 |
|
 |
|
 |
 |
|
 |
 |
 |
 |
 |
 |
 |
|
|
|
|
|
|
|
|
|
|
|
|
|
|
|
|
|
|
* Department of Oral and Maxillofacial Surgery, Barzilai Medical Center, Ashkelon, affiliated with the Hebrew University-Hadassah School of Dental Medicine, Jerusalem, Israel
Department of Oral and Maxillofacial Surgery Barzilai Medical Center, Ashkelon
Pediatric Department, Barzilai Medical Center, Ashkelon, Israel
|| Department of Oral Medicine, Hebrew University-Hadassah School of Dental Medicine, Jerusalem, Barzilai Medical Center, Ashkelon, Israel
 |
ABSTRACT |
|---|
 TOP ABSTRACT METHODSRESULTSDISCUSSIONCONCLUSIONREFERENCES |
|---|
Methods. Twenty-six cases symptomatic JRP in children were diagnosed and treated with a combined endoscopic approach. Sialography and sialoendoscopy were performed bilaterally in all children. The treatment modality was composed of lavage, ductal dilation, and hydrocortisone injection.
Results. Sialography showed multiple sialectasis in the affected gland and in the contralateral one as well. Dilations and strictures were noticed in the main duct, and kinks could be identified in 31% of the glands. The main endoscopic finding was a white appearance of the ductal layer without the healthy blood vessel coverage. Recurrence of the symptoms occurred in only 2 (8%) children.
Conclusions. The endoscopic technique provides the possibility of a correct diagnosis and treatment. In this article, we present a new treatment modality of irrigation and dilation under direct vision by endoscopically guided miniature surgical instruments.
Key Words: juvenile parotitis • sialoendoscopy • sialography • sialectasis • therapeutic technique
Abbreviations: JRP, juvenile recurrent parotitis
Juvenile recurrent parotitis (JRP), or recurrent acute parotitis, is the second most common salivary gland disease in children, next to mumps, and in young children it is difficult to differentiate between the two. Clinical symptoms of JRP include recurrent nonobstructive, nonsuppurative parotid inflammation (Fig 1A). JRP is usually unilateral, but bilateral exacerbation can occur, with symptoms usually more prominent on 1 side. Diagnosis of JRP is made mainly through the history, by parental report, of unilateral or bilateral parotid gland recurrent infections.
|
Treatment of an acute phase of JRP is divided into conservative observation and antibiotic treatment. Cohen et al4 recommended low-dose antibiotic cover or prophylactic administration early in an attack, when a deficiency in salivary immunoglobulin A is observed. Antibiotic treatment prevents additional damage to the glandular parenchyma.
No preventive therapy against JRP has been available. A regimen of massage, encouragement of fluid intake, warmth, and use of chewing gum and sialogogues can be helpful, as can duct probing and dilation. Performing sialography proved to be useful in improving the symptoms.7 Other treatment methods mentioned in the literature are much more invasive, such as duct ligation, parotidectomy, and tympanic neurectomy.6 In this article, we present our experience with 26 children whose JRP was diagnosed and treated with a combined endoscopic approach.
|
METHODS |
|---|
|
TOPABSTRACTMETHODSRESULTSDISCUSSIONCONCLUSIONREFERENCES |
|---|
All patients were referred to ultrasonography, most of them by the pediatrician, and the rest during our examination before surgery. Our indication for endoscopic intervention was at least 2 swelling events occurring during a 12-month period. All of the procedures were performed under general anesthesia. Bilateral sialography under constant fluoroscopy (dye, Ultravist 300 Iopromide; Schering, Berlin, Germany) was conducted in all patients to determine a primary diagnosis.
After this procedure, bilateral sialoendoscopy with Nahlieli sialoendoscope diagnostic type (1.3 mm) was performed.8,9 The Stensen's duct was examined, and secondary ducts were viewed as well when it was possible. The gland was lavaged thoroughly with 60 mL of normal saline (each side). The sialoendoscope was also used to dilate the duct with saline pressure, and, when necessary, a dilation balloon was used.
After the lavage and dilation, hydrocortisone (100 mg) was injected via the endoscope into the gland. After the procedure, all patients were given intravenous Augmentin (amoxicillin-clavulanic acid 25 mg/kg). Follow-up was performed up to 36 months after surgery.
|
RESULTS |
|---|
|
TOPABSTRACTMETHODSRESULTSDISCUSSIONCONCLUSIONREFERENCES |
|---|
All patients had a dominant affected gland, although 6 had intermittent swelling episodes of the contralateral gland. One of the 26 children had bronchial asthma, 1 had glomerulonephritis, and another had Down syndrome. All other children were systemically healthy. One patient underwent right superficial parotidectomy at another medical center and shortly thereafter had contracted JRP of the left parotid gland.
Clinical Findings
The most pathognomonic sign is Stensen's papilla (Fig 1B). When visualized carefully with magnification loops (x3.5), the orifice (in both glands) is usually wide and yellow plaques can be seen in the saliva extracted from the gland. A firm mass can be felt when the symptomatic gland is palpated.
Sialographic Results
The main pathology of the parotid gland was multiple sialectasis of the affected gland. The contralateral gland was affected as well in 19 (73%) of the 26 patients involved in this study. Dilations and strictures were noticed in the main duct (Fig 2A), and kinks could be identified in 8 (31%) of the glands (Fig 2B), all of which were in the affected ones.
|
|
Treatment Result
In 2 (8%) children, we had to repeat our endoscopic technique because of recurrence of the symptoms. In these 2 cases, the affected gland was the contralateral gland. In 2 patients, we had to use a balloon (Fogarty, 2 Fr) to dilate the duct of the affected gland. All other children were free of symptoms during follow-up ranging from 4 to 36 months.
In the first few hours after the procedure, bilateral swelling of the parotid glands was noticed as a result of the irrigation. The swelling resolved spontaneously within 12 hours after the procedure.
|
DISCUSSION |
|---|
|
TOPABSTRACTMETHODSRESULTSDISCUSSIONCONCLUSIONREFERENCES |
|---|
The main disease from which the physician has to differentiate JRP is mumps, caused by a paramyxovirus transmitted by direct contact with salivary drops. In this case, the child will develop fever, malaise, headache, and chills, whereas with JRP, the symptoms are usually more local to the parotid gland, with systemic fever developing occasionally. Moreover, 90% of the population provided with mumps vaccine gain immunity for life. Other conditions such as juvenile Sjögren syndrome, pneumoparotid, lymphoma, and acquired immune deficiency syndrome can be also taken into consideration when making the differential diagnosis.
Histopathologic inspection of the diseased gland reveals lymphocyte infiltrate that tends to form lymphoid follicles and small ductal dilations. These intraductal cyst-like dilations are demonstrated clearly in sialography10 and are called sialectases. When examined, the children usually exhibit sialectases of the asymptomatic contralateral gland as well.1 Ultrasonographic examinations demonstrate multiple small hypoechogenic areas and punctate calcifications, corresponding to the sialectases demonstrated on sialography.
The history of JRP includes various theories about the cause of the disease. Its self-limiting nature, the reports of a gender distribution favoring boys, and the absence of autoimmune antibodies indicate that autoimmune disease is a less probable cause of JRP. Nevertheless, we have to consider primary Sjögren syndrome when JRP is assessed, despite the rareness of this condition.3
Ericson et al1 found no support for the suggestion that JRP is an allergic condition. Reid et al2 found a pattern of autosomal dominant inheritance with incomplete penetrance, suggesting that, at least in some cases, genetic factors are involved. One of the most probable causes of JRP is retrograde infection, mostly during dehydration of the child, as occurs in upper respiratory tract infection. The dehydration causes a decrease in salivary secretion of the parotid gland, which is low even under normal conditions, providing a ground for bacterial infection.5
As our technology skills improve, our knowledge progresses. In the past, only indirect diagnostic techniques were available. Today, introducing miniature endoscopic telescopes into the parotid glands provides an excellent possibility for correct diagnosis of intraductal pathology. Moreover, inserting miniature surgical instruments and injecting medications into the gland opens a new treatment modality.8,9
From our demographic data and from the literature,1,4–6 it is clear that the majority (54%) of JRP cases involve boys. The first symptom occurred at an average age of 3.8 years, and the symptoms for boys were exhibited >1 year earlier than for girls (3.3 boys, 4.7 girls).
The sialographic studies showed a combination of sialectasis, strictures, dilations, and kinks. In most (73%) of the cases, the radiologic findings were bilateral.
The clinical intraoral examination shows a wide opening of the Stensen's papilla with plaque exudates. Endoscopic examination leaves no doubt that there is an avascular appearance of the ductal layer (when compared with normal glands). The avascular nature of the Stensen's ductal layer hints at the possible cause of JRP. We can see such phenomena in adults who have long-standing chronic recurrent parotitis. Because the affected glands were symptomatic only for short periods (first symptom, age 3.8; average age, 7), we think that it is more than likely that the ductal avascular appearance is primary. Our assumption is that the lack of vascularity in the ductal layer interferes with the sphincteral system of the parotid gland8,9 and with the ability of the ductal system to drain the saliva out of the gland, which leads to stricture obstruction and chronic infection of the gland.
Our treatment modality is directed to lavage the ductal system and the sialectases from plaques and to dilate strictures. The first usage of lavage to treat JRP was done by Galili et al,7 who used sialography and reported good results. The advantage of our technique lies in the possibility to irrigate the ductal system of the gland, to insert surgical instruments, and to inject medications, all under direct vision. The goal of the treatment is to stop the recurrent swellings and infections of the glands before puberty and to prevent irreversible changes in the parotid glands.
A search of the literature reveals no episodes of JRP after puberty. We do not know the reason for this self-limiting behavior of the affected glands, but we assume that most of them become atrophic. We encounter few adult patients with chronic recurrent parotitis for whom, when reviewing their history, we discovered that they used to have parotid swelling episodes during their childhood.
|
CONCLUSION |
|---|
|
TOPABSTRACTMETHODSRESULTSDISCUSSIONCONCLUSIONREFERENCES |
|---|
|
FOOTNOTES |
|---|
Reprint requests to (O.N.) Oral and Maxillofacial Surgery Department, Barzilai Medical Center, 78306-Ashkelon, Israel. E-mail: nahlieli{at}yahoo.com
|
REFERENCES |
|---|
|
TOPABSTRACTMETHODSRESULTSDISCUSSIONCONCLUSIONREFERENCES |
|---|
This article has been cited by other articles:
 |
|
 |
|
  S. Quenin, I. Plouin-Gaudon, F. Marchal, P. Froehlich, F. Disant, and F. Faure Juvenile Recurrent Parotitis: Sialendoscopic Approach Arch Otolaryngol Head Neck Surg, July 1, 2008; 134(7): 715 - 719. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 D. A. Brooks and R. Clover Pertussis Infection in the United States: Role for Vaccination of Adolescents and Adults J Am Board Fam Med, November 1, 2006; 19(6): 603 - 611. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 O. Nahlieli, L. H. Nakar, Y. Nazarian, and M. D. Turner Sialoendoscopy: A new approach to salivary gland obstructive pathology. J Am Dent Assoc, October 1, 2006; 137(10): 1394 - 1400. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 K. M. Tatti, K.-H. Wu, G. N. Sanden, P. Greer, J. Sumner, J. Guarner, C. D. Paddock, and S. R. Zaki Molecular Diagnosis of Bordetella pertussis Infection by Evaluation of Formalin-Fixed Tissue Specimens. J. Clin. Microbiol., March 1, 2006; 44(3): 1074 - 1076. [Abstract] [Full Text] [PDF]
|
|
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||
 |
||
|
||
Home | My Pediatrics | Journal Information | Current Issue | Past Issues | Subscriptions & Services | Contact Us Click here for faster international access © 2004 American Academy of Pediatrics. All rights reserved. |
||
|
||
