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Pediatric sialendoscopy under local anesthesia: Limitations and potentials
International Journal of Pediatric Otorhinolaryngology 75 (2011) 245–249
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Pediatric sialendoscopy under local anesthesia: Limitations and potentials I. Konstantinidis *, A. Chatziavramidis, E. Tsakiropoulou, H. Malliari, J. Constantinidis Sialendoscopy Clinic, 2nd ORL Department, Aristotle University, Papageorgiou Hospital, Ring Road, Efkarpia, Thessaloniki 56403, Greece
A R T I C L E I N F O
A B S T R A C T
Article history: Received 31 August 2010 Received in revised form 2 November 2010 Accepted 3 November 2010 Available online 4 December 2010
Objective: Sialendoscopy in children is a recently described procedure. The small amount of existed literature presents the procedure under general anesthesia. We report our experience on pediatric sialendoscopy under local anesthesia at an outpatient setting. Methods: In a period of 1.5 years 9 children with age >8 years suffering from recurrent parotid swellings were assessed with sialendoscopy. In 8 out of 9 cases the parents accepted a procedure under local anesthesia. In 7 cases the procedure was completed uneventfully however in one case was discontinued and repeated under general anesthesia. The endoscope used was a 1.1 mm Marchal type. Prior to endoscopy xylocaine solution 4% was applied on the papilla area for 15 min. Intraductal injection of xylocaine 2% (5 ml) was performed prior the insertion of the endoscope. During the procedure parents evaluated pain by means of a 6-point smiley scale. Social life and school activity were also evaluated by means of an 11-point scale pre- and 1 year post-sialendoscopy. Results: Seven out of eight children (8 sialendoscopies) tolerated and completed the sialendoscopy assessment. The mean duration of the procedure was 39.2 min. No major complications were reported at the early post-endoscopy period. Four children presented no further swellings, two experienced one recurrence and one needed a repeat sialendoscopy (3 recurrent episodes). Sialendoscopy findings showed fibrinous debris in 4 children, mucous plugs in 2, evidence of sialodochitis with purulent debris in one child and stenosis in 3 children. The diagnosis in our cases was Juvenile Recurrent Parotitis in 6 children and chronic microbial parotitis in one case. Social life and school activity were improved in 6 children 1-year post-sialendoscopy according to parent’s ratings. Conclusions: Sialendoscopy under local anesthesia can be an alternative option in children of age >8 years and satisfactory cooperative skills, avoiding unnecessary general anesthesias and hospital stay. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Sialendoscopy Children Local anesthesia Parotid gland
1. Introduction Sialendoscopy is a new concept in the management of salivary gland disorders in childhood. It was initially described by Nahlieli et al. 10 years ago for sialolithiasis [1] and recurrent juvenile parotitis [2]. Pediatric cases were reported as part of large series (e.g. sialolithiasis papers), but not as a specific study group. Only a few papers on pediatric sialendoscopy have been published, describing the procedure, almost exclusively, under general anesthesia [3–7]. Nahlieli et al. reported a small number of children with submandibular sialolithiasis assessed under local anesthesia [1]. Due to the small diameter of child’s salivary ductal system, the sialendoscopic exploration is more challenging in children than in adults. However the need for minimal invasive methods at an outpatient basis led our team to assess the efficacy of the technique under local anesthesia, in children with an age
allowing an accepted level of co-operation. The majority of pediatric sialendoscopies concern non-lithiasic cases [4] and thus the procedure can be less interventional and potentially tolerated under local anesthesia. Numerous options have been suggested for the treatment of recurrent salivary gland swellings, including prophylactic antibiotics, duct ligation, gland excision, tympanic neurectomy, and sialendoscopy [8,9]. As a minimal invasive technique, sialendoscopy in adults decreased hospital stay, unnecessary anesthesias and open procedures. Based on these facts, we assessed the efficacy of sialendoscopy for recurrent parotid gland swelling in children under local anesthesia at a single tertiary-care Otorhinolaryngology Department. 2. Materials and methods 2.1. Patients
* Corresponding author. E-mail addresses: [email protected], [email protected] (I. Konstantinidis). 0165-5876/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2010.11.009
In a period of 1.5 years, 9 children (5 females and 4 males) with an age >8 years (mean age: 9.6 years, range 8–12) suffering from recurrent parotid swellings were referred in the Sialendoscopy
246
I. Konstantinidis et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 245–249
Clinic of our Academic Otorhinolaryngology Department. Indication for sialendoscopy was at least 2 episodes of swellings within the last 6 months. All children had prior mumps vaccination and were in good general health condition. Imaging studies were performed for all cases in the referral centres, including ultrasound in all children and sialography in two cases. No suspicion of sialolithiasis occurred in our patients. Sialendoscopy procedure was explained in full detail to the parents, offering the standard option of general anesthesia and the alternative of local anesthesia. Parents were informed that in case of a non-tolerated sialendoscopy under local anesthesia the procedure will be discontinued and a second sialendoscopy under general anesthesia will be scheduled. The Ethic committee of the Aristotle University, Thessaloniki, Greece, initially approved the study and then the procedures had the approval of the Papageorgiou Hospital Ethic Committee. The parents of 8 out of 9 children accepted a procedure under local anesthesia and signed an informed consent. In 7 children the procedure was completed without problems however in one case it was discontinued at an initial stage due to pain intolerance and anxiety and repeated under general anesthesia. In a case with bilateral episodes, parotid glands were assessed in different sessions. The mean follow-up was 14 months (minimum 12 months, range 12–17 months). The first follow up appointment was one week post-endoscopy and then every 3 months for the next year. 2.2. Procedure Initially a swab soaked with xylocaine 4% was applied on the papilla region for 10 min. Identification and dilatation of the papilla followed with the use of customized dilators (Karl Storz Co., Tuttlingen, Germany). When the insertion of a 16 gauge venous catheter was possible an injection of 5–7 ml xylocaine solution 4% was performed. Sialendoscopy assessment was performed with the child in a lying position and the head elevated in 308 (Fig. 1) at an outpatient setting. All procedures were completed under continuous monitoring. A semirigid optic endoscopic device with an external diameter of 1.1 mm was used (Karl Storz Co., Tuttlingen, Germany). Adequate view of the duct was obtained by its dilation and cleansing of the endoscope tip, using [()TD$FIG]intermittent rinsing through the endoscope with a local anesthetic
solution (half xylocaine 2% and half NaCl 0.9%). The endoscope was advanced with gentle irrigation to the branching point of the ductal system. Care was taken to avoid puncturing or lacerating the duct. The gland was lavaged during sialendoscopy with approximately 60 ml of the rinsing solution. The level of pain at every stage of the procedure was recorded by one of the parents using a 6-point smiley scale. In all cases an injection of steroids (prednisolone 150 mg) in 10 ml of NaCl 0.9% was performed at the end of the procedure. Sialendoscopy was considered successful when the entire ductal lumen and its branches were clear of any disease. Patients received oral antibiotic prophylaxis (Amoxicilline–Clavulanic acid) for 48 h, covering one day prior to endoscopy and the assessment day. Parents were asked to evaluate school activity and social life of their children as risk factors for psychological and psychosocial maladjustment [10]. Ratings were performed prior and one year after sialendoscopy, by means of an 11-point Likert scales where 0 represents ‘‘no problem’’ and 10 stands for ‘‘major problem’’. According to Walters et al. parents assessed school activity, rating two factors: (1) school absences; (2) performance in class, and social activity, rating children’s: (1) participation in after-school activities and hobbies; (2) avoidance of relations with friends and schoolmates. The mean of the two factors’ ratings was the final score for each category. The small sample size does not allow direct comparisons and this study presents only descriptive statistics of the pediatric patients included. Statistics are presented as means SD, and as percentages of the study group. 3. Results Seven children (3 males/4 females, mean age 9.8 years) completed the assessment under local anesthesia. In total eight sialendoscopies were performed as in one case a bilateral assessment was needed. The mean duration of the disease (first onset – sialendoscopy) was 6.2 years. The diagnosis was Juvenile Recurrent Parotitis (JRP) in 6 cases and chronic microbial parotitis in 1 case. All cases are analytically presented in Table 1. Diagnosis of JRP was based on history (recurrent episodes of non-suppurative parotitis) and characteristic imaging (sialectasia) and/or endoscopy findings (white ductal wall without vessels,
Fig. 1. Sialendoscopic assessment under local anesthesia of a 9 year-old girl in a lying position at an outpatient setting.
I. Konstantinidis et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 245–249
247
Table 1 Demographics, sialadenitis episodes and sialendoscopy findings (st: stenosis, fd: fibrinous debris, mp: mucous plugs, pd: purulent debris/JRP: Juvenile Recurrent Parotitis, CMP: chronic microbial parotitis). Patients
Gender
Age
Disease duration
Episodes/ 1 year prior
Episodes/ 1 year follow up
Complication
Endoscopic findings
Diagnosis
Repeat sialendoscopy
1 2
m f
8.3 10.5
6 4.5
8.5 11 9.2 12 9.3
5.5 6 7 7 8
– – – – – – – Pain/swelling
St/fd mp mp Loc st/fd fd mp sialodochitis/pd Loc st/fd
+
m f f f m
3 0 0 0 1 0 1 0
JRP JRP
3 4 5 6 7
6 R4 L3 4 5 6 7 4
stenosis). The diagnosis of chronic microbial parotitis was based on history of recurrent suppurative episodes of parotid swelling, endoscopic findings suggestive of chronic inflammation and saliva culture positive for Staph. Aureus. The main endoscopic finding in all JRP cases was a whitish appearance of the ductal layer without the healthy blood vessel coverage (Fig. 2A). Wide orifice of Stensen’s papilla was noticed in four children. Localised stenosis was present in 2 cases (Fig. 2B) and diffuse stenosis in one case. Endoscopic findings of the child with chronic microbial parotitis were suggestive of sialodochitis with intense vasculitis like red spots on the walls of Stensen’s duct (Fig. 2C). Fibrinous debris was present in four cases and mucous plugs in two cases. Sialendoscopy was therapeutic for six children. Four of them had no symptoms during the post-intervention year, and two had one episode of recurrent swelling. Only one child required a repeat sialendoscopy as he experienced 3 episodes of swellings within 6 months. Specifically the mean episode prior to sialendoscopy was 5.1/year and reduced to 0.7 at the post-intervention year (Table 1). The smiley scale results showed moderate tolerance of the procedure with a mean score of 2.6. The scores presented a gradual increase from papilla dilatation to the steroid injection as seen in Fig. 3 with the last stage being the most annoying. The mean procedure duration was 35.2 min (range 28–40 min). Swelling of the assessed gland was noticed within the first few hours after the procedure, as a result of the irrigation and steroid injection. The swelling resolved spontaneously within 24 h after the procedure with the exception of one child requiring further antibiotic treatment and massaging with resolution of symptoms in a week. No major complications such as facial palsy, bleeding or duct laceration were encountered. No open salivary gland resection was necessary. Subjective ratings of parents regarding social life and school activity indicated improvement of both domains in 6 children and no change in one. These ratings are analytically presented in Fig. 4.
[()TD$FIG]
JRP JRP JRP CMP JRP
4. Discussion In the limited literature on pediatric sialendoscopy, children account approximately the 10% of all cases of recurrent salivary gland swellings [4]. Due to the small diameter of the child’s salivary ductal system, a sialendoscopy assessment presents difficulties compared with this in adults. This is probably the main reason that almost all authors described the procedure in children under general anesthesia [3–7]. However the every day clinical praxis demands a decrease of the hospital stay, increasing the use of minimal invasive methods at an outpatient basis. The purpose of this study was to assess if this technique is applicable under local anesthesia in children and explore its limitations. The etiology of parotid swellings in children remains often unknown. Various factors have been proposed, including autoimmune, genetic, ductal obstruction and congenital abnormalities [1,3,4]. Juvenile Recurrent Parotitis (JRP) is the second most common cause of salivary gland disease in children after mumps, and one of the major causes of appointments in sialendoscopy clinics. The peak incidence occurs between age of 3 and 6, with a second peak at age 9–10 [2,11]. At this second peak many children can co-operate for an intervention as sialendoscopy under local anesthesia. In inflammatory swellings of salivary glands, dilatation and clearing of the ductal system from the debris due to continuous irrigation along with steroid injection is usually efficient and results a significant improvement of symptoms [2,3,12]. In those cases sialendoscopy provides a less invasive and time-consuming approach. On the contrary sialolithiasis cases need complex manipulations for stone retrieval, using baskets or drilling, etc. and their endoscopic assessment demands more time and patient’s tolerance [13,14]. Specifically for an interventional sialendoscopy due to sialolithiasis the mean time ranges around 70 min in the adult literature [13,15]. Nahlieli et al. reported six pediatric sialolithiasis cases treated with sialendoscopy under local anesthesia without
Fig. 2. (A) Typical white appearance of the Stensen duct with fibrinous debris in the lumen. (B) Localised stenosis not allowing further exploration of the ductal system. (C) Sialodochitis findings with vasculitis red spots on the ductal walls. (For interpretation of the references to color in this figure legend, the reader is referred to the web version of the article.)
[()TD$FIG]
I. Konstantinidis et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 245–249
248
Fig. 3. Schematic presentation of the smiley scale scoring (above) and diagram of scoring results during the sialendoscopy procedure (below).
details regarding age, location of stones and duration of the procedure in these patients. The authors of this study believe that sialolithiasis cases are not the optimal group of paediatric patients for an interventional sialendoscopy under local anesthesia. However this technique can be an alternative option in children of age >8 years old, having small stones at a proximal location. In any case sialolithiasis in children is rare ranging between 3 and 5% of all sialolithiasis cases [1,16,17]. Thus a significant percentage of sialendoscopies in children, concerning non-lithiasic cases, can be less interventional and time consuming and therefore tolerated under local anesthesia. This technique is based on the potential to irrigate the ductal system of the gland, and to inject medications under direct vision. The goal of the treatment is to stop or significantly decrease the recurrent swellings and infections of the glands and to prevent irreversible structural changes in the parotid glands. Despite the limited number of children in our series, the significant improvement in the vast majority of them is encouraging evidence. The recurrent episodes in other series range from 10 to 40% of patients. However in the vast majority of them, patients have large
[()TD$FIG]
A
symptom free intervals and a small number of them need a repeat sialendoscopy [3,4,6]. Although the majority of children tolerated and completed the procedure, the reported pain was at a moderate level. This fact does not mean that sialendoscopy cannot be applied in children under local anesthesia. However further studies are needed to assess different anesthetics, time of application or new instrumentation in order to improve the quality of the procedure in terms of pain tolerance. It is noticeable that complications did not present higher incidence under local anesthesia although one could expect that a sudden movement of the child’s head during the procedure can result injury of the duct. The detailed prior explanation of the procedure in children and what the examiner expects from them is of great value. However larger series are needed to compare safety of sialendoscopy between local and general anesthesia. In our series, it was not possible to explore the secondary branches in 3 cases out of 7. However this should be interpreted not as a limitation of local anesthesia but as a result of the JRP causing stenosis of the ductal system [2,3].
B
10
10
9
9
8
8
7
7
6
6
5
5
4
4
3
3
2
2
1
1
0
school act pre
school act post
0
social life pre
social life post
Fig. 4. Diagram presenting parents’ ratings of school activity and social life scoring pre- and post-sialendoscopy.
I. Konstantinidis et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 245–249
Previous studies showed that chronic pain conditions can result in activity restriction and school absences of children [10]. Thus activity reduction is considered as an important measure of response to chronic pain conditions [10]. The limited number of children in our series does not permit clear conclusions regarding a positive effect of sialendoscopy in social life and school activities of children. However we should keep in mind that the majority of parents’ ratings showed improvement in the above life domains. Our results should be considered as an initial indication that minimally invasive techniques under local anesthesia can improve children’s every day life without the need of a general anesthesia. Conflict of interest None declared. References [1] O. Nahlieli, E. Eliav, O. Hasson, A. Zagury, A.M. Baruchin, Pediatric sialolithiasis, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 90 (6) (2000) 709–712. [2] O. Nahlieli, R. Shacham, M. Shlesinger, E. Eliav, Juvenile recurrent parotitis: a new method of diagnosis and treatment, Paediatrics 114 (2004) 9–12. [3] S. Quenin, I. Plouin-Gaudon, F. Marchal, P. Froehlich, F. Disant, F. Faure, Juvenile recurrent parotitis sialendoscopic approach, Arch. Otolaryngol. Head Neck Surg. 134 (7) (2008) 715–719. [4] N. Jabbour, R. Tibesar, T. Lander, J. Sidman, Sialendoscopy in children, Int. J. Pediatr. Otorhinolaryngol. 74 (2010) 347–350.
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[5] F. Faurea, P. Froehlicha, F. Marchal, Paediatric sialendoscopy, Curr. Opin. Otolaryngol. Head Neck Surg. 16 (2008) 60–63. [6] F. Faure, S. Querin, P. Dulguerov, P. Froehlich, F. Disant, F. Marchal, Pediatric salivary gland obstructive swelling: sialendoscopic approach, Laryngoscope 117 (8) (2007) 1364–1367. [7] C. Martins-Carvalho, I. Plouin-Gaudon, S. Quenin, J. Lesniak, P. Froehlich, F. Marchal, et al., Pediatric sialendoscopy: a 5-year experience at a single institution, Arch. Otolaryngol. Head Neck Surg. 136 (1) (2010) 33–36. [8] M.A. Arriaga, E.N. Myers, The surgical management of chronic parotitis, Laryngoscope 100 (12) (1990) 1270–1275. [9] M. Motamed, D. Laugharne, P.J. Bradley, Management of chronic parotitis: a review, J. Laryngol. Otol. 117 (7) (2003) 521–526. [10] A.S. Walters, G.M. Williamson, The role of activity restriction in the association between pain and depression: a study of pediatric patients with chronic pain, Child. Health Care 28 (1) (1999) 33–50. [11] P. Katz, D.M. Hartl, A. Guerre, Treatment of juvenile recurrent parotitis, Otolaryngol. Clin. North Am. 42 (6) (2009) 1087–1091. [12] R. Shacham, E.B. Droma, D. London, T. Bar, O. Nahlieli, Long term experience with endoscopic diagnosis and treatment of juvenile recurrent parotitis, J. Oral Maxillofac. Surg. 67 (1) (2009) 162–167. [13] F. Marchal, P. Dulguerov, M. Becker, G. Barki, F. Disant, W. Lehmann, Specificity of parotid sialendoscopy, Laryngoscope 111 (2) (2001) 264–271. [14] H. Iro, J. Zenk, M. Koch, Modern concepts for the diagnosis and therapy of sialolithiasis, HNO 58 (3) (2010) 211–217. [15] F. Marchal, P. Dulguerov, M. Becker, G. Barki, F. Disant, W. Lehmann, Submandibular diagnostic and interventional sialendoscopy: new procedure for ductal disorders, Ann. Otol. Rhinol. Laryngol. 111 (1) (2002) 27–35. [16] L. Bodner, D.M. Fliss, Parotid and submandibular calculi in children, Int. J. Pediatr. Otorhinolaryngol. 31 (1) (1995) 35–42. [17] M.K. Chung, H.S. Jeong, M.H. Ko, H.J. Cho, N.G. Ryu, D.Y. Cho, et al., Pediatric sialolithiasis: what is different from adult sialolithiasis? Int. J. Pediatr. Otorhinolaryngol. 71 (5) (2007) 787–791.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Pediatric sialendoscopy under local anesthesia: Limitations and potentials I. Konstantinidis *, A. Chatziavramidis, E. Tsakiropoulou, H. Malliari, J. Constantinidis Sialendoscopy Clinic, 2nd ORL Department, Aristotle University, Papageorgiou Hospital, Ring Road, Efkarpia, Thessaloniki 56403, Greece
A R T I C L E I N F O
A B S T R A C T
Article history: Received 31 August 2010 Received in revised form 2 November 2010 Accepted 3 November 2010 Available online 4 December 2010
Objective: Sialendoscopy in children is a recently described procedure. The small amount of existed literature presents the procedure under general anesthesia. We report our experience on pediatric sialendoscopy under local anesthesia at an outpatient setting. Methods: In a period of 1.5 years 9 children with age >8 years suffering from recurrent parotid swellings were assessed with sialendoscopy. In 8 out of 9 cases the parents accepted a procedure under local anesthesia. In 7 cases the procedure was completed uneventfully however in one case was discontinued and repeated under general anesthesia. The endoscope used was a 1.1 mm Marchal type. Prior to endoscopy xylocaine solution 4% was applied on the papilla area for 15 min. Intraductal injection of xylocaine 2% (5 ml) was performed prior the insertion of the endoscope. During the procedure parents evaluated pain by means of a 6-point smiley scale. Social life and school activity were also evaluated by means of an 11-point scale pre- and 1 year post-sialendoscopy. Results: Seven out of eight children (8 sialendoscopies) tolerated and completed the sialendoscopy assessment. The mean duration of the procedure was 39.2 min. No major complications were reported at the early post-endoscopy period. Four children presented no further swellings, two experienced one recurrence and one needed a repeat sialendoscopy (3 recurrent episodes). Sialendoscopy findings showed fibrinous debris in 4 children, mucous plugs in 2, evidence of sialodochitis with purulent debris in one child and stenosis in 3 children. The diagnosis in our cases was Juvenile Recurrent Parotitis in 6 children and chronic microbial parotitis in one case. Social life and school activity were improved in 6 children 1-year post-sialendoscopy according to parent’s ratings. Conclusions: Sialendoscopy under local anesthesia can be an alternative option in children of age >8 years and satisfactory cooperative skills, avoiding unnecessary general anesthesias and hospital stay. ß 2010 Elsevier Ireland Ltd. All rights reserved.
Keywords: Sialendoscopy Children Local anesthesia Parotid gland
1. Introduction Sialendoscopy is a new concept in the management of salivary gland disorders in childhood. It was initially described by Nahlieli et al. 10 years ago for sialolithiasis [1] and recurrent juvenile parotitis [2]. Pediatric cases were reported as part of large series (e.g. sialolithiasis papers), but not as a specific study group. Only a few papers on pediatric sialendoscopy have been published, describing the procedure, almost exclusively, under general anesthesia [3–7]. Nahlieli et al. reported a small number of children with submandibular sialolithiasis assessed under local anesthesia [1]. Due to the small diameter of child’s salivary ductal system, the sialendoscopic exploration is more challenging in children than in adults. However the need for minimal invasive methods at an outpatient basis led our team to assess the efficacy of the technique under local anesthesia, in children with an age
allowing an accepted level of co-operation. The majority of pediatric sialendoscopies concern non-lithiasic cases [4] and thus the procedure can be less interventional and potentially tolerated under local anesthesia. Numerous options have been suggested for the treatment of recurrent salivary gland swellings, including prophylactic antibiotics, duct ligation, gland excision, tympanic neurectomy, and sialendoscopy [8,9]. As a minimal invasive technique, sialendoscopy in adults decreased hospital stay, unnecessary anesthesias and open procedures. Based on these facts, we assessed the efficacy of sialendoscopy for recurrent parotid gland swelling in children under local anesthesia at a single tertiary-care Otorhinolaryngology Department. 2. Materials and methods 2.1. Patients
* Corresponding author. E-mail addresses: [email protected], [email protected] (I. Konstantinidis). 0165-5876/$ – see front matter ß 2010 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2010.11.009
In a period of 1.5 years, 9 children (5 females and 4 males) with an age >8 years (mean age: 9.6 years, range 8–12) suffering from recurrent parotid swellings were referred in the Sialendoscopy
246
I. Konstantinidis et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 245–249
Clinic of our Academic Otorhinolaryngology Department. Indication for sialendoscopy was at least 2 episodes of swellings within the last 6 months. All children had prior mumps vaccination and were in good general health condition. Imaging studies were performed for all cases in the referral centres, including ultrasound in all children and sialography in two cases. No suspicion of sialolithiasis occurred in our patients. Sialendoscopy procedure was explained in full detail to the parents, offering the standard option of general anesthesia and the alternative of local anesthesia. Parents were informed that in case of a non-tolerated sialendoscopy under local anesthesia the procedure will be discontinued and a second sialendoscopy under general anesthesia will be scheduled. The Ethic committee of the Aristotle University, Thessaloniki, Greece, initially approved the study and then the procedures had the approval of the Papageorgiou Hospital Ethic Committee. The parents of 8 out of 9 children accepted a procedure under local anesthesia and signed an informed consent. In 7 children the procedure was completed without problems however in one case it was discontinued at an initial stage due to pain intolerance and anxiety and repeated under general anesthesia. In a case with bilateral episodes, parotid glands were assessed in different sessions. The mean follow-up was 14 months (minimum 12 months, range 12–17 months). The first follow up appointment was one week post-endoscopy and then every 3 months for the next year. 2.2. Procedure Initially a swab soaked with xylocaine 4% was applied on the papilla region for 10 min. Identification and dilatation of the papilla followed with the use of customized dilators (Karl Storz Co., Tuttlingen, Germany). When the insertion of a 16 gauge venous catheter was possible an injection of 5–7 ml xylocaine solution 4% was performed. Sialendoscopy assessment was performed with the child in a lying position and the head elevated in 308 (Fig. 1) at an outpatient setting. All procedures were completed under continuous monitoring. A semirigid optic endoscopic device with an external diameter of 1.1 mm was used (Karl Storz Co., Tuttlingen, Germany). Adequate view of the duct was obtained by its dilation and cleansing of the endoscope tip, using [()TD$FIG]intermittent rinsing through the endoscope with a local anesthetic
solution (half xylocaine 2% and half NaCl 0.9%). The endoscope was advanced with gentle irrigation to the branching point of the ductal system. Care was taken to avoid puncturing or lacerating the duct. The gland was lavaged during sialendoscopy with approximately 60 ml of the rinsing solution. The level of pain at every stage of the procedure was recorded by one of the parents using a 6-point smiley scale. In all cases an injection of steroids (prednisolone 150 mg) in 10 ml of NaCl 0.9% was performed at the end of the procedure. Sialendoscopy was considered successful when the entire ductal lumen and its branches were clear of any disease. Patients received oral antibiotic prophylaxis (Amoxicilline–Clavulanic acid) for 48 h, covering one day prior to endoscopy and the assessment day. Parents were asked to evaluate school activity and social life of their children as risk factors for psychological and psychosocial maladjustment [10]. Ratings were performed prior and one year after sialendoscopy, by means of an 11-point Likert scales where 0 represents ‘‘no problem’’ and 10 stands for ‘‘major problem’’. According to Walters et al. parents assessed school activity, rating two factors: (1) school absences; (2) performance in class, and social activity, rating children’s: (1) participation in after-school activities and hobbies; (2) avoidance of relations with friends and schoolmates. The mean of the two factors’ ratings was the final score for each category. The small sample size does not allow direct comparisons and this study presents only descriptive statistics of the pediatric patients included. Statistics are presented as means SD, and as percentages of the study group. 3. Results Seven children (3 males/4 females, mean age 9.8 years) completed the assessment under local anesthesia. In total eight sialendoscopies were performed as in one case a bilateral assessment was needed. The mean duration of the disease (first onset – sialendoscopy) was 6.2 years. The diagnosis was Juvenile Recurrent Parotitis (JRP) in 6 cases and chronic microbial parotitis in 1 case. All cases are analytically presented in Table 1. Diagnosis of JRP was based on history (recurrent episodes of non-suppurative parotitis) and characteristic imaging (sialectasia) and/or endoscopy findings (white ductal wall without vessels,
Fig. 1. Sialendoscopic assessment under local anesthesia of a 9 year-old girl in a lying position at an outpatient setting.
I. Konstantinidis et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 245–249
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Table 1 Demographics, sialadenitis episodes and sialendoscopy findings (st: stenosis, fd: fibrinous debris, mp: mucous plugs, pd: purulent debris/JRP: Juvenile Recurrent Parotitis, CMP: chronic microbial parotitis). Patients
Gender
Age
Disease duration
Episodes/ 1 year prior
Episodes/ 1 year follow up
Complication
Endoscopic findings
Diagnosis
Repeat sialendoscopy
1 2
m f
8.3 10.5
6 4.5
8.5 11 9.2 12 9.3
5.5 6 7 7 8
– – – – – – – Pain/swelling
St/fd mp mp Loc st/fd fd mp sialodochitis/pd Loc st/fd
+
m f f f m
3 0 0 0 1 0 1 0
JRP JRP
3 4 5 6 7
6 R4 L3 4 5 6 7 4
stenosis). The diagnosis of chronic microbial parotitis was based on history of recurrent suppurative episodes of parotid swelling, endoscopic findings suggestive of chronic inflammation and saliva culture positive for Staph. Aureus. The main endoscopic finding in all JRP cases was a whitish appearance of the ductal layer without the healthy blood vessel coverage (Fig. 2A). Wide orifice of Stensen’s papilla was noticed in four children. Localised stenosis was present in 2 cases (Fig. 2B) and diffuse stenosis in one case. Endoscopic findings of the child with chronic microbial parotitis were suggestive of sialodochitis with intense vasculitis like red spots on the walls of Stensen’s duct (Fig. 2C). Fibrinous debris was present in four cases and mucous plugs in two cases. Sialendoscopy was therapeutic for six children. Four of them had no symptoms during the post-intervention year, and two had one episode of recurrent swelling. Only one child required a repeat sialendoscopy as he experienced 3 episodes of swellings within 6 months. Specifically the mean episode prior to sialendoscopy was 5.1/year and reduced to 0.7 at the post-intervention year (Table 1). The smiley scale results showed moderate tolerance of the procedure with a mean score of 2.6. The scores presented a gradual increase from papilla dilatation to the steroid injection as seen in Fig. 3 with the last stage being the most annoying. The mean procedure duration was 35.2 min (range 28–40 min). Swelling of the assessed gland was noticed within the first few hours after the procedure, as a result of the irrigation and steroid injection. The swelling resolved spontaneously within 24 h after the procedure with the exception of one child requiring further antibiotic treatment and massaging with resolution of symptoms in a week. No major complications such as facial palsy, bleeding or duct laceration were encountered. No open salivary gland resection was necessary. Subjective ratings of parents regarding social life and school activity indicated improvement of both domains in 6 children and no change in one. These ratings are analytically presented in Fig. 4.
[()TD$FIG]
JRP JRP JRP CMP JRP
4. Discussion In the limited literature on pediatric sialendoscopy, children account approximately the 10% of all cases of recurrent salivary gland swellings [4]. Due to the small diameter of the child’s salivary ductal system, a sialendoscopy assessment presents difficulties compared with this in adults. This is probably the main reason that almost all authors described the procedure in children under general anesthesia [3–7]. However the every day clinical praxis demands a decrease of the hospital stay, increasing the use of minimal invasive methods at an outpatient basis. The purpose of this study was to assess if this technique is applicable under local anesthesia in children and explore its limitations. The etiology of parotid swellings in children remains often unknown. Various factors have been proposed, including autoimmune, genetic, ductal obstruction and congenital abnormalities [1,3,4]. Juvenile Recurrent Parotitis (JRP) is the second most common cause of salivary gland disease in children after mumps, and one of the major causes of appointments in sialendoscopy clinics. The peak incidence occurs between age of 3 and 6, with a second peak at age 9–10 [2,11]. At this second peak many children can co-operate for an intervention as sialendoscopy under local anesthesia. In inflammatory swellings of salivary glands, dilatation and clearing of the ductal system from the debris due to continuous irrigation along with steroid injection is usually efficient and results a significant improvement of symptoms [2,3,12]. In those cases sialendoscopy provides a less invasive and time-consuming approach. On the contrary sialolithiasis cases need complex manipulations for stone retrieval, using baskets or drilling, etc. and their endoscopic assessment demands more time and patient’s tolerance [13,14]. Specifically for an interventional sialendoscopy due to sialolithiasis the mean time ranges around 70 min in the adult literature [13,15]. Nahlieli et al. reported six pediatric sialolithiasis cases treated with sialendoscopy under local anesthesia without
Fig. 2. (A) Typical white appearance of the Stensen duct with fibrinous debris in the lumen. (B) Localised stenosis not allowing further exploration of the ductal system. (C) Sialodochitis findings with vasculitis red spots on the ductal walls. (For interpretation of the references to color in this figure legend, the reader is referred to the web version of the article.)
[()TD$FIG]
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Fig. 3. Schematic presentation of the smiley scale scoring (above) and diagram of scoring results during the sialendoscopy procedure (below).
details regarding age, location of stones and duration of the procedure in these patients. The authors of this study believe that sialolithiasis cases are not the optimal group of paediatric patients for an interventional sialendoscopy under local anesthesia. However this technique can be an alternative option in children of age >8 years old, having small stones at a proximal location. In any case sialolithiasis in children is rare ranging between 3 and 5% of all sialolithiasis cases [1,16,17]. Thus a significant percentage of sialendoscopies in children, concerning non-lithiasic cases, can be less interventional and time consuming and therefore tolerated under local anesthesia. This technique is based on the potential to irrigate the ductal system of the gland, and to inject medications under direct vision. The goal of the treatment is to stop or significantly decrease the recurrent swellings and infections of the glands and to prevent irreversible structural changes in the parotid glands. Despite the limited number of children in our series, the significant improvement in the vast majority of them is encouraging evidence. The recurrent episodes in other series range from 10 to 40% of patients. However in the vast majority of them, patients have large
[()TD$FIG]
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symptom free intervals and a small number of them need a repeat sialendoscopy [3,4,6]. Although the majority of children tolerated and completed the procedure, the reported pain was at a moderate level. This fact does not mean that sialendoscopy cannot be applied in children under local anesthesia. However further studies are needed to assess different anesthetics, time of application or new instrumentation in order to improve the quality of the procedure in terms of pain tolerance. It is noticeable that complications did not present higher incidence under local anesthesia although one could expect that a sudden movement of the child’s head during the procedure can result injury of the duct. The detailed prior explanation of the procedure in children and what the examiner expects from them is of great value. However larger series are needed to compare safety of sialendoscopy between local and general anesthesia. In our series, it was not possible to explore the secondary branches in 3 cases out of 7. However this should be interpreted not as a limitation of local anesthesia but as a result of the JRP causing stenosis of the ductal system [2,3].
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Fig. 4. Diagram presenting parents’ ratings of school activity and social life scoring pre- and post-sialendoscopy.
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Previous studies showed that chronic pain conditions can result in activity restriction and school absences of children [10]. Thus activity reduction is considered as an important measure of response to chronic pain conditions [10]. The limited number of children in our series does not permit clear conclusions regarding a positive effect of sialendoscopy in social life and school activities of children. However we should keep in mind that the majority of parents’ ratings showed improvement in the above life domains. Our results should be considered as an initial indication that minimally invasive techniques under local anesthesia can improve children’s every day life without the need of a general anesthesia. Conflict of interest None declared. References [1] O. Nahlieli, E. Eliav, O. Hasson, A. Zagury, A.M. Baruchin, Pediatric sialolithiasis, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 90 (6) (2000) 709–712. [2] O. Nahlieli, R. Shacham, M. Shlesinger, E. Eliav, Juvenile recurrent parotitis: a new method of diagnosis and treatment, Paediatrics 114 (2004) 9–12. [3] S. Quenin, I. Plouin-Gaudon, F. Marchal, P. Froehlich, F. Disant, F. Faure, Juvenile recurrent parotitis sialendoscopic approach, Arch. Otolaryngol. Head Neck Surg. 134 (7) (2008) 715–719. [4] N. Jabbour, R. Tibesar, T. Lander, J. Sidman, Sialendoscopy in children, Int. J. Pediatr. Otorhinolaryngol. 74 (2010) 347–350.
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[5] F. Faurea, P. Froehlicha, F. Marchal, Paediatric sialendoscopy, Curr. Opin. Otolaryngol. Head Neck Surg. 16 (2008) 60–63. [6] F. Faure, S. Querin, P. Dulguerov, P. Froehlich, F. Disant, F. Marchal, Pediatric salivary gland obstructive swelling: sialendoscopic approach, Laryngoscope 117 (8) (2007) 1364–1367. [7] C. Martins-Carvalho, I. Plouin-Gaudon, S. Quenin, J. Lesniak, P. Froehlich, F. Marchal, et al., Pediatric sialendoscopy: a 5-year experience at a single institution, Arch. Otolaryngol. Head Neck Surg. 136 (1) (2010) 33–36. [8] M.A. Arriaga, E.N. Myers, The surgical management of chronic parotitis, Laryngoscope 100 (12) (1990) 1270–1275. [9] M. Motamed, D. Laugharne, P.J. Bradley, Management of chronic parotitis: a review, J. Laryngol. Otol. 117 (7) (2003) 521–526. [10] A.S. Walters, G.M. Williamson, The role of activity restriction in the association between pain and depression: a study of pediatric patients with chronic pain, Child. Health Care 28 (1) (1999) 33–50. [11] P. Katz, D.M. Hartl, A. Guerre, Treatment of juvenile recurrent parotitis, Otolaryngol. Clin. North Am. 42 (6) (2009) 1087–1091. [12] R. Shacham, E.B. Droma, D. London, T. Bar, O. Nahlieli, Long term experience with endoscopic diagnosis and treatment of juvenile recurrent parotitis, J. Oral Maxillofac. Surg. 67 (1) (2009) 162–167. [13] F. Marchal, P. Dulguerov, M. Becker, G. Barki, F. Disant, W. Lehmann, Specificity of parotid sialendoscopy, Laryngoscope 111 (2) (2001) 264–271. [14] H. Iro, J. Zenk, M. Koch, Modern concepts for the diagnosis and therapy of sialolithiasis, HNO 58 (3) (2010) 211–217. [15] F. Marchal, P. Dulguerov, M. Becker, G. Barki, F. Disant, W. Lehmann, Submandibular diagnostic and interventional sialendoscopy: new procedure for ductal disorders, Ann. Otol. Rhinol. Laryngol. 111 (1) (2002) 27–35. [16] L. Bodner, D.M. Fliss, Parotid and submandibular calculi in children, Int. J. Pediatr. Otorhinolaryngol. 31 (1) (1995) 35–42. [17] M.K. Chung, H.S. Jeong, M.H. Ko, H.J. Cho, N.G. Ryu, D.Y. Cho, et al., Pediatric sialolithiasis: what is different from adult sialolithiasis? Int. J. Pediatr. Otorhinolaryngol. 71 (5) (2007) 787–791.