Br. J. Surg. Vol. 62 (1975) 468470
Bilateral idiopathic parotid duct strictures M. A D I S E S H I A H * SUMMARY
Details of 2 patients with idiopathic bilateral parotid duct strictures are presented. The possible aetiology and treatment m e discussed.
OBSTRUCTION of Stensen’s duct has been attributed to calculus, neuromuscular incoordination, stricture, foreign bodies, mucous plugs and trauma from erupting teeth or ill-fitting dentures (Barsony, 1925 ; Rose, 1954; Patey, 1965). Two patients have recently been seen with bilateral strictures of the parotid duct in which there was no obvious causative factor. No reports of similar cases have been found in the I i terat ure. Case reports Case 1 : A 43-year-old Asian male presented in April 1973 complaining of intermittent bilateral parotid gland swelling of 1 year’s duration. The swelling caused him discomfort; he had learned to massage his glands when swollen and this would result in a gush of saliva inside his mouth with resultant relief of his discomfort. Physical examination revealed a fit young man. Both parotid glands were slightly distended and tender. The teeth, gums and orifices of the parotid ducts looked normal. The patient was able to demonstrate the emptying of his parotid glands by massaging. A gush of saliva appeared at the duct orifice. Subsequently the gland felt normal again. Plain X-ray of his parotid glands and ducts did not reveal a calculus. Parotid sialography was performed and a film of the right side is shown in Fig. 1. There is a narrowing of the terminal 2 cm of the parotid duct. Proximally there is uniform dilatation of the main duct, but the small ducts and acini appear normal. Similar changes were found on the left side.
Fig. 1. Case 1. Right parotid sialogram showing dilatation of the main duct only with narrowing of the terminal portion.
468
Fig. 2. Ca.w 2. Left parotid sialogram showing dilatation of the main duct only with narrowing of the terminal portion.
Case 2: A 29-year-old Asian female presented in October 1972 complaining of intermittent painful bilateral parotid swelling of 1 year’s duration. The pain and swelling were invariably present on rising in the morning, and 2-3 hours later there was a sudden gush of saliva accompanied by relief of the pain and swelling. Physical examination revealed a fit young woman with slightly tender swollen parotid glands. The teeth, gums and orifices of Stensen’s duct were normal. When the gland was massaged there was a sudden gush of saliva through the orifice with deflation of the gland on that side. The parotid sialogram of the left side is shown in Fig. 2. The terminal 0.5 cm of duct is narrow and the proximal part of thc main duct is dilated. The small ducts and acini appear normal. Similar changes were seen on the right side. The patient was treated by dilatation of thc terminal portion of the duct with lacrimal duct dilators 3 and 6 months after presentation, but her symptoms remained unchanged. Subsequently she underwent parotid terminal dochoplasty in which the termination of the parotid duct was slit open up to the dilated portion. The strictured portion was resected on each side and submitted for histological examination. The duct mucosa was sutured to the buccal mucosa with interrupted 4/0 plain catgut sutures. Thc final result immediately after surgery is shown in Fig. 3. On histological examination both stricturcs showed an essentially similar nature. The normal pseudocolumnar epithelial lining had undergone squamous metaplasia, deep to which was dense fibrosis with collections of chronic inflammatory cells (mainly lymphocytes and macrophages) without any specific features. No microcalculi or mucous plugs were seen, or any iron pigment which might have indicated previous trauma. This is illustrated in Fig. 4. In contrast, Fig. 5 shows the appearance of a normal parotid duct (obtained from a 56-year-old man a t autopsy). Pseudocolumnar epithelium overlies a narrow zone of loose connective tissue with little
* University
College Hospital, London.
Bilateral idiopathic parotid duct strictures
Fig. 3. Case 2. Appearance of new right duct orifice at the completion of dochoplasty.
collagen and of scanty cellularity, outside which is buccinator muscle. Tcn days aftcr operation the patient suffered an attack of pain in each parotid region. This was thought to be due to infection and responded well to systemic antibiotic treatment. Ten months after operation she was pain-free. There was a free Row o f saliva from each duct orifice.
Discussion BarsonY (1925) described a case of idiopathic bilateral parotid duct obstruction. He suggested that obstruction may be due to neurOmuScu~arincoordination which would result in the main duct failing to release saliva. The lesion may be similar to that seen in the oesophagus in achalasia or in the colon in Hirschsprung’s disease. However, in our second patient there was histological evidence of fibrotic stricture. Rose (1954) described a ‘papillary’ variety of parotid duct stenosis. This occurred in young people, normally at the time of eruption of the third molar tooth. Food is chewed on the opposite side of the mouth with resultant trauma to the orifice of Stensen’s duct on that side. In our patients the onset ofsymptoms was unrelated to eruption of the wisdom teeth or indeed to trauma. Further, in our patients the condition was bilateral. All of Rose’s patients’ symptoms cleared up after one or two dilatations, whereas the symptoms of the present Case 2 were unrelieved by two dilatations at a n interval of 3 months. Patey (1965) has shown that ductal obstruction may be caused by mucous plugs, the mucus being secreted by glands in the parotid gland and duct itself. However, the sialographic and histological appearances in our patients were not characteristic of the disease that Patey described. Patey mentioned the triad of obstruction, depression of secretion and infection acting in a viscious cycle that might cause strictures, but in our patients there was no evidence of infection or of diminution of salivary flow. The gush of a large quantity of clear saliva produced on massaging the glands was easily demonstrable. The salivary gushing was recorded on 16-mm moving cine film in Case 2 prior to
Fig. 4. Cuse 2. Histology of the right parotid duct stricture. Note the dense fibrous tissue extending from the metaplastic squamous epithelial lining to a bundle of skeletal muscle fibres (buccinatorh HE, (‘ 75’)
Fig. 5. Histology of a normal parotid duct. HE. ( x 75.)
469
M. Adiseshiah dochoplasty. In recurrent parotitis the secretion is usually viscid, scanty and sometimes contains pus and debris. The symmetrical nature of the disease suggests that there may be a constitutional and/or dietary factor involved. Both patients rarely chew betel nut and clinically there was no evidence of submucous fibrosis. However, it is possible that a constituent used in spices in Indian cooking may be responsible. Case 1 obviously required no treatment, having learned to massage his glands and thereby empty them. Case 2 was unrelieved by dilatation and had been unable to empty the glands by massage. Hence parotid terminal dochoplasty was performed. This was followed by immediate relief of symptoms; she had one episode of infection which responded to antibiotic therapy. Subsequently she became symptomfree. A possible complication in the long term is the development of ascending parotitis. Prophylactic measures against this include adequate hydration at
470
all times and the use of sialogogues (e.g. citrousflavoured sweets).
Acknowledgements My thanks are due to Professor C. G. Clark and Mr D. A. Bailey for allowing me to report on their patients. I would like to express my gratitude to Mr M. J. Hobsley for discussing the problem of idiopathic parotid duct obstruction and to Dr P. M. Sutton and Dr C. Fisher for help with the histology. References (1925) Kasuistische mitteilung. Idiopathischestenongang-dilatation. Klin. Wochenschr. 4, 2500-2501. PATEY D. H. (1965) Inflammation of the salivary glands with particular reference to chronic and recurrent parotitis. Ann. R. Coll. Surg. Engl. 36, 26-44. ROSE s. s. (1954) A clinical and radiological survey of 192 cases of recurrent swelling of the salivary glands. Ann. R . Coll. Surg. Engl. 15, 374401. BARSONY T.
Bilateral idiopathic parotid duct strictures M. A D I S E S H I A H * SUMMARY
Details of 2 patients with idiopathic bilateral parotid duct strictures are presented. The possible aetiology and treatment m e discussed.
OBSTRUCTION of Stensen’s duct has been attributed to calculus, neuromuscular incoordination, stricture, foreign bodies, mucous plugs and trauma from erupting teeth or ill-fitting dentures (Barsony, 1925 ; Rose, 1954; Patey, 1965). Two patients have recently been seen with bilateral strictures of the parotid duct in which there was no obvious causative factor. No reports of similar cases have been found in the I i terat ure. Case reports Case 1 : A 43-year-old Asian male presented in April 1973 complaining of intermittent bilateral parotid gland swelling of 1 year’s duration. The swelling caused him discomfort; he had learned to massage his glands when swollen and this would result in a gush of saliva inside his mouth with resultant relief of his discomfort. Physical examination revealed a fit young man. Both parotid glands were slightly distended and tender. The teeth, gums and orifices of the parotid ducts looked normal. The patient was able to demonstrate the emptying of his parotid glands by massaging. A gush of saliva appeared at the duct orifice. Subsequently the gland felt normal again. Plain X-ray of his parotid glands and ducts did not reveal a calculus. Parotid sialography was performed and a film of the right side is shown in Fig. 1. There is a narrowing of the terminal 2 cm of the parotid duct. Proximally there is uniform dilatation of the main duct, but the small ducts and acini appear normal. Similar changes were found on the left side.
Fig. 1. Case 1. Right parotid sialogram showing dilatation of the main duct only with narrowing of the terminal portion.
468
Fig. 2. Ca.w 2. Left parotid sialogram showing dilatation of the main duct only with narrowing of the terminal portion.
Case 2: A 29-year-old Asian female presented in October 1972 complaining of intermittent painful bilateral parotid swelling of 1 year’s duration. The pain and swelling were invariably present on rising in the morning, and 2-3 hours later there was a sudden gush of saliva accompanied by relief of the pain and swelling. Physical examination revealed a fit young woman with slightly tender swollen parotid glands. The teeth, gums and orifices of Stensen’s duct were normal. When the gland was massaged there was a sudden gush of saliva through the orifice with deflation of the gland on that side. The parotid sialogram of the left side is shown in Fig. 2. The terminal 0.5 cm of duct is narrow and the proximal part of thc main duct is dilated. The small ducts and acini appear normal. Similar changes were seen on the right side. The patient was treated by dilatation of thc terminal portion of the duct with lacrimal duct dilators 3 and 6 months after presentation, but her symptoms remained unchanged. Subsequently she underwent parotid terminal dochoplasty in which the termination of the parotid duct was slit open up to the dilated portion. The strictured portion was resected on each side and submitted for histological examination. The duct mucosa was sutured to the buccal mucosa with interrupted 4/0 plain catgut sutures. Thc final result immediately after surgery is shown in Fig. 3. On histological examination both stricturcs showed an essentially similar nature. The normal pseudocolumnar epithelial lining had undergone squamous metaplasia, deep to which was dense fibrosis with collections of chronic inflammatory cells (mainly lymphocytes and macrophages) without any specific features. No microcalculi or mucous plugs were seen, or any iron pigment which might have indicated previous trauma. This is illustrated in Fig. 4. In contrast, Fig. 5 shows the appearance of a normal parotid duct (obtained from a 56-year-old man a t autopsy). Pseudocolumnar epithelium overlies a narrow zone of loose connective tissue with little
* University
College Hospital, London.
Bilateral idiopathic parotid duct strictures
Fig. 3. Case 2. Appearance of new right duct orifice at the completion of dochoplasty.
collagen and of scanty cellularity, outside which is buccinator muscle. Tcn days aftcr operation the patient suffered an attack of pain in each parotid region. This was thought to be due to infection and responded well to systemic antibiotic treatment. Ten months after operation she was pain-free. There was a free Row o f saliva from each duct orifice.
Discussion BarsonY (1925) described a case of idiopathic bilateral parotid duct obstruction. He suggested that obstruction may be due to neurOmuScu~arincoordination which would result in the main duct failing to release saliva. The lesion may be similar to that seen in the oesophagus in achalasia or in the colon in Hirschsprung’s disease. However, in our second patient there was histological evidence of fibrotic stricture. Rose (1954) described a ‘papillary’ variety of parotid duct stenosis. This occurred in young people, normally at the time of eruption of the third molar tooth. Food is chewed on the opposite side of the mouth with resultant trauma to the orifice of Stensen’s duct on that side. In our patients the onset ofsymptoms was unrelated to eruption of the wisdom teeth or indeed to trauma. Further, in our patients the condition was bilateral. All of Rose’s patients’ symptoms cleared up after one or two dilatations, whereas the symptoms of the present Case 2 were unrelieved by two dilatations at a n interval of 3 months. Patey (1965) has shown that ductal obstruction may be caused by mucous plugs, the mucus being secreted by glands in the parotid gland and duct itself. However, the sialographic and histological appearances in our patients were not characteristic of the disease that Patey described. Patey mentioned the triad of obstruction, depression of secretion and infection acting in a viscious cycle that might cause strictures, but in our patients there was no evidence of infection or of diminution of salivary flow. The gush of a large quantity of clear saliva produced on massaging the glands was easily demonstrable. The salivary gushing was recorded on 16-mm moving cine film in Case 2 prior to
Fig. 4. Cuse 2. Histology of the right parotid duct stricture. Note the dense fibrous tissue extending from the metaplastic squamous epithelial lining to a bundle of skeletal muscle fibres (buccinatorh HE, (‘ 75’)
Fig. 5. Histology of a normal parotid duct. HE. ( x 75.)
469
M. Adiseshiah dochoplasty. In recurrent parotitis the secretion is usually viscid, scanty and sometimes contains pus and debris. The symmetrical nature of the disease suggests that there may be a constitutional and/or dietary factor involved. Both patients rarely chew betel nut and clinically there was no evidence of submucous fibrosis. However, it is possible that a constituent used in spices in Indian cooking may be responsible. Case 1 obviously required no treatment, having learned to massage his glands and thereby empty them. Case 2 was unrelieved by dilatation and had been unable to empty the glands by massage. Hence parotid terminal dochoplasty was performed. This was followed by immediate relief of symptoms; she had one episode of infection which responded to antibiotic therapy. Subsequently she became symptomfree. A possible complication in the long term is the development of ascending parotitis. Prophylactic measures against this include adequate hydration at
470
all times and the use of sialogogues (e.g. citrousflavoured sweets).
Acknowledgements My thanks are due to Professor C. G. Clark and Mr D. A. Bailey for allowing me to report on their patients. I would like to express my gratitude to Mr M. J. Hobsley for discussing the problem of idiopathic parotid duct obstruction and to Dr P. M. Sutton and Dr C. Fisher for help with the histology. References (1925) Kasuistische mitteilung. Idiopathischestenongang-dilatation. Klin. Wochenschr. 4, 2500-2501. PATEY D. H. (1965) Inflammation of the salivary glands with particular reference to chronic and recurrent parotitis. Ann. R. Coll. Surg. Engl. 36, 26-44. ROSE s. s. (1954) A clinical and radiological survey of 192 cases of recurrent swelling of the salivary glands. Ann. R . Coll. Surg. Engl. 15, 374401. BARSONY T.
