Research
Original Investigation
Treatment of Juvenile Recurrent Parotitis of Childhood An Analysis of Effectiveness Brianne Barnett Roby, MD; Jameson Mattingly, MD; Emily L. Jensen, BS; Dexiang Gao, PhD; Kenny H. Chan, MD
IMPORTANCE Juvenile recurrent parotitis (JRP) is characterized by recurrent painful swelling of the parotid gland that occurs in the pediatric population. Sialendoscopy with and without ductal corticosteroid infusion (DCI) has been found to be effective in the treatment of JRP and autoimmune parotitis.
Supplemental content at jamaotolaryngology.com
OBJECTIVE To determine the utility of instrumentation vs pharmacotherapy alone for juvenile recurrent parotitis. DESIGN, SETTING, AND PARTICIPANTS A retrospective medical record review of pediatric patients undergoing DCI without sialendoscopy at a tertiary pediatric hospital was conducted. The medical records were reviewed to determine the frequency of parotitis events before and after treatment. A multiquestion telephone survey of patients and their parents who underwent the procedure was then conducted to determine patient satisfaction. INTERVENTIONS Ductal corticosteroid infusion with hydrocortisone through catheter
inserted in the parotid duct. MAIN OUTCOMES AND MEASURES Frequency of symptoms before and after treatment and parental satisfaction with the treatment. RESULTS Twelve patients with a mean age of 6.7 years were identified. The mean duration and frequency of symptoms before the procedure were 22 months and every 2 months, respectively. Five patients had a recurrence, on average 4 months after the procedure. Four patients underwent repeated surgical procedures. All had a longer duration between episodes compared with before DCI. Eight parents participated in the survey on satisfaction with the procedure, and 75% reported improvement in their child’s life postprocedure. CONCLUSIONS AND RELEVANCE Current literature shows sialendoscopy with corticosteroid application is successful in treating JRP, but it is unclear whether corticosteroid application alone would treat JRP equally. This study shows that DCI alone has similar results as sialendoscopy with corticosteroid application, indicating that it is the corticosteroid application and not the sialendoscopy causing improvement in symptoms. Because JRP must be differentiated from sialolithiasis, we recommend ultrasonography of the involved parotid(s) prior to using DCI alone to ensure no stone is present. Sialendoscopy should be reserved to rule out other parotid ductal pathologic conditions. Author Affiliations: Department of Otolaryngology, University of Colorado School of Medicine, Aurora (Roby, Mattingly, Jensen, Chan); Department of Pediatric Otolaryngology, Children’s Hospital Colorado, Aurora, Colorado (Roby, Mattingly, Jensen, Chan); Department of Biostatistics and Informatics, Colorado School of Public Health, Aurora (Gao).
JAMA Otolaryngol Head Neck Surg. 2015;141(2):126-129. doi:10.1001/jamaoto.2014.3036 Published online December 4, 2014. 126
Corresponding Author: Brianne Barnett Roby, MD, Children’s Hospital of Colorado, 13123 E 16th Ave, Ste B158, Aurora, CO 80045 ([email protected]). (Reprinted) jamaotolaryngology.com
Copyright 2015 American Medical Association. All rights reserved.
Downloaded From: http://archotol.jamanetwork.com/ by a New York University User on 05/27/2015
Treating Juvenile Recurrent Parotitis of Childhood
Original Investigation Research
J
uvenile recurrent parotitis (JRP) is an inflammatory process that results in recurrent, painful swelling of the parotid gland. The etiology is unknown, although autoimmune, ductal obstruction, immune deficiency, and infectious causes have all been proposed.1-3 It is the second most common pediatric salivary gland disorder after mumps.4 While the most common age for it to appear is 3 to 6 years old, it ranges from a couple of months of age to puberty, at which time it usually self-resolves.2,4 To diagnose JRP, a patient must have a clinical history characterized by multiple episodes of same-sided swelling and pain. It also must be differentiated from sialolithiasis and other causes of unilateral parotid swelling, which can occasionally occur in pediatric patients. Often this is done using imaging such as ultrasonography.1,2 Treatment of JRP often begins with symptomatic treatment, including antibiotics, analgesics, warm compresses, and sialagogues. Other treatments for recurrent inflammation have included injection of the duct with a sclerosing agent, radiation, ligature of the parotid duct, tympanic neurectomy, and parotidectomy.1,2 Many of those treatments, including radiation and neurectomy, have fallen out of favor. More recently, sialendoscopy has become more common. This involves using a rigid endoscope to examine the duct for both diagnostic and therapeutic purposes. Sialendoscopy with and without ductal corticosteroid infusion (DCI) has been found to be effective in the treatment of JRP and autoimmune parotitis.1,2,5,6 However, all previous studies using corticosteroid application for JRP also involved sialendoscopy. Because not all institutions have sialendoscopy equipment, treatment with sialendoscopy is not an option in all cases. To our knowledge, this is the first study to look at corticosteroid application alone as a treatment option for JRP, which is a treatment option even at institutions without sialendoscopy equipment. Furthermore, this study also looks at the patient satisfaction with corticosteroid application.
Methods A 5-year retrospective medical record review of all pediatric patients undergoing DCI alone was conducted. Prior to beginning the study, institutional review board approval was obtained from the Children’s Hospital of Colorado. Written informed consent was not required per the institutional review board, but verbal consent was obtained from the participants when performing the survey. Demographic and clinical data collected included the following: sex, age at time of surgical procedure, ethnicity, frequency of parotitis preoperatively and postoperatively, length of time from surgery to first recurrence, number of surgical procedures, imaging, and autoimmune workup prior to surgery. In addition to the medical record review, a multiquestion telephone survey (see eFigure in the Supplement) of patients and their parents who underwent the procedure was then conducted to determine patient satisfaction on a 5-point Likert scale along with their impression for the frequency of sympjamaotolaryngology.com
Table 1. Study Population Demographics Demographic
Patients (N=12)
Sex, No. (%) Male Female Age at time of procedure, mean (range), y
7 (58) 5 (42) 6.7 (3.5-15.9)
Ethnicity, No. (%) White
7 (58)
Hispanic/Latino
3 (25)
African American
1 (8)
Multiracial
1 (8)
toms of parotitis (parotid pain, swelling, and tenderness) before and after treatment to compare with the results from the medical record review.
Results Twelve patients with a mean age (range) of 6.7 (3.5-15.9) years were identified (Table 1). Clinical data are summarized in Table 2. The mean duration and frequency of symptoms before the procedure were 22.1 months and every 2 months, respectively. Preoperative imaging studies were completed in 9 patients. No patient had sialolithiasis, but imaging showed evidence of inflammation and sialectasis in multiple patients. One patient had intraparotid lymph nodes. Laboratory tests and autoimmune workup were completed in 7 patients. One patient was positive for antineutrophil cytoplasmic antibodies (ANCA) and anti-Ro (SSA) and anti-La (SSB) antibodies, while another patient was positive for antinuclear antibodies (ANA). Rheumatology consultation was obtained in both cases, and at this time neither patient had been given a diagnosis of an autoimmune disorder that would be the cause of parotitis. All patients underwent ductal cannulation under general anesthesia until an 18-gauge angiocatheter easily fit within the duct. Hydrocortisone, 100 mg, was then instilled in the duct. The mean (range) follow-up mean was 3.8 (0.5-12.0) months. Five patients had a recurrence on average 4 months (range, 0.5-12.0 months) after the procedure. Seven patients had no recurrences after the procedure. Overall, 4 patients underwent a second procedure. All had a longer duration between episodes when compared with before DCI, although this was not statistically significant (P = .52). The Figure shows the difference in frequency with preoperative and postoperative episodes of parotitis. Eight parents participated in the survey on satisfaction with the procedure. The eFigure in the Supplement shows the questions on the survey, while Table 3 gives the results of the survey. Four parents were “extremely satisfied” according to a 5-point Likert scale, 3 parents were neutral, and 1 parent was dissatisfied with the procedure. The patient with the dissatisfied response on the survey underwent 1 procedure and was
(Reprinted) JAMA Otolaryngology–Head & Neck Surgery February 2015 Volume 141, Number 2
Copyright 2015 American Medical Association. All rights reserved.
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127
Research Original Investigation
Treating Juvenile Recurrent Parotitis of Childhood
Table 2. Parotitis Events
Table 3. Parent Satisfaction With Child’s Procedure Patients (N=12)
Preoperative Imaging, No. (%)a US
3 (25)
MRI
2 (17)
CT
6 (50)
Sialogram
2 (17)
None
3 (25) 7 (58)
Length of time with recurrent parotitis, mean (range), mo
22.1 (3-60)
Postoperative Recurrence of parotitis, No. (%)
5 (42)
Length of time between treatment and first recurrence, mean (range), wk
19.6 (2-52)
>1 Surgical procedure, No. (%)
4 (33)
Length of time between procedures, mean (range), mo
8.3 (4-15)
Abbreviations: CT, computed tomography; MRI, magnetic resonance imaging; US, ultrasonography. Total may add up to more than 12 because multiple options were selected.
Figure. Frequency of Parotitis Episodes Before and After Treatment With Ductal Corticosteroid Infusion 8 Preoperative 7
Postoperative
Patients, No.
6 5 4 3 2 1 0
More Than Once per mo
Once per mo
Once per 3 mo
Once per 6 mo
Never
Frequency of Parotitis Episodes
not seen at the follow-up appointment. The parents of the patients who underwent more than 1 procedure were “extremely satisfied” on the survey. Six parents (75%) believed that there was improvement in quality of life related to parotitis after the procedure.
Discussion Juvenile recurrent parotitis causes recurrent episodes of swelling and pain of the parotid gland. It is often self-limiting, occurring over a 5- to 10-year time frame, but symptoms can be bothersome in the interim. As the Figure shows, most of the patients in this study were having recurrent symptoms either 128
Total reachable
Parents, No. (%) 8 (67)
Overall parent satisfaction with procedure
Blood work, No. (%)
a
Variable
Very satisfied
4 (50)
Neutral
3 (38)
Very dissatisfied
1 (13)
Improvement in child’s life Significant improvement
4 (50)
Quite a bit of improvement
1 (13)
Some improvement
1 (13)
Little improvement
1 (13)
No improvement
1 (13)
Overall parent satisfaction with current parotitis Very satisfied
5 (63)
Satisfied
1 (13)
Neutral
1 (13)
Very dissatisfied
1 (13)
monthly or every couple of months before deciding to undergo treatment. In recent years, multiple articles have shown that sialendoscopy with corticosteroid application is successful in treating JRP.1,2,5,6 Table 4 summarizes the results of other studies on sialendoscopy with corticosteroid application. Yet it has remained unclear whether corticosteroid application alone would treat JRP equally as well as sialendoscopy with DCI. This is important because not all institutions have appropriate sialendoscopy equipment for pediatric patients. Previous studies have indicated that the therapeutic nature of sialendoscopy may be due to the irrigation of the duct rather than the sialendoscopy itself.2 Comparing our study of DCI alone with previous studies, DCI alone had a recurrence rate of 42%, compared with a recurrence rate of 11% to 50% noted in previous studies. Of the 5 studies, 3 used an identical dose of 100-mg hydrocortisone,1,4,6 1 used 100-mg prednisolone,2 and 1 failed to state the dose.5 Therefore, our study used the identical dose of 100-mg hydrocortisone as used in 3 of the published studies.1,4,6 All of the studies were small and assessed recurrence rates differently, making it somewhat challenging to directly compare studies. The follow-up period in other studies ranged from 10 to 30 months. Both DCI alone and sialendoscopy with DCI are low risk, with no complications being reported in the present study. With DCI alone, older children may tolerate the procedure in the clinic without general anesthesia, although that was not attempted in our study. This is in contrast to some of the more invasive historical treatments for JRP, such as tympanic neurectomy or parotidectomy. Historically, parotidectomy has been considered the gold standard for severe JRP but has the risks of facial nerve injury, ear lobe numbness, and Frey syndrome.1,4,7 The mean (range) follow-up time in this study was 3.8 0.512.0) months. This follow-up consisted of actual appointments in clinic and not follow-up with the telephone survey. The telephone survey follow-up ranged from 6 months to 5 years after procedure. On the basis of the telephone survey, a
JAMA Otolaryngology–Head & Neck Surgery February 2015 Volume 141, Number 2 (Reprinted)
Copyright 2015 American Medical Association. All rights reserved.
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jamaotolaryngology.com
Treating Juvenile Recurrent Parotitis of Childhood
Original Investigation Research
Table 4. Summary of Previous Studies on Sialendoscopy With Corticosteroid Application for Juvenile Recurrent Parotitis
Source
No. of Patients
Jabbour et al,1 2010
6
Corticosteroid Type With Dose
No Recurrences After Treatment, No. of Patients
Recurrences But Decreased Frequency After Treatment, No. of Patients
Repeated Surgery, No. of Patients
Pretherapy Frequency, Mean, mo
Posttherapy Frequency, Mean, mo
3
3
1
NA
NA
15
NA
NA
2
0.6
0.2
12
Hydrocortisone, 100 mg
Follow-up Duration, Mean, mo
Schneider et al,2 2014
15
Prednisolone, 100 mg
Cappacio et al,4 2012
14
Hydrocortisone, 100 mg
8
6
3
NA
NA
30
Hackett et al,5 2012
12
NA
8
4
3
NA
NA
10
Hydrocortisone, 100 mg
8
1
0
NA
NA
14.5
Mikolajczak et al,6 2014
9
Abbreviation: NA, data not available.
majority of those patients with short follow-up periods were satisfied with the treatment, had improvement in their symptoms, and did not believe they needed to continue follow-up. However, the short follow-up time is a weakness of the study. It is possible that other patients had recurrences that were not captured in either the medical record review or the telephone survey. Furthermore, there is always some inherent bias in telephone surveys. Pathologic conditions for pediatric parotitis are much less likely to be sialolithiasis or autoimmune disorders compared with adult populations. The literature debates whether ultrasonography, MRI, or sialography is the best imaging modality for JRP.4,7 However, in most cases JRP must be differentiated from sialolithiasis, and an ultrasound examination of the involved parotid(s) should be able to diagnosis a stone before proceeding with DCI alone. Sialendoscopy should be reserved to rule out other parotid ductal pathologic conditions. The results of the survey indicate that the majority of patients had benefited from treatment: 4 of 8 were “extremely
ARTICLE INFORMATION Submitted for Publication: June 3, 2014; final revision received September 10, 2014; accepted October 22, 2014. Published Online: December 4, 2014. doi:10.1001/jamaoto.2014.3036. Author Contributions: Drs Roby and Chan had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. Study concept and design: Roby, Chan. Acquisition, analysis, or interpretation of data: Roby, Mattingly, Jensen, Gao. Drafting of the manuscript: Roby, Mattingly. Critical revision of the manuscript for important intellectual content: All authors. Statistical analysis: Roby, Mattingly, Jensen, Gao.
jamaotolaryngology.com
satisfied” with the treatment, and 6 of 8 noted improvement in quality of life related to parotitis symptoms. Furthermore, 5 of 8 patients were “extremely satisfied” with the current parotitis symptoms. Even the patients who had recurrence of symptoms viewed the treatment favorably and believed they had benefited from the treatment. This survey indicates that patients are satisfied with the minimally invasive DCI alone treatment.
Conclusions This study shows that DCI alone had similar results compared with previous studies looking at sialendoscopy with DCI, indicating that it may be the corticosteroid application and not the sialendoscopy causing improvement in symptoms. We conclude that JRP can be treated effectively with DCI alone, which can be beneficial at institutions where pediatric sialendoscopy equipment is not available.
Administrative, technical, or material support: Mattingly. Study supervision: Chan. Conflict of Interest Disclosures: None reported. Previous Presentation: These data were presented as a poster at the American Society of Pediatric Otolaryngologists Annual Meeting; May 16-18, 2014; Las Vegas, Nevada. REFERENCES 1. Jabbour N, Tibesar R, Lander T, Sidman J. Sialendoscopy in children. Int J Pediatr Otorhinolaryngol. 2010;74(4):347-350. 2. Schneider H, Koch M, Künzel J, et al. Juvenile recurrent parotitis: a retrospective comparison of sialendoscopy versus conservative therapy. Laryngoscope. 2014;124(2):451-455.
3. Chitre VV, Premchandra DJ. Recurrent parotitis. Arch Dis Child. 1997;77(4):359-363. 4. Capaccio P, Sigismund PE, Luca N, Marchisio P, Pignataro L. Modern management of juvenile recurrent parotitis. J Laryngol Otol. 2012;126(12): 1254-1260. 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(10):912-915. 6. Mikolajczak S, Meyer MF, Beutner D, Luers JC. Treatment of chronic recurrent juvenile parotitis using sialendoscopy. Acta Otolaryngol. 2014;134(5): 531-535. 7. Katz P, Hartl DM, Guerre A. Treatment of juvenile recurrent parotitis. Otolaryngol Clin North Am. 2009;42(6):1087-1091.
(Reprinted) JAMA Otolaryngology–Head & Neck Surgery February 2015 Volume 141, Number 2
Copyright 2015 American Medical Association. All rights reserved.
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129
Original Investigation
Treatment of Juvenile Recurrent Parotitis of Childhood An Analysis of Effectiveness Brianne Barnett Roby, MD; Jameson Mattingly, MD; Emily L. Jensen, BS; Dexiang Gao, PhD; Kenny H. Chan, MD
IMPORTANCE Juvenile recurrent parotitis (JRP) is characterized by recurrent painful swelling of the parotid gland that occurs in the pediatric population. Sialendoscopy with and without ductal corticosteroid infusion (DCI) has been found to be effective in the treatment of JRP and autoimmune parotitis.
Supplemental content at jamaotolaryngology.com
OBJECTIVE To determine the utility of instrumentation vs pharmacotherapy alone for juvenile recurrent parotitis. DESIGN, SETTING, AND PARTICIPANTS A retrospective medical record review of pediatric patients undergoing DCI without sialendoscopy at a tertiary pediatric hospital was conducted. The medical records were reviewed to determine the frequency of parotitis events before and after treatment. A multiquestion telephone survey of patients and their parents who underwent the procedure was then conducted to determine patient satisfaction. INTERVENTIONS Ductal corticosteroid infusion with hydrocortisone through catheter
inserted in the parotid duct. MAIN OUTCOMES AND MEASURES Frequency of symptoms before and after treatment and parental satisfaction with the treatment. RESULTS Twelve patients with a mean age of 6.7 years were identified. The mean duration and frequency of symptoms before the procedure were 22 months and every 2 months, respectively. Five patients had a recurrence, on average 4 months after the procedure. Four patients underwent repeated surgical procedures. All had a longer duration between episodes compared with before DCI. Eight parents participated in the survey on satisfaction with the procedure, and 75% reported improvement in their child’s life postprocedure. CONCLUSIONS AND RELEVANCE Current literature shows sialendoscopy with corticosteroid application is successful in treating JRP, but it is unclear whether corticosteroid application alone would treat JRP equally. This study shows that DCI alone has similar results as sialendoscopy with corticosteroid application, indicating that it is the corticosteroid application and not the sialendoscopy causing improvement in symptoms. Because JRP must be differentiated from sialolithiasis, we recommend ultrasonography of the involved parotid(s) prior to using DCI alone to ensure no stone is present. Sialendoscopy should be reserved to rule out other parotid ductal pathologic conditions. Author Affiliations: Department of Otolaryngology, University of Colorado School of Medicine, Aurora (Roby, Mattingly, Jensen, Chan); Department of Pediatric Otolaryngology, Children’s Hospital Colorado, Aurora, Colorado (Roby, Mattingly, Jensen, Chan); Department of Biostatistics and Informatics, Colorado School of Public Health, Aurora (Gao).
JAMA Otolaryngol Head Neck Surg. 2015;141(2):126-129. doi:10.1001/jamaoto.2014.3036 Published online December 4, 2014. 126
Corresponding Author: Brianne Barnett Roby, MD, Children’s Hospital of Colorado, 13123 E 16th Ave, Ste B158, Aurora, CO 80045 ([email protected]). (Reprinted) jamaotolaryngology.com
Copyright 2015 American Medical Association. All rights reserved.
Downloaded From: http://archotol.jamanetwork.com/ by a New York University User on 05/27/2015
Treating Juvenile Recurrent Parotitis of Childhood
Original Investigation Research
J
uvenile recurrent parotitis (JRP) is an inflammatory process that results in recurrent, painful swelling of the parotid gland. The etiology is unknown, although autoimmune, ductal obstruction, immune deficiency, and infectious causes have all been proposed.1-3 It is the second most common pediatric salivary gland disorder after mumps.4 While the most common age for it to appear is 3 to 6 years old, it ranges from a couple of months of age to puberty, at which time it usually self-resolves.2,4 To diagnose JRP, a patient must have a clinical history characterized by multiple episodes of same-sided swelling and pain. It also must be differentiated from sialolithiasis and other causes of unilateral parotid swelling, which can occasionally occur in pediatric patients. Often this is done using imaging such as ultrasonography.1,2 Treatment of JRP often begins with symptomatic treatment, including antibiotics, analgesics, warm compresses, and sialagogues. Other treatments for recurrent inflammation have included injection of the duct with a sclerosing agent, radiation, ligature of the parotid duct, tympanic neurectomy, and parotidectomy.1,2 Many of those treatments, including radiation and neurectomy, have fallen out of favor. More recently, sialendoscopy has become more common. This involves using a rigid endoscope to examine the duct for both diagnostic and therapeutic purposes. Sialendoscopy with and without ductal corticosteroid infusion (DCI) has been found to be effective in the treatment of JRP and autoimmune parotitis.1,2,5,6 However, all previous studies using corticosteroid application for JRP also involved sialendoscopy. Because not all institutions have sialendoscopy equipment, treatment with sialendoscopy is not an option in all cases. To our knowledge, this is the first study to look at corticosteroid application alone as a treatment option for JRP, which is a treatment option even at institutions without sialendoscopy equipment. Furthermore, this study also looks at the patient satisfaction with corticosteroid application.
Methods A 5-year retrospective medical record review of all pediatric patients undergoing DCI alone was conducted. Prior to beginning the study, institutional review board approval was obtained from the Children’s Hospital of Colorado. Written informed consent was not required per the institutional review board, but verbal consent was obtained from the participants when performing the survey. Demographic and clinical data collected included the following: sex, age at time of surgical procedure, ethnicity, frequency of parotitis preoperatively and postoperatively, length of time from surgery to first recurrence, number of surgical procedures, imaging, and autoimmune workup prior to surgery. In addition to the medical record review, a multiquestion telephone survey (see eFigure in the Supplement) of patients and their parents who underwent the procedure was then conducted to determine patient satisfaction on a 5-point Likert scale along with their impression for the frequency of sympjamaotolaryngology.com
Table 1. Study Population Demographics Demographic
Patients (N=12)
Sex, No. (%) Male Female Age at time of procedure, mean (range), y
7 (58) 5 (42) 6.7 (3.5-15.9)
Ethnicity, No. (%) White
7 (58)
Hispanic/Latino
3 (25)
African American
1 (8)
Multiracial
1 (8)
toms of parotitis (parotid pain, swelling, and tenderness) before and after treatment to compare with the results from the medical record review.
Results Twelve patients with a mean age (range) of 6.7 (3.5-15.9) years were identified (Table 1). Clinical data are summarized in Table 2. The mean duration and frequency of symptoms before the procedure were 22.1 months and every 2 months, respectively. Preoperative imaging studies were completed in 9 patients. No patient had sialolithiasis, but imaging showed evidence of inflammation and sialectasis in multiple patients. One patient had intraparotid lymph nodes. Laboratory tests and autoimmune workup were completed in 7 patients. One patient was positive for antineutrophil cytoplasmic antibodies (ANCA) and anti-Ro (SSA) and anti-La (SSB) antibodies, while another patient was positive for antinuclear antibodies (ANA). Rheumatology consultation was obtained in both cases, and at this time neither patient had been given a diagnosis of an autoimmune disorder that would be the cause of parotitis. All patients underwent ductal cannulation under general anesthesia until an 18-gauge angiocatheter easily fit within the duct. Hydrocortisone, 100 mg, was then instilled in the duct. The mean (range) follow-up mean was 3.8 (0.5-12.0) months. Five patients had a recurrence on average 4 months (range, 0.5-12.0 months) after the procedure. Seven patients had no recurrences after the procedure. Overall, 4 patients underwent a second procedure. All had a longer duration between episodes when compared with before DCI, although this was not statistically significant (P = .52). The Figure shows the difference in frequency with preoperative and postoperative episodes of parotitis. Eight parents participated in the survey on satisfaction with the procedure. The eFigure in the Supplement shows the questions on the survey, while Table 3 gives the results of the survey. Four parents were “extremely satisfied” according to a 5-point Likert scale, 3 parents were neutral, and 1 parent was dissatisfied with the procedure. The patient with the dissatisfied response on the survey underwent 1 procedure and was
(Reprinted) JAMA Otolaryngology–Head & Neck Surgery February 2015 Volume 141, Number 2
Copyright 2015 American Medical Association. All rights reserved.
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127
Research Original Investigation
Treating Juvenile Recurrent Parotitis of Childhood
Table 2. Parotitis Events
Table 3. Parent Satisfaction With Child’s Procedure Patients (N=12)
Preoperative Imaging, No. (%)a US
3 (25)
MRI
2 (17)
CT
6 (50)
Sialogram
2 (17)
None
3 (25) 7 (58)
Length of time with recurrent parotitis, mean (range), mo
22.1 (3-60)
Postoperative Recurrence of parotitis, No. (%)
5 (42)
Length of time between treatment and first recurrence, mean (range), wk
19.6 (2-52)
>1 Surgical procedure, No. (%)
4 (33)
Length of time between procedures, mean (range), mo
8.3 (4-15)
Abbreviations: CT, computed tomography; MRI, magnetic resonance imaging; US, ultrasonography. Total may add up to more than 12 because multiple options were selected.
Figure. Frequency of Parotitis Episodes Before and After Treatment With Ductal Corticosteroid Infusion 8 Preoperative 7
Postoperative
Patients, No.
6 5 4 3 2 1 0
More Than Once per mo
Once per mo
Once per 3 mo
Once per 6 mo
Never
Frequency of Parotitis Episodes
not seen at the follow-up appointment. The parents of the patients who underwent more than 1 procedure were “extremely satisfied” on the survey. Six parents (75%) believed that there was improvement in quality of life related to parotitis after the procedure.
Discussion Juvenile recurrent parotitis causes recurrent episodes of swelling and pain of the parotid gland. It is often self-limiting, occurring over a 5- to 10-year time frame, but symptoms can be bothersome in the interim. As the Figure shows, most of the patients in this study were having recurrent symptoms either 128
Total reachable
Parents, No. (%) 8 (67)
Overall parent satisfaction with procedure
Blood work, No. (%)
a
Variable
Very satisfied
4 (50)
Neutral
3 (38)
Very dissatisfied
1 (13)
Improvement in child’s life Significant improvement
4 (50)
Quite a bit of improvement
1 (13)
Some improvement
1 (13)
Little improvement
1 (13)
No improvement
1 (13)
Overall parent satisfaction with current parotitis Very satisfied
5 (63)
Satisfied
1 (13)
Neutral
1 (13)
Very dissatisfied
1 (13)
monthly or every couple of months before deciding to undergo treatment. In recent years, multiple articles have shown that sialendoscopy with corticosteroid application is successful in treating JRP.1,2,5,6 Table 4 summarizes the results of other studies on sialendoscopy with corticosteroid application. Yet it has remained unclear whether corticosteroid application alone would treat JRP equally as well as sialendoscopy with DCI. This is important because not all institutions have appropriate sialendoscopy equipment for pediatric patients. Previous studies have indicated that the therapeutic nature of sialendoscopy may be due to the irrigation of the duct rather than the sialendoscopy itself.2 Comparing our study of DCI alone with previous studies, DCI alone had a recurrence rate of 42%, compared with a recurrence rate of 11% to 50% noted in previous studies. Of the 5 studies, 3 used an identical dose of 100-mg hydrocortisone,1,4,6 1 used 100-mg prednisolone,2 and 1 failed to state the dose.5 Therefore, our study used the identical dose of 100-mg hydrocortisone as used in 3 of the published studies.1,4,6 All of the studies were small and assessed recurrence rates differently, making it somewhat challenging to directly compare studies. The follow-up period in other studies ranged from 10 to 30 months. Both DCI alone and sialendoscopy with DCI are low risk, with no complications being reported in the present study. With DCI alone, older children may tolerate the procedure in the clinic without general anesthesia, although that was not attempted in our study. This is in contrast to some of the more invasive historical treatments for JRP, such as tympanic neurectomy or parotidectomy. Historically, parotidectomy has been considered the gold standard for severe JRP but has the risks of facial nerve injury, ear lobe numbness, and Frey syndrome.1,4,7 The mean (range) follow-up time in this study was 3.8 0.512.0) months. This follow-up consisted of actual appointments in clinic and not follow-up with the telephone survey. The telephone survey follow-up ranged from 6 months to 5 years after procedure. On the basis of the telephone survey, a
JAMA Otolaryngology–Head & Neck Surgery February 2015 Volume 141, Number 2 (Reprinted)
Copyright 2015 American Medical Association. All rights reserved.
Downloaded From: http://archotol.jamanetwork.com/ by a New York University User on 05/27/2015
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Treating Juvenile Recurrent Parotitis of Childhood
Original Investigation Research
Table 4. Summary of Previous Studies on Sialendoscopy With Corticosteroid Application for Juvenile Recurrent Parotitis
Source
No. of Patients
Jabbour et al,1 2010
6
Corticosteroid Type With Dose
No Recurrences After Treatment, No. of Patients
Recurrences But Decreased Frequency After Treatment, No. of Patients
Repeated Surgery, No. of Patients
Pretherapy Frequency, Mean, mo
Posttherapy Frequency, Mean, mo
3
3
1
NA
NA
15
NA
NA
2
0.6
0.2
12
Hydrocortisone, 100 mg
Follow-up Duration, Mean, mo
Schneider et al,2 2014
15
Prednisolone, 100 mg
Cappacio et al,4 2012
14
Hydrocortisone, 100 mg
8
6
3
NA
NA
30
Hackett et al,5 2012
12
NA
8
4
3
NA
NA
10
Hydrocortisone, 100 mg
8
1
0
NA
NA
14.5
Mikolajczak et al,6 2014
9
Abbreviation: NA, data not available.
majority of those patients with short follow-up periods were satisfied with the treatment, had improvement in their symptoms, and did not believe they needed to continue follow-up. However, the short follow-up time is a weakness of the study. It is possible that other patients had recurrences that were not captured in either the medical record review or the telephone survey. Furthermore, there is always some inherent bias in telephone surveys. Pathologic conditions for pediatric parotitis are much less likely to be sialolithiasis or autoimmune disorders compared with adult populations. The literature debates whether ultrasonography, MRI, or sialography is the best imaging modality for JRP.4,7 However, in most cases JRP must be differentiated from sialolithiasis, and an ultrasound examination of the involved parotid(s) should be able to diagnosis a stone before proceeding with DCI alone. Sialendoscopy should be reserved to rule out other parotid ductal pathologic conditions. The results of the survey indicate that the majority of patients had benefited from treatment: 4 of 8 were “extremely
ARTICLE INFORMATION Submitted for Publication: June 3, 2014; final revision received September 10, 2014; accepted October 22, 2014. Published Online: December 4, 2014. doi:10.1001/jamaoto.2014.3036. Author Contributions: Drs Roby and Chan had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. Study concept and design: Roby, Chan. Acquisition, analysis, or interpretation of data: Roby, Mattingly, Jensen, Gao. Drafting of the manuscript: Roby, Mattingly. Critical revision of the manuscript for important intellectual content: All authors. Statistical analysis: Roby, Mattingly, Jensen, Gao.
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satisfied” with the treatment, and 6 of 8 noted improvement in quality of life related to parotitis symptoms. Furthermore, 5 of 8 patients were “extremely satisfied” with the current parotitis symptoms. Even the patients who had recurrence of symptoms viewed the treatment favorably and believed they had benefited from the treatment. This survey indicates that patients are satisfied with the minimally invasive DCI alone treatment.
Conclusions This study shows that DCI alone had similar results compared with previous studies looking at sialendoscopy with DCI, indicating that it may be the corticosteroid application and not the sialendoscopy causing improvement in symptoms. We conclude that JRP can be treated effectively with DCI alone, which can be beneficial at institutions where pediatric sialendoscopy equipment is not available.
Administrative, technical, or material support: Mattingly. Study supervision: Chan. Conflict of Interest Disclosures: None reported. Previous Presentation: These data were presented as a poster at the American Society of Pediatric Otolaryngologists Annual Meeting; May 16-18, 2014; Las Vegas, Nevada. REFERENCES 1. Jabbour N, Tibesar R, Lander T, Sidman J. Sialendoscopy in children. Int J Pediatr Otorhinolaryngol. 2010;74(4):347-350. 2. Schneider H, Koch M, Künzel J, et al. Juvenile recurrent parotitis: a retrospective comparison of sialendoscopy versus conservative therapy. Laryngoscope. 2014;124(2):451-455.
3. Chitre VV, Premchandra DJ. Recurrent parotitis. Arch Dis Child. 1997;77(4):359-363. 4. Capaccio P, Sigismund PE, Luca N, Marchisio P, Pignataro L. Modern management of juvenile recurrent parotitis. J Laryngol Otol. 2012;126(12): 1254-1260. 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(10):912-915. 6. Mikolajczak S, Meyer MF, Beutner D, Luers JC. Treatment of chronic recurrent juvenile parotitis using sialendoscopy. Acta Otolaryngol. 2014;134(5): 531-535. 7. Katz P, Hartl DM, Guerre A. Treatment of juvenile recurrent parotitis. Otolaryngol Clin North Am. 2009;42(6):1087-1091.
(Reprinted) JAMA Otolaryngology–Head & Neck Surgery February 2015 Volume 141, Number 2
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