The surgical treatment of drooling in Leicester: 12 years experience

British Journal of Plastic Surgery (1999), 52, 335–338 © 1999 The British Association of Plastic Surgeons

The surgical treatment of drooling in Leicester: 12 years experience S. W. Wilson and H. P. Henderson* Department of Plastic Surgery, St James’ Hospital, Leeds, UK and *Department of Plastic Surgery, Leicester Royal Infirmary, Leicester, UK. SUMMARY. The experience of the surgical treatment of drooling in the 12-year period 1985–1997 has been retrospectively reviewed. Details of 71 out of a total of 79 patients were available. Of these, 69% underwent bilateral submandibular duct diversion and unilateral parotid duct ligation. Excellent or good control of drooling was obtained in 65 patients (92%) which compares favourably with other series. A ranula occurred in 10% and settled spontaneously. Transient airway obstruction occurred in one patient. Keywords: Drooling, saliva, submandibular duct diversion, parotid duct ligation.

Drooling is a common feature of both cerebral palsy (seen in approximately 10%)1 and mental retardation, and is a cause of distress to both patient and carer alike. In addition to the obvious social embarrassment, drooling can disrupt schoolwork by soiling books, and result in staining of clothes. A further feature of drooling is cheilitis,2 seen in 67% of our patients. The senior author has performed 79 surgical procedures for the correction of drooling since 1985. The early results were reported by Varma, Henderson and Cotton in 1991.3 This retrospective review includes follow-up details of those patients, in addition to details of those treated since.

is placed on the history given by the carers. Techniques to quantify drooling have been described to measure volumes of radio-labelled saliva secreted pre and postoperatively in order to assess the effectiveness of antidrooling procedures.1,4 Similar studies have also been used to confirm continued submandibular gland function following submandibular duct transposition.5 We felt it inappropriate to repeat these studies in our patients, and therefore relied on the assessment of parents or carers. Markers of the extent of drooling include the number of bibs or T-shirts used daily (range 2–20, mean 5), and the presence of cheilitis (present in 67% of our series). Drooling at meal times only is, in our opinion, an insufficient indication for surgery; drooling must be continuous throughout the waking hours. All carers were provided with an information sheet before surgery.

Patients and methods In the 12 years 1985–1997, 79 patients from all over the UK underwent a surgical procedure in Leicester to correct drooling. A retrospective review of the case records was performed. By means of subsequent telephone or written questionnaires, we were able to obtain details of 71 patients. Of the remaining eight, two patients had died of causes unrelated to the surgical procedure, and six were lost to follow-up. Details of the questionnaire are shown in Table 1. Assessment of drooling is carried out in the outpatient clinic and again on the ward before surgery. Objective measurement of drooling is difficult in children, particularly handicapped children, and emphasis Table 1

Results Details were obtained in 71 cases (44 male, 27 female). The age range was 4–59 years (mean 15 years), and the mean postoperative stay was 2 nights (range 1–11 nights). Average follow-up was 24 months (range 9–136 months). The aetiology of the drooling is shown in Table 2. The surgical procedure undertaken is shown in Table 3. The majority of our patients (69%) underwent bilateral submandibular duct diversion and unilateral Table 2

Questionnaire details

Aetiology of drooling

Cerebral palsy Mental retardation Angelman’s syndrome Down’s syndrome Learning difficulties Idiopathic Freeman–Sheldon syndrome Total

Extent of drooling before surgery? Extent of drooling after surgery? Number of daily changes of bibs/T-shirts pre- and postoperatively? Adverse effects or side effects following surgery? Dental problems following surgery? Late deterioration in drooling postoperatively? Effect of operation on social interactions? Any adverse effects on behaviour?

335

33 23 7 2 3 2 1 71

336 Table 3

British Journal of Plastic Surgery Surgical procedure

Bilateral submandibular duct diversion + unilateral parotid duct ligation Bilateral submandibular duct diversion alone Unilateral submandibular duct diversion Bilateral submandibular duct diversion + Wilkie procedure Bilateral submandibular duct diversion + reversal of Wilkie procedure

Table 4

Results of surgery (Wilkie and Brody criteria6)

Results 49 16 4 1

Number (%)

Excellent (patient dry all the time) Good (occasional salivary loss) Fair (improved but still drooling) Poor (excessively wet or too dry)

31 (44) 34 (48) 5 (7) 1 (1)

1

Table 5

parotid duct ligation; details of this procedure have been discussed previously.3 In patients deemed to have a lesser degree of drooling, diversion of both submandibular ducts alone was carried out (16 patients, 23%). In four cases, a unilateral submandibular duct diversion was performed. Our reasons for choosing a less radical operation in these four patients were as follows: two of these patients had mild drooling, one had previously undergone surgery on both parotid ducts, and one patient had undergone prior submandibular gland excision. The remainder of our series comprised one patient who had previously been treated elsewhere with unilateral parotid duct ligation and who underwent diversion of both submandibular ducts plus a Wilkie procedure6 on the unoperated parotid duct. Another patient complained of a painful cheek swelling following a previous Wilkie procedure, and was therefore treated with reversal of the Wilkie procedure combined with bilateral submandibular duct diversion. The lead surgeon was the senior author (HPH) in 67 cases, and a registrar with consultant supervision (SWW) in 4 cases. Overall, excellent or good control of drooling was obtained in 65 patients (92%). Five patients were still wet despite surgery (Tables 4 and 5). One patient, who initially had a good result following bilateral submandibular duct diversion plus parotid duct ligation, subsequently suffered a deterioration (bulbar palsy) in his neurological condition. Due to the risk of aspiration of diverted saliva from the pharynx, the antidrooling surgery had to be revised; this was easily and successfully achieved by marsupialisation of the rerouted submandibular ducts at the lateral border of the tongue. Cheilitis complicated the drooling in two thirds (48) of our patients and resolved in 47 patients following surgery. The remaining patient remained slightly wet following bilateral submandibular duct diversion alone, and is awaiting supplementary parotid duct ligation. Transient cheek, floor of mouth or submandibular swelling was a common finding after surgery on the parotid or submandibular ducts. Seventeen patients had transient oral or cheek swelling within the first week following surgery, and all cases resolved within a few days. Swelling of the cheek or submandibular region was noted in 11 patients after the first postoperative week. These episodes lasted several weeks, but all cases resolved spontaneously. The early postoperative complications (encountered in the first week; were: airway problem (1 patient) and oral candidiasis (1 patient). The airway

Results showing procedure vs outcome Outcome

Procedure BSDD + PDL BSDD alone Unilateral SDD BSDD + Wilkie BSDD + reversal Wilkie

Excellent

Good

Fair

Poor

23 6 2

24 6 2 1 1

1 4 0

1 0 0

problem was related to bleeding and respiratory distress, and settled following an overnight stay on ITU. Later postoperative complications occurring after the first week following surgery were ranula (7 patients) and cheek cyst requiring excision (1 patient). The cheek cyst followed parotid duct ligation, and required excision under general anaesthetic. Subsequent healing was uneventful. All ranulas in our series settled spontaneously without requiring further surgery. Other than the two patients with dental problems postoperatively reported in the earlier study,3 there were no long term dental sequelae. Regular dental surveillance for caries was recommended to all patients. None of the carers reported any alteration in patients’ behaviour after surgery, although 39 patients (54%) were noticed to enjoy better social contacts as a result of being dry. Several respondents noticed temporary worsening of the drooling during intercurrent illness; this has also been noticed by other authors.7 One patient, who initially had an excellent result from bilateral submandibular duct diversion plus unilateral parotid duct ligation, developed worsening drooling after he fell and fractured his mandible 1 year after surgery. The drooling persisted, and the patient was treated elsewhere with a re-routing of the remaining parotid duct (Wilkie procedure) combined with bilateral sublingual gland excision. We are not aware of any other late recurrences of drooling in our series. Discussion The majority of our patients were treated by the combination of bilateral submandibular duct diversion plus parotid duct ligation (BSDD + PDL) (49 patients), a procedure first used in this department in 1985.3 Forty-seven of these patients (96%) had excellent or good control of drooling after surgery. In those patients considered to have less severe drooling who underwent bilateral submandibular duct diversion alone (BSDD) (16 patients), 12 had excellent or good control of drooling (75%). Overall these two groups of patients (BSDD + PDL and BSDD alone) enjoyed

Surgical treatment of drooling

excellent or good control in 91% of cases. We can compare these results with other series reporting submandibular duct diversion surgery. Cotton and Richardson8 reported ‘adequate’ improvement in 96% of their patients; Bailey and Wadsworth2 reported that 88% of their patients were ‘better or much better’; Crysdale and White9 produced ‘excellent’ (chin dry) or ‘good’ (drool < once per day) control in 67%; in Burton’s series,7 90% were ‘better or much better’, O’Dwyer and Conlon10 reported 68% of patients ‘much improved’ and 24% ‘improved’. Webb et al11 reviewed 39 patients who underwent bilateral submandibular duct diversion plus unilateral parotid duct ligation in Victoria, Australia. A comparatively low 61% of patients found surgery ‘helpful’, although objective clinical measurement of drooling did show a significant improvement in severity and frequency of drooling in the majority. Our overall excellent or good control rate (92%) can also be compared with control rates in series using alternative procedures. Wilkie and Brody 86% good or excellent control;6 Dundas and Peterson 86% good or excellent;12 Brundage and Moore 86% good or excellent control.13 (All three of these series used Wilkie and Brody criteria6 for assessment of results.) Two patients had disappointing results following BSDD + PDL: one patient was rendered too dry, and now requires increased fluids at mealtimes. One patient still drools slightly, but his carers are pleased with the result and do not wish further treatment. Our standard operation, comprising bilateral submandibular duct diversion plus unilateral parotid duct ligation, seems to be an excellent procedure for the control of drooling in the majority of these patients. The procedure produces a physiological diversion of submandibular salivary flow, and also avoids the risk of parotid duct stenosis encountered with the Wilkie procedure.6 We also avoid the external scars produced by submandibular excision procedures.12,13 In patients with less severe drooling, a less radical procedure (BSDD alone) can be performed, with the possibility of additional anti-drooling surgery later on, although this has only been requested in three out of the four patients who continued to drool following BSSD. These three patients are now awaiting supplementary parotid duct ligation. The problems encountered after surgery were generally transient and self-limiting. Oral and submandibular swelling is seen in association with submandibular duct diversion, cheek swelling is related to parotid duct surgery. Post-surgical swelling has also been a feature of other authors’ series.2,7,9,13,14 When seen immediately after surgery, the swelling is related to inflammation around the operated ducts. Swelling found after the first postoperative week (which usually takes longer to settle) is likely to be related to back pressure on the salivary gland. Two patients who suffered facial swelling in the early postoperative period required further treatment with intravenous antibiotics, but the swelling settled quickly. Patient dissatisfaction with this, and other side effects of surgery were marked in one series.11 This is in direct contrast, however, to our own experience in Leicester, where patient and carer satisfaction has been very high.

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The most serious complication in our series was transient airway obstruction (one patient) which has been encountered in other series.9,11 The risk of development of airway obstruction requires vigilance by the ward staff and surgical team postoperatively, and awareness by the anaesthetist during preoperative assessment. The oral candidiasis seen in one patient was antibiotic related; this resolved with cessation of antibiotics and a short course of anti-fungal treatment. Seven of our patients (10%) developed ranulas after surgery on the submandibular duct. Ranulas are thought to arise from obstructed sublingual glands in this situation9 and similar incidences have been reported by others (Bailey 12%,2 Guerin 11%14). In Crysdale’s series,9 7% of patients required sublingual gland excision for treatment of ranula. None of our patients, however, required re-operation for ranulas, and all settled spontaneously. The low incidence of dental problems following surgery has already been noted; an increased susceptibility to dental caries was noticed following submandibular duct diversion and sublingual gland excision in one series.15 Other than the two patients reported in our original paper,3 there has been no further incidence of dental caries following anti-drooling procedures in our department. In conclusion, the quality of life was enhanced in the majority of our patients by a relatively simple operation. The operation is safe, with few serious complications. The procedure can be tailored to the individual patient, and can, if necessary, be supplemented at a later date if the patient remains wet. We would recommend bilateral submandibular duct diversion plus unilateral parotid duct ligation for patients with troublesome drooling. References 1. Ekedahl C. Surgical treatment of drooling. Acta Otolaryngol (Stockh) 1974; 77: 215–20. 2. Bailey CM, Wadsworth PV. Treatment of the drooling child by submandibular duct transposition. J Laryngol Otol 1985; 99: 1111–17. 3. Varma SK, Henderson HP, Cotton BR. Treatment of drooling by parotid duct ligation and submandibular duct diversion. Br J Plast Surg 1991; 44: 415–17. 4. Becmeur F, Horta-Geraud P, Brunot B, Maniere MC, Prulhiere Y, Sauvage P. Diversion of salivary flow to treat drooling in patients with cerebral palsy. J Pediatr Surg 1996; 31: 1629–33. 5. Hotaling AJ, Madgy DN, Kuhns LR, Filipek L, Belenky WM. Postoperative technetium scanning in patients with submandibular duct diversion. Arch Otolaryngol Head Neck Surg 1992; 118: 1331–3. 6. Wilkie TF, Brody GS. The surgical treatment of drooling: a tenyear review. Plast Reconstr Surg 1977; 59: 791–7. 7. Burton MJ, Leighton SEJ, Lund WS. Long-term results of submandibular duct transposition for drooling. J Laryngol Otol 1991; 105: 101–3. 8. Cotton RT, Richardson MA. The effect of submandibular duct rerouting in the treatment of sialorrhea in children. Otolaryngol Head Neck Surg 1981; 89: 535–41. 9. Crysdale WS, White A. Submandibular duct relocation for drooling: a 10-year experience with 194 patients. Otolaryngol Head Neck Surg 1989; 101: 87–92. 10. O’Dwyer TP, Conlon BJ. The surgical management of drooling – a 15 years follow-up. Clin Otolaryngol 1997; 22: 284–7. 11. Webb K, Reddihough DS, Johnson H, Bennett CS, Byrt T. Long-term outcome of saliva-control surgery. Dev Med Child Neurol 1995; 37: 755–62.

338 12. Dundas DF, Peterson RA. Surgical treatment of drooling by bilateral parotid duct ligation and submandibular gland resection. Plast Reconstr Surg 1979; 64: 47–51. 13. Brundage SR, Moore WD. Submandibular gland resection and bilateral parotid duct ligation as a management for chronic drooling in cerebral palsy. Plast Reconstr Surg 1989; 83: 443–6. 14. Guerin RL. Surgical management of drooling. Arch Otolaryngol 1979; 105: 535–7. 15. Ericson T, Nordblom A, Ekedahl C. Effect on caries susceptibility after surgical treatment of drooling in patients with neurological disorders. Acta Otolaryngol (Stockh) 1973; 75: 71–4.

British Journal of Plastic Surgery

The Authors Stuart W. Wilson, FRCS, FRCS(Plast), Senior Registrar, Department of Plastic Surgery, St James’ Hospital, Leeds, UK. H. P. Henderson, FRCS, Department of Plastic Surgery, Leicester Royal Infirmary, Leicester, UK. Correspondence to Mr S. W. Wilson. Paper received 29 September 1998. Accepted 3 February, 1999.