscopy is emerging as a safe, minimally invasive procedure that the authors have found to have a fast learning curve and a satisfactory rate of success at resolving cases of recurrent pediatric sialadenitis, particularly concerning the parotid gland. Because of these benefits, sialendoscopy has now become fully integrated as the preferred treatment modality in our pediatric otolaryngology practice for children with recurrent sialadenitis, prior to consideration of more invasive procedures such as parotidectomy.
BIBLIOGRAPHY 1. Shacham R, Bar Droma E, London D, Bar T, Nahlieli O. Long-term experience with endoscopic diagnosis and treatment of juvenile recurrent Parotitis. J Oral Maxillofac Surg 2009;67:162–167. 2. Jabbour N, Tibesar R, Lander T, Sidman J. Sialendoscopy in children. Int J Pediatr Otolaryngology 2010;74:347–350. 3. Martins-Cavalho C, Plouin-Gaudon I, Quenin S, et al. Pediatric sialendoscopy. A 5-year experience at a single institution. Arch Otolaryngol Head Neck Surg 2010;136:33–36. 4. Konstantinidis I, Chatziavramidis E, Tsakiropoulou E, Malliari H, Constantinidis J. Pediatric sialendoscopy under local anesthesia: limitations and potentials. Int J Pediatr Otorhinolaryngol 2011;75:245–249. 5. Hackett AM, Baranano CF, Reed M, Duvvuri U, Smith RJ, Mehta D. Sialoendoscopy for the treatment of pediatric salivary gland disorders. Arch Otolaryngol Head Neck Surg 2012;138:912–915. 6. Nguyen AM, Francis CL, Larsen CG. Salivary endoscopy in a pediatric patient with HLA-B27 seropositivity and recurrent submandibular sialadenitis. Int J Pediatr Otorhinolaryngol 2013;77:1045–1047. 7. Mikolajczak S, Meyer MF, Beutner D, Luers JC. Treatment of chronic recurrent juvenile parotitis using sialendoscopy. Acta Otolaryngol 2014; 134:531–535. 8. Katz P. New method of examination of the salivary glands: the fiberscope. Inf Dent 1990;72:785–786. 9. Nahlieli O, Baruchin AM. Sialendoscopy: three years’ experience as a diagnostic and treatment modality. J Oral Maxillofac Surg 1997;55:912–918. 10. Marchal F, Dulguerov P, Lehmann W. Interventional sialendoscopy. N Engl J Med 1999;341:1242–1243.
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