Success and complications of four-duct ligation for sialorrhea☆

1. Introduction 

Sialorrhea is defined as the inability to control oral secretions. The condition is not due to excessive production of saliva but rather is secondary to poor swallowing function resulting from impaired neurological control of otopharyngeal muscles. The major cause of impaired motor control in children is cerebral palsy. Chronic drooling has a negative impact on the patient's social interactions, and adds to the burden of patient care.

Anticholinergic drugs such as atropine can control drooling but are not appropriate for long-term use because the high doses needed cause side effects such as urinary retention and constipation that pose greater risks to the patient's health than sialorrhea. When physical therapy, behavioral modification, and other noninvasive treatments for severe chronic drooling have proven unsuccessful, surgery is often indicated.

1.2. Duct ligation/rerouting and gland resection procedures 

One of the earliest reported surgical treatments for sialorrhea is bilateral parotid duct rerouting and bilateral SM gland excision, first performed by Wilkie in 1964 [2]. The operation gave good to excellent control of sialorrhea in 106 (86%) of 123 patients treated over a 10-year period and evaluated 1.5 years or more postoperatively. However, the complication rate in this series was 30% (Table 1).

Table 1. Success and complications of duct ligation/rerouting and/or gland resection procedures for sialorrhea

	Procedure	Reference (year)	Long-term success (%)	Major complications (cases, %)
	Parotid duct rerouting+SM gland removal	Wilkie and Brody [2] (1974)	106/123 (86%)	Wound dehiscence (3, 2%); Duct stenosis or cyst (25, 20%); Oral/dental problems (9, 7%); Septic parotitis (1, 1%); Total: 30%
	SM duct rerouting	Cotton and Richardson [1] (1981)	24/25 (96%)	Floor of mouth injection (1, 4%); Swelling requiring staging (1, 4%); Ranula (2, 8%); Total: 16%
	SM duct rerouting	Shott et al. [3] (1989)	0/6 (0%)	Not specified
	SM duct rerouting	Crysdale [4] (1989)	95/107 (89%)	Ranula (7/235, 3%); Duct obstruction (7/490, 2%); Total: 5%
	SM duct rerouting	Burton et al. [5] (1991)	17/20 (85%)	Reoperation for retention cysts (2, 10%); Resolved retention cysts (2, 10%); Total: 20%
	SM duct rerouting	Mandarious et al. [6] (1999)	47/59 (80%)	Ranula (7/79, 9%); SM gland infection (1/79, 1%); Total: 10%
	SM duct rerouting	Wilson and Henderson [7] (1999)	12/16 (75%)	Ranula (7/71, 10%); Transient airway obstruction (1/71, 1%); Total: 11%
	SM duct rerouting+SL duct ligation	Ekedahl [8] (1974)	6/11 (54%)	No major complications reported; Total: 0%
	SM duct rerouting+unilat. parotid duct ligation	Wilson and Henderson [7] (1999)	47/49 (96%)	Cheek cyst (1, 2%); Total: 2%
	Parotid duct ligation+SM gland resection	Dundas and Peterson [9] (1979)	12/14 (86%)	(unrelated?) TMJ ankylosis 3.5 years postoperatively (1); Total: 7%?
	Parotid duct ligation+SM gland resection	Brundage and Moore [10] (1989)	50/58 (86%)	Wound dehiscence (2, 3%); Septic parotitis (1, 2%); Total: 5%
	Parotid duct ligation+SM gland resection	Shott et al. [3] (1989)	21/24 (88%)	Not specified
	Four-duct (parotid+SM) ligation	Klem and Mair [11] (1999)	5/5 (100%)	No major complications reported; Total: 0%

SL, sublingual; SM, submandibular; unilat., unilateral.

SM duct rerouting alone was reported by Cotton and Richardson in 1981 to have a 96% success rate in 25 patients [1], although in later reports the success of this procedure ranged from 0% in 6 patients [3] to between 75 and 89% in four series with a total of 202 patients [4], [5], [6], [7]. Ranulas occurred in between 3 and 10% of the total of 436 patients who underwent SM duct rerouting in the five series that reported on complications [3], [4], [5], [6], [7].

In the first reported study of SM duct rerouting, with SL duct ligation, no major complications were reported but nursing staff judged postoperative sialorrhea to be improved in only six (54%) of 11 patients [8].

Bilateral parotid duct ligation and SM gland resection was reported to have a success rate of 86–88% for three series totaling 96 patients [3], [9], [10]. Complications were not specified in one series [3]. In the other two series, one case of temporomandibular joint ankylosis occurred 3.5 years after surgery [9] and wound dehiscence or septic parotitis occurred in three cases (5% of that series) [10].

In 1999, Klem and Mair reported on a new procedure for sialorrhea, four-duct (both parotid ducts and both SM ducts) ligation [11]. The success rate in their first five patients, 10–15 months postoperatively, was 100% and there were no major complications. Due to the favorable results from our study and the simplicity of the intraoral procedure, we now perform four-duct ligation in our pediatric patients for whom operative control of sialorrhea was indicated. This report documents our results on our first 21 patients we treated using this procedure.

2. Methods 

We conducted a retrospective medical record review of children in whom we performed four-duct ligation to treat sialorrhea between August 1999 and September 2000. We conducted a follow-up telephone interview with each primary caregiver. We then compared our results with those reported in the literature for four-duct ligation and other procedures to treat sialorrhea.

2.2. Evaluation of results 

Each caregiver who could be reached by telephone was asked the same set of questions regarding pre- and postoperative symptoms and complications of surgery (Fig. 1). The success of surgery was based on the caregiver's answer to the question, ‘Would you classify the patient as ‘much better, better, same, or worse’ postoperatively?’ Much better was defined for the parents as having to rarely if ever having to change the patient's clothes, not having to wear a bib, and not having to carry a towel or something similar to wipe the child's mouth. Better was defined as significantly improved but still required occasional changes of clothing or bibs.

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Fig. 1. Questionnaire for telephone follow-up with primary caregiver after four-duct ligation for sialorrhea. Four-Duct Ligation Questionnaire.

Complications were based on the caregiver's answers regarding ‘complications’ on the questionnaire and the results of medical record review.

Multiple other questions were asked regarding the patient's pre and postoperative condition and these can be seen on an example of the questionnaire in Fig. 1.

3. Results 

Caregivers of all 21 patients completed the telephone questionnaire. Caregivers classified the patient as ‘much better’ in 11 cases (52%), ‘better’ in six cases (29%), and the ‘same’ in the remaining four cases (19%). Surgery was considered successful in the 81% of cases that were rated ‘much better’ or ‘better’ after four-duct ligation.

All of the children had cerebral palsy or some neurodegenerative disorder. Their functional status was as follows. Ten patients could walk (at least three requiring a walker) and 11 patient were wheelchair bound. Nine patients could feel themselves to some degree, and 12 patients could not. Only four of the patients had intelligible language skills, the remaining 17 could not.

Preoperatively every child had to have his/her shirt or bib changed from four to ten times a day, with the large majority between six and eight times a day. Thirteen of the patients specifically report of having to constantly wipe the child's mouth in the preoperative state, with the rest referring to multiple times.

Major complications occurred in two patients. One patient had a persistent ranula and the other had chronic intermittent painful SMG sialadenitis, both of which were successfully treated with surgery. The ranula was excised and the patient with chronic sialadenitis had a bilateral submandibular gland excision and had no further problems.

Minor complications occurred in a total of four patients. One patient had a ranula that resolved; two patients had intermittent SM gland swelling that was not associated with any discomfort and eventually resolved; and one patient had postoperative tongue swelling that required care in the step-down unit, prolonging hospitalization. All patients in the immediate postoperative period had some degree of floor of mouth, submandibular and parotid swelling. The severity of this swelling, which was sometimes associated with significant discomfort was what usually determined the length of the hospital stay.

Five (24%) of the 21 patients were discharged from the hospital the day of surgery, seven (33%) were discharged after an overnight stay, and the other nine (43%) spent two to three nights in the hospital after surgery.

The parents were questioned on their subjective assessment of their child's postoperative pain on a scale of 1–5 as described in the questionnaire. The child's pain the first evening of the surgery averaged 3.475, and the pain the following day averaged 2.875. Seven parents rated the pain as a five the evening of the surgery and five felt that it continued as a five the following day. Six parents reported a level of 1 or 2 the day of the surgery and eleven reported a level of 1 or 2 the day following surgery.

Five families only after specific questioning noticed that their child's secretions were more thick postoperatively. Two families felt the secretions were considerably thicker and three thought they were mildly thicker; however, none of the families felt, this gave them any problem. One family felt their child's mouth odor worsened after the surgery, while one was very pleased at how their child's oral odor had significantly improved. Only one child had problems with dental caries afterward, in this case the parent was unsure if the caries could have been their before the operation as the visit to the dentist was the first one in some time, and only 5 months after the operation.

Overall 16 of the families were pleased with the results. One of the patients in the better group was significantly improved but was still requiring around two shirt/bib changes a day and some wiping of the mouth. The family was not satisfied and we offered SMG excision, which was performed and the patient had complete cessation of drooling. Thus, this patient and the four failures were not pleased with the initial result.

Five patients said they would not do the surgery again. These were not the same five as the previously mentioned group who were displeased with the results of the surgery. The two patients who required surgical treatment for control of a ranula and SMG sialadenitis, in spite of their ultimate good result, said that due to the complications they would not do the surgery again. The parents of a third child that had a good result said they would not repeat the surgery if given the chance because they thought the postoperative pain was too high a cost. The other two who regretted the decision to proceed with surgery were two of the failures. The other two failures were disappointed the surgery did not work but did not regret the decision to proceed with surgery.

4. Discussion 

One of the earliest reported surgical treatments for sialorrhea is bilateral parotid duct rerouting and bilateral SM gland excision, first performed by Wilkie in 1964 [2]. The operation gave good to excellent control of sialorrhea in 106 (86%) of 123 patients treated over a 10-year period and evaluated 1.5 years or more postoperatively. However, the complication rate in this series was 30% (Table 1).

SM duct rerouting alone was reported by Cotton and Richardson in 1981 to have a 96% success rate in 25 patients [1], although in later reports the success of this procedure ranged from 0% in six patients [3] to between 75 and 89% in four series with a total of 202 patients [4], [5], [6], [7]. Ranulas occurred in between 3 and 10% of the total of 436 patients who underwent SM duct rerouting in the five series that reported on complications [3], [4], [5], [6], [7].

In the first reported study of SM duct rerouting, with SL duct ligation, no major complications were reported but nursing staff judged postoperative sialorrhea to be improved in only six (54%) of 11 patients [8].

Bilateral parotid duct ligation and SM gland resection was reported to have a success rate of 86–88% for three series totaling 96 patients [3], [9], [10]. Complications were not specified in one series [3]. In the other two series, one case of temporomandibular joint ankylosis occurred 3.5 years after surgery [9] and wound dehiscence or septic parotitis occurred in three cases (5% of that series) [10].

Compared with other procedures to manage sialorrhea, we found four-duct ligation to be relatively simple to perform and 57% of our patients were able to be discharged from the hospital the same day or the day after surgery. However, the post of course can be associated with significant discomfort. In addition, our major complication rate of 10% compares well with ‘total’ complication rates reported in the literature for other procedures (0–30%; Table 1).

Our success rate of 81% in 21 patients is slightly lower than typical rates reported in the literature for most other procedures to manage sialorrhea: 86% for parotid duct rerouting and SM gland removal in 123 patients [2], a median of 85% (range 75–96%) for SM duct rerouting alone in 227 patients [1], [4], [5], [6], [7], 54% for SM duct rerouting and parotid duct ligation in 11 patients [2], 96% for SM duct rerouting with unilateral parotid duct ligation in 49 patients [7], and 86–88% for bilateral parotid duct ligation and SM gland resection in 96 patients [3], [9], [10].

Klem and Mair reported a 100% success and 0% complication rate for four-duct ligation in their initial study. However, their study included only five patients. The success and complication rates we report in our 21 patients are more consistent with those reported in the literature for other procedures and thus probably are a better reflection of the success and complication rates to be expected with four-duct ligation. Due to the simplicity of this intraoral procedure and its favorable success and complication rates, we consider four-duct ligation to be a good option when surgical management of sialorrhea is indicated.