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Sublingual gland salivary fistula and sialocele
Sublingual gland salivary fistula and sialocele Sidney L. Bronstein, D.D.S., M.Sc.D..* UNIVERSITY
and Morris S. Clark, D.D.S., ** Denver, Colo.
OF COLORADO SCHOOL OF DENTISTRY
A case of cutaneous salivary fistula and sialocele in the submental area, arising from the sublingual gland, is reported and its etiology, development, and management are discussed. The entity of plunging ranula is compared to the lesion the case presented, and the literature regarding this lesion is reviewed. The successful surgical treatment of this case supports the theory that the sublingual gland is the source of the disorder. The effect of the altered salivary function on the adjacent submandibular gland and duct presented an operative complication. (ORAL SURG. 57:357-36 1, 1984.)
S
ubmental sublingual gland salivary fistula with sialocele occurs infrequently and has not been reported in the literature. Fistulas that develop following parotid surgery or injury are more numerous and better documented,’ and an involvement with sialocele formation has been described.‘,* A variety of lesions are common to the floor of the mouth and may be seen extraorally as swellings of the submental region. These include dental abscess, minor salivary gland mucocele, oral ranula, major salivary glarid adenitis or neoplasm, dermoid cyst, branchial cleft cyst, thyroglossal duct cyst, and plunging ranula. 3*4Rarely will any of these lesions fistulate unless traumatized.4 It is the purpose of this article to report a salivary fistula and sialocele which followed surgical trauma, to discuss the diagnosis and pathogenesis of this lesion ai a plunging ranula, and to describe its surgical manag,ement. CASE REPORT
In November, 1974, the patient, then a 6%year-old girl, underwent treatment of several primary teeth, including pulpotomies of two lower primary molars. About 7 to 10 days later a midline swelling appeared under the chin adjacent to the mandible. This swelling was occasionally painful and somewhat tender but never appeared to be inflammatory. Erythromycin was ineffective, but Ampicil-
*Associate Professor and Chairman, Division of Oral and Maxil-
lofacial Surgery,Schoolof Dentistry. **Assistant Professor,Division of Oral and Maxillofacial Surgery, School of Dentistry.
lin therapy was instituted and within a week the swelling decreasedbut never completely disappeared. Approximately 2 months later, on Jan. 19, 1975, her pediatrician wanted the lesion biopsied and, with a presumptive diagnosis of thyroglossal duct cyst, the child was admitted for surgery. The procedure was described as quite difficult since the tissue was not well formed and appeared necrotic. The microscopic diagnosis was reported as “necrotic and inflammatory granulation tissue.” Healing progressednormally for about 3 months, but then the incision openedand drained a clear, viscous, mucoid material. This would “stretch out like an elastic band,” according to the patient’s mother, and could be broken only with difficulty. The area did not appear infected at this time; recurrent swelling alternating with drainage occurred. A dental cause was investigated but was discarded. In July, 1976, the area was operated on again and findings similar to those of the original operation were reported. Within 2 weeksthe lesion reappearedand the thick mucoid drainage resumed. At this time the surgeons believed that the surgical intervention had been minimal, and in May, 1977, a classic thryoglossal duct cyst excision, which included the central portion of the hyoid bone, was performed. Microscopic diagnosis was “tissue consistent with thyroglossal duct cyst and chronic inflammation.” After a short time healing was interrupted by drainage exactly as before. After moving to Denver, Colorado, the patient was referred by the Otolaryngology Service of University Hospital and was seenin February and again in November of 1981. At this latter appointment the patient reported increased discomfort at mealtimes, culminating in increased mucous secretion from the submental fistula. There was equivocal intraoral tendernessonly over the left sublingual gland. Surgical removal of one or possibly both sublingual glands was contemplated and submandibular 357
358
Bronstein and Clark
Oral Surg.
April, I984
Fig. 1. Sialogram of right submandibular and sublingual glands. Seen are ectatic submandibular ducts (solid arrows) and prominent sublingual ducts (open arrows) leaving enlarged sublingual gland to enter submandibular duct.
sialography was scheduled. The left glands were reported as normal and, indeed, showed active reflux during fluorography; the right submandibular gland showed ductal dilatation, and the right sublingual gland was enlarged to two to three times its normal size and showed ductal dilatation also. The only laboratory tests that had ever been performed were an amylase test in 1977, which showed a very low level, and an acid-fast culture by the Otolaryngology Service in 1981, which was negative. With this extreme chronic history, evidence of increasing adenitis, continued drainage, and an abnormal sialogram, removal of the right sublingual gland and of the fistula was advised. On Jan. 15, 1982, with the patient under general anesthesia, following preservation of the submandibular duct by probing, an incision was made just lateral to the duct and the gland was excised along with two large dilated secondary ducts seento enter it from the submandibular duct; these enlarged ducts (Fig. 1) were ligated prior to excision. Blunt dissection with peanut spongeswas used mainly to protect the duct and the lingual nerve. The gland was noted to be approximately three to four times normal size. A portion of the gland was closely connected to the anterior part of the mylohyoid muscle, but no definite extension could be identified below the muscle. After thorough exploration the lingual nerve was noted to be intact, but there was a tear in the submandibular duct at its exit from the gland. The duct was cannulated with a length of 18-gauge polyethylene tubing and was fixed in place by sutures to provide luminal supports and patency. A good flow of clear saliva was obtained. The oral incision was closed in normal fashion, and the cutaneous fistula was excised after preparation and redraping of the submental area. No tract upward into the floor of the mouth could be located, but a well-defined
scarred mass with a central, saccular outpouching was identified and removed after suture ligation of its deep attachment. The tissues were closed in layers, the wound was dressed,and the procedure was terminated. Specimensof gland, duct, and fistula were submitted for histopathologic examination and were reported as ectatic gland (Fig. 2) normal duct, and “focally ulcerated squamous epithelium the saccular outpouching is distally lined by columnar epithelial cells. Proximally the outpouching is ulcerated and lined by inflamed, hemorrhagic granulation tissue” (Fig. 3). Postoperatively, maintenance of the submandibular gland was quite difficult, as the tubing was continually displaced from the gland and could be replaced only with difficulty. However, clear saliva could be obtained when the tubing was correctly situated. Ten days postsurgically the tubing had to be removed but normal drainage could be noted from what appeared to be an oral fistula. Unfortunately, this fistula did not develop and the patient reported increasing discomfort in the right submandibular area at mealtimes, although the symptoms were gone within 20 minutes after meals. These symptoms gradually decreased in severity and by 2 months postoperatively discomfort was noted only upon stimulation with sweet and sour foods. In May, 1982, the patient returned with right submandibular swelling, not associatedwith eating. This was very tender, of sudden onset, and resolved completely within 4 days with penicillin therapy. Only minor episodesof diminishing submandibular adenitis recurred for a time and then ceased. The submental area healed normally, and no drainage ever occurred postoperatively. At examination on July 25, 1983, the patient was completely asymptomatic and was enjoying a normal diet. Plastic repair of the submental scar is anticipated. Lingual nerve sensation, decreased in the first few months postoperatively, was normal. DISCUSSION
The management of this case was complicated by the history of dental treatment, with a submental swelling appearing within a short interval afterward; however, the nature and location of the swelling were not typical of a dental infection. It appeared to be, according to the patient’s mother, like a “sebaceous cyst,” close to the skin, without signs of inflammation. However, it did respond to Ampicillin, so infection was present and the dental treatment may have contributed to an alteration of sublingual gland structure or function, promoting formation of the
sialocele and fistula. During the more than 2 months from the decrease of the swelling to the initial surgery, the simple extravasation and accompanying mucus-escape phenomenon developed. The plunging ranula, also known as an oral ranula
with a cervical extension, it formed by extravasation of salivary secretion, usually from the sublingual gland.6 Differentiation
has been made between the
Sublingual gland salivary jistula and sialocele 359
Volume 57 Number 4
Fig.
2. High-power photomicrograph showing thinning of ductal epithelium.
Fig. 3. Low-power photomicrograph of fistulous opening with saccular structure lined with ductal epithelium.
oral ranula as being an extravasation cyst from the
sublingual gland generally and the plunging ranula as being formed from the posterior portion only of this gland. L Neither of these lesions is now considered as a cyst because no epithelial lining can be found; rather, they are considered extravasation phenomena.) It has been postulated that an aberrant sublingual duct may allow stasis in the deep lobe of the sublingual gland with obstruction and extravasation.6 Hiatuses in the mylohyoid muscle may allow a portion of the sublingual gland to protrude into the submental spaceand permit the mucous-escapephe-
nomenon to occur.8 Ectopic sublingual glands9 or portions’O have also been reported below the mylohyoid. Animal studies have failed to support the theory that a ranula or mucocele could be caused by simple ligation of a salivary duct, but these could be produced following severance of the duct.4 Some workers concluded that human mucocele results from a traumatic defect of a salivary duct with release of saliva into the surrounding tissue,*’ Standish and ShaferI suggested also that partial salivary retention would allow smaller dilated ducts to rup-
360
Bronstein and Clark
ture and excrete saliva into the tissues. This might occur as a result of a sialolith producing distention and subsequent rupture.j3 since, with each occurrence of obstruction, more destruction and fibrosis occurs in the gland parenchyma.14 A lining consisting of granulation4 or fibrous6 tissue has been reported in cavities produced by the mucous-escapephenomenon. The increase in pressure and subsequent degeneration of the original epithelial lining are thought to be responsible for this altered lining4 Trauma, such as might be incurred during an incisional biopsy, can lead to persistent adenitis and a resultant fistula14and require gland removaLL4This was well illustrated in the present case following the initial surgery. Suturing of areas of duct injury so that an oral fistula develops is often the best treatment for a transected duct,5,‘4 and it is this same feature which led to development of the cutaneous fistula. An early correct diagnosis would have altered considerably the management and clinical course of this patient. The suspicion or recognition of a problem confined primarily to the sublingual gland would have suggestedtreatment for something other than a thyroglossal duct cyst. After the original biopsy, and certainly after the secondoperation, somethought of salivary origin should have been entertained. Bilateral sialography with aqueous contrast material to depict the altered glandular structure is necessaryto confirm the diagnosis and evaluate the other glands in the floor of the mouth. Although cannulation of the sublingual ducts is extremely difficult, if not impossible, the gland can be visualized if its major ducts are large and empty directly into the submandibular duct.14Filling of the sublingual duct may be enhanced if the cannula is not passedvery far into the submandibular duct but is allowed to fill the submandibular duct near its orifice.15This allows retrograde filling, but only on a sporadic basis.K” Subtraction techniques are also useful in visualizing the sublingual sialogram against the superimposed mandibular bone.“.‘* Computerized tomography has been reported for imaging the salivary glands, but it appears to be useful primarily for detecting massesin or near the gland.20Oil-base contract material should be injected into the glands for optimum visualization. Venography can be used at the same examination for visualization of the vessels in the area or of vascular tumors.‘9 Amylase and protein activity have also been utilized in the diagnosis of mucus-extravasation phenomena.’ Ranula contents are normally quite high in amylase and low in protein, as compared to serum.
Oral Surg. April, 1984
Histochemically, amylase activity has been detected only in the posterior portion of the sublingual gland.’ Treatment of the oral ranula or its deeper counterpart, the plunging ranula, is by excision of the sublingual gland.4*b,20 It appears to be unnecessaryto dissect cervical ramifications associated with the plunging ranula; these will disappear with removal of the secretory source.6 In the case reported here surgical intervention was also necessaryto eliminate the sialocele and fistula. Surgery, based on the pathogenesis of the lesion, can be seen logically to prevent recurrences4; at one time an external approach through a cervical incision was suggested.’ Treatment of sialocele must either restore oral flow by repair of the duct, if injured, or prevent or remove glandular secretory activity. This has been achieved by ligating the proximal end of the duct, administering anticholinergic drugs with daily aspiration of the cavity, radiotherapy, or ablation of the secretomotor supply via the chorda tympani in the middle ear.‘, ” Protection of the lingual nerve and artery and of the submandibular duct is necessary to reduce postoperative morbidity. In this patient, weaknessof the duct, possibly secondary to stasis dilatation, required catheterization which proved unsuccessful and the duct becamenonpatent. Apparently, there was retrograde pressure from the enlarged sublingual gland, causing ductal dilatation and stasis in the submandibular gland, as confirmed in the sialogram (Fig. 2); this, in turn, may have produced additional pressure forward into the sublingual gland. It has been demonstrated that complete duct ligation will Iead to gland atrophy” but that partial ligation will not.23 SUMMARY
A case of salivary fistula and sialocele of the submental area secondary to development of a plunging ranula is reported. The probable pathogenesis, the anatomic basis of development, and the surgical treatment have been discussed. REFERENCES I. Avery BS: A sialocele and unusual parotid tistula-a case report. Br J Oral Surg 18: 40-44, 1980. 2. Dierks EJ. Granite EL: Parotid sialocele and fistula after mandibular osteotomv. J Oral Surg 35: 299-300. 1977. 3. Rayne J: Plunging raiula-report:f a case. Br J Oral Surg 11: 139-142, 1973. 4. Catone GA, Merrill RG, Henny FA: Sublingual gland mucus escape phenomenon-treatment by excision of sublingual gland. J Oral Surg 27: 774-786, 1969. 5. Rankow RM, Polayes IM: Diseases of the salivary glands, Philadelphia, 1976, W. B. Saunders Company, p. 232.
Volume 57 Number 4 6. Roediger WEW, Kay S: Pathogenesis and treatment of plunging ranula. Surg Gynecol Obstet 144: 862-864, 1977. 7. Roediger WEW, Lloyd P, Lawson HH: Mucus extravasation theory as a cause of plunging ranulas. Br J Oral Surg. 60: 720-722, 1973. 8. Seward GR: Dermoid cysts of the floor of the mouth. Br J Oral Surg 3: 36-47, 1965. 9. Castello WA, Huelke DF, Celis A: Some basic anatomic features in paralingual space surgery. ORAL SURG 27: 613621, 1969.
10. Braun TW, Soteranos GC: Cervical ranula due to an ectopic sublingual gland. J Maxillofac Surg 10: 56-58, 1982. 11, Bhaskar SN, Bolden TE, Weinmann JP: Pathogenesis of mucoceles. J Dent Res 35: 863, 1956. 12. Standish SM. Shafer WG: Mucus retention uhenomenon. J Oral Surg 17: 15, 1959. 13. Chaudhry AP, et al: Clinical and experimental study of mucocele (retention cyst). J Dent Res 39: 1253, 1960. 14. Rankow RM, Polayes IM: Diseases of the salivary glands, Philadelphia, 1976, W.B. Saunders Company, pp. 233, 244, 309, 62-63. 15. Liliequist B, Welander U: Sialography of the sublingual gland. Acta Radio1 8: 187-192, 1969. 16. Wackens G, De Smedt E: Sublingual sialography. Acta Radio1 IO: 181-192, 1970.
Sublingual gland salivary fistula and sialocele 361 17. Corbett RH: Submandibular sialography: An unusual complication. Br J Radio1 18: 1032-1033, 1975. 18. Liliequist B, Welander LJ: Sialography: New application of the subtraction technique. Acta Radio1 7: 228-234, 1968. 19. Carter BL, Karmody CS, Blickman JR, Panders AK: Computed tomography and sialography. 1. Normal anatomy. J Comput Assist Tomogr 5: 42-45, 1981. 20. Rankow RM, Polayes IM: Diseases of the salivary glands, Philadelphia, 1976, W.B. Saunders Company, p. 162. 21. Moore KL: Clinically oriented anatomy, Baltimore, 1980, Williams & Wilkins Company, p. 1015. 22. Bhaskar SN, Bolden TE, Weinmann JP: Experimental obstructive adenitis in the mouse, J Dent Res 35: 852, 1956. 23. Patey DH: In Irvine WT (editor): Modern trends in surgery, ed. 3, London, 1971, Butterworth & Co., p. 261. Reprint requests to:
Dr. Sidney L. Bronstein Division of Oral and Maxillofacial Surgery School of Dentistry University of Colorado Health Science Center Campus Box C284 4200 East Ninth Ave. Denver, CO 80261
and Morris S. Clark, D.D.S., ** Denver, Colo.
OF COLORADO SCHOOL OF DENTISTRY
A case of cutaneous salivary fistula and sialocele in the submental area, arising from the sublingual gland, is reported and its etiology, development, and management are discussed. The entity of plunging ranula is compared to the lesion the case presented, and the literature regarding this lesion is reviewed. The successful surgical treatment of this case supports the theory that the sublingual gland is the source of the disorder. The effect of the altered salivary function on the adjacent submandibular gland and duct presented an operative complication. (ORAL SURG. 57:357-36 1, 1984.)
S
ubmental sublingual gland salivary fistula with sialocele occurs infrequently and has not been reported in the literature. Fistulas that develop following parotid surgery or injury are more numerous and better documented,’ and an involvement with sialocele formation has been described.‘,* A variety of lesions are common to the floor of the mouth and may be seen extraorally as swellings of the submental region. These include dental abscess, minor salivary gland mucocele, oral ranula, major salivary glarid adenitis or neoplasm, dermoid cyst, branchial cleft cyst, thyroglossal duct cyst, and plunging ranula. 3*4Rarely will any of these lesions fistulate unless traumatized.4 It is the purpose of this article to report a salivary fistula and sialocele which followed surgical trauma, to discuss the diagnosis and pathogenesis of this lesion ai a plunging ranula, and to describe its surgical manag,ement. CASE REPORT
In November, 1974, the patient, then a 6%year-old girl, underwent treatment of several primary teeth, including pulpotomies of two lower primary molars. About 7 to 10 days later a midline swelling appeared under the chin adjacent to the mandible. This swelling was occasionally painful and somewhat tender but never appeared to be inflammatory. Erythromycin was ineffective, but Ampicil-
*Associate Professor and Chairman, Division of Oral and Maxil-
lofacial Surgery,Schoolof Dentistry. **Assistant Professor,Division of Oral and Maxillofacial Surgery, School of Dentistry.
lin therapy was instituted and within a week the swelling decreasedbut never completely disappeared. Approximately 2 months later, on Jan. 19, 1975, her pediatrician wanted the lesion biopsied and, with a presumptive diagnosis of thyroglossal duct cyst, the child was admitted for surgery. The procedure was described as quite difficult since the tissue was not well formed and appeared necrotic. The microscopic diagnosis was reported as “necrotic and inflammatory granulation tissue.” Healing progressednormally for about 3 months, but then the incision openedand drained a clear, viscous, mucoid material. This would “stretch out like an elastic band,” according to the patient’s mother, and could be broken only with difficulty. The area did not appear infected at this time; recurrent swelling alternating with drainage occurred. A dental cause was investigated but was discarded. In July, 1976, the area was operated on again and findings similar to those of the original operation were reported. Within 2 weeksthe lesion reappearedand the thick mucoid drainage resumed. At this time the surgeons believed that the surgical intervention had been minimal, and in May, 1977, a classic thryoglossal duct cyst excision, which included the central portion of the hyoid bone, was performed. Microscopic diagnosis was “tissue consistent with thyroglossal duct cyst and chronic inflammation.” After a short time healing was interrupted by drainage exactly as before. After moving to Denver, Colorado, the patient was referred by the Otolaryngology Service of University Hospital and was seenin February and again in November of 1981. At this latter appointment the patient reported increased discomfort at mealtimes, culminating in increased mucous secretion from the submental fistula. There was equivocal intraoral tendernessonly over the left sublingual gland. Surgical removal of one or possibly both sublingual glands was contemplated and submandibular 357
358
Bronstein and Clark
Oral Surg.
April, I984
Fig. 1. Sialogram of right submandibular and sublingual glands. Seen are ectatic submandibular ducts (solid arrows) and prominent sublingual ducts (open arrows) leaving enlarged sublingual gland to enter submandibular duct.
sialography was scheduled. The left glands were reported as normal and, indeed, showed active reflux during fluorography; the right submandibular gland showed ductal dilatation, and the right sublingual gland was enlarged to two to three times its normal size and showed ductal dilatation also. The only laboratory tests that had ever been performed were an amylase test in 1977, which showed a very low level, and an acid-fast culture by the Otolaryngology Service in 1981, which was negative. With this extreme chronic history, evidence of increasing adenitis, continued drainage, and an abnormal sialogram, removal of the right sublingual gland and of the fistula was advised. On Jan. 15, 1982, with the patient under general anesthesia, following preservation of the submandibular duct by probing, an incision was made just lateral to the duct and the gland was excised along with two large dilated secondary ducts seento enter it from the submandibular duct; these enlarged ducts (Fig. 1) were ligated prior to excision. Blunt dissection with peanut spongeswas used mainly to protect the duct and the lingual nerve. The gland was noted to be approximately three to four times normal size. A portion of the gland was closely connected to the anterior part of the mylohyoid muscle, but no definite extension could be identified below the muscle. After thorough exploration the lingual nerve was noted to be intact, but there was a tear in the submandibular duct at its exit from the gland. The duct was cannulated with a length of 18-gauge polyethylene tubing and was fixed in place by sutures to provide luminal supports and patency. A good flow of clear saliva was obtained. The oral incision was closed in normal fashion, and the cutaneous fistula was excised after preparation and redraping of the submental area. No tract upward into the floor of the mouth could be located, but a well-defined
scarred mass with a central, saccular outpouching was identified and removed after suture ligation of its deep attachment. The tissues were closed in layers, the wound was dressed,and the procedure was terminated. Specimensof gland, duct, and fistula were submitted for histopathologic examination and were reported as ectatic gland (Fig. 2) normal duct, and “focally ulcerated squamous epithelium the saccular outpouching is distally lined by columnar epithelial cells. Proximally the outpouching is ulcerated and lined by inflamed, hemorrhagic granulation tissue” (Fig. 3). Postoperatively, maintenance of the submandibular gland was quite difficult, as the tubing was continually displaced from the gland and could be replaced only with difficulty. However, clear saliva could be obtained when the tubing was correctly situated. Ten days postsurgically the tubing had to be removed but normal drainage could be noted from what appeared to be an oral fistula. Unfortunately, this fistula did not develop and the patient reported increasing discomfort in the right submandibular area at mealtimes, although the symptoms were gone within 20 minutes after meals. These symptoms gradually decreased in severity and by 2 months postoperatively discomfort was noted only upon stimulation with sweet and sour foods. In May, 1982, the patient returned with right submandibular swelling, not associatedwith eating. This was very tender, of sudden onset, and resolved completely within 4 days with penicillin therapy. Only minor episodesof diminishing submandibular adenitis recurred for a time and then ceased. The submental area healed normally, and no drainage ever occurred postoperatively. At examination on July 25, 1983, the patient was completely asymptomatic and was enjoying a normal diet. Plastic repair of the submental scar is anticipated. Lingual nerve sensation, decreased in the first few months postoperatively, was normal. DISCUSSION
The management of this case was complicated by the history of dental treatment, with a submental swelling appearing within a short interval afterward; however, the nature and location of the swelling were not typical of a dental infection. It appeared to be, according to the patient’s mother, like a “sebaceous cyst,” close to the skin, without signs of inflammation. However, it did respond to Ampicillin, so infection was present and the dental treatment may have contributed to an alteration of sublingual gland structure or function, promoting formation of the
sialocele and fistula. During the more than 2 months from the decrease of the swelling to the initial surgery, the simple extravasation and accompanying mucus-escape phenomenon developed. The plunging ranula, also known as an oral ranula
with a cervical extension, it formed by extravasation of salivary secretion, usually from the sublingual gland.6 Differentiation
has been made between the
Sublingual gland salivary jistula and sialocele 359
Volume 57 Number 4
Fig.
2. High-power photomicrograph showing thinning of ductal epithelium.
Fig. 3. Low-power photomicrograph of fistulous opening with saccular structure lined with ductal epithelium.
oral ranula as being an extravasation cyst from the
sublingual gland generally and the plunging ranula as being formed from the posterior portion only of this gland. L Neither of these lesions is now considered as a cyst because no epithelial lining can be found; rather, they are considered extravasation phenomena.) It has been postulated that an aberrant sublingual duct may allow stasis in the deep lobe of the sublingual gland with obstruction and extravasation.6 Hiatuses in the mylohyoid muscle may allow a portion of the sublingual gland to protrude into the submental spaceand permit the mucous-escapephe-
nomenon to occur.8 Ectopic sublingual glands9 or portions’O have also been reported below the mylohyoid. Animal studies have failed to support the theory that a ranula or mucocele could be caused by simple ligation of a salivary duct, but these could be produced following severance of the duct.4 Some workers concluded that human mucocele results from a traumatic defect of a salivary duct with release of saliva into the surrounding tissue,*’ Standish and ShaferI suggested also that partial salivary retention would allow smaller dilated ducts to rup-
360
Bronstein and Clark
ture and excrete saliva into the tissues. This might occur as a result of a sialolith producing distention and subsequent rupture.j3 since, with each occurrence of obstruction, more destruction and fibrosis occurs in the gland parenchyma.14 A lining consisting of granulation4 or fibrous6 tissue has been reported in cavities produced by the mucous-escapephenomenon. The increase in pressure and subsequent degeneration of the original epithelial lining are thought to be responsible for this altered lining4 Trauma, such as might be incurred during an incisional biopsy, can lead to persistent adenitis and a resultant fistula14and require gland removaLL4This was well illustrated in the present case following the initial surgery. Suturing of areas of duct injury so that an oral fistula develops is often the best treatment for a transected duct,5,‘4 and it is this same feature which led to development of the cutaneous fistula. An early correct diagnosis would have altered considerably the management and clinical course of this patient. The suspicion or recognition of a problem confined primarily to the sublingual gland would have suggestedtreatment for something other than a thyroglossal duct cyst. After the original biopsy, and certainly after the secondoperation, somethought of salivary origin should have been entertained. Bilateral sialography with aqueous contrast material to depict the altered glandular structure is necessaryto confirm the diagnosis and evaluate the other glands in the floor of the mouth. Although cannulation of the sublingual ducts is extremely difficult, if not impossible, the gland can be visualized if its major ducts are large and empty directly into the submandibular duct.14Filling of the sublingual duct may be enhanced if the cannula is not passedvery far into the submandibular duct but is allowed to fill the submandibular duct near its orifice.15This allows retrograde filling, but only on a sporadic basis.K” Subtraction techniques are also useful in visualizing the sublingual sialogram against the superimposed mandibular bone.“.‘* Computerized tomography has been reported for imaging the salivary glands, but it appears to be useful primarily for detecting massesin or near the gland.20Oil-base contract material should be injected into the glands for optimum visualization. Venography can be used at the same examination for visualization of the vessels in the area or of vascular tumors.‘9 Amylase and protein activity have also been utilized in the diagnosis of mucus-extravasation phenomena.’ Ranula contents are normally quite high in amylase and low in protein, as compared to serum.
Oral Surg. April, 1984
Histochemically, amylase activity has been detected only in the posterior portion of the sublingual gland.’ Treatment of the oral ranula or its deeper counterpart, the plunging ranula, is by excision of the sublingual gland.4*b,20 It appears to be unnecessaryto dissect cervical ramifications associated with the plunging ranula; these will disappear with removal of the secretory source.6 In the case reported here surgical intervention was also necessaryto eliminate the sialocele and fistula. Surgery, based on the pathogenesis of the lesion, can be seen logically to prevent recurrences4; at one time an external approach through a cervical incision was suggested.’ Treatment of sialocele must either restore oral flow by repair of the duct, if injured, or prevent or remove glandular secretory activity. This has been achieved by ligating the proximal end of the duct, administering anticholinergic drugs with daily aspiration of the cavity, radiotherapy, or ablation of the secretomotor supply via the chorda tympani in the middle ear.‘, ” Protection of the lingual nerve and artery and of the submandibular duct is necessary to reduce postoperative morbidity. In this patient, weaknessof the duct, possibly secondary to stasis dilatation, required catheterization which proved unsuccessful and the duct becamenonpatent. Apparently, there was retrograde pressure from the enlarged sublingual gland, causing ductal dilatation and stasis in the submandibular gland, as confirmed in the sialogram (Fig. 2); this, in turn, may have produced additional pressure forward into the sublingual gland. It has been demonstrated that complete duct ligation will Iead to gland atrophy” but that partial ligation will not.23 SUMMARY
A case of salivary fistula and sialocele of the submental area secondary to development of a plunging ranula is reported. The probable pathogenesis, the anatomic basis of development, and the surgical treatment have been discussed. REFERENCES I. Avery BS: A sialocele and unusual parotid tistula-a case report. Br J Oral Surg 18: 40-44, 1980. 2. Dierks EJ. Granite EL: Parotid sialocele and fistula after mandibular osteotomv. J Oral Surg 35: 299-300. 1977. 3. Rayne J: Plunging raiula-report:f a case. Br J Oral Surg 11: 139-142, 1973. 4. Catone GA, Merrill RG, Henny FA: Sublingual gland mucus escape phenomenon-treatment by excision of sublingual gland. J Oral Surg 27: 774-786, 1969. 5. Rankow RM, Polayes IM: Diseases of the salivary glands, Philadelphia, 1976, W. B. Saunders Company, p. 232.
Volume 57 Number 4 6. Roediger WEW, Kay S: Pathogenesis and treatment of plunging ranula. Surg Gynecol Obstet 144: 862-864, 1977. 7. Roediger WEW, Lloyd P, Lawson HH: Mucus extravasation theory as a cause of plunging ranulas. Br J Oral Surg. 60: 720-722, 1973. 8. Seward GR: Dermoid cysts of the floor of the mouth. Br J Oral Surg 3: 36-47, 1965. 9. Castello WA, Huelke DF, Celis A: Some basic anatomic features in paralingual space surgery. ORAL SURG 27: 613621, 1969.
10. Braun TW, Soteranos GC: Cervical ranula due to an ectopic sublingual gland. J Maxillofac Surg 10: 56-58, 1982. 11, Bhaskar SN, Bolden TE, Weinmann JP: Pathogenesis of mucoceles. J Dent Res 35: 863, 1956. 12. Standish SM. Shafer WG: Mucus retention uhenomenon. J Oral Surg 17: 15, 1959. 13. Chaudhry AP, et al: Clinical and experimental study of mucocele (retention cyst). J Dent Res 39: 1253, 1960. 14. Rankow RM, Polayes IM: Diseases of the salivary glands, Philadelphia, 1976, W.B. Saunders Company, pp. 233, 244, 309, 62-63. 15. Liliequist B, Welander U: Sialography of the sublingual gland. Acta Radio1 8: 187-192, 1969. 16. Wackens G, De Smedt E: Sublingual sialography. Acta Radio1 IO: 181-192, 1970.
Sublingual gland salivary fistula and sialocele 361 17. Corbett RH: Submandibular sialography: An unusual complication. Br J Radio1 18: 1032-1033, 1975. 18. Liliequist B, Welander LJ: Sialography: New application of the subtraction technique. Acta Radio1 7: 228-234, 1968. 19. Carter BL, Karmody CS, Blickman JR, Panders AK: Computed tomography and sialography. 1. Normal anatomy. J Comput Assist Tomogr 5: 42-45, 1981. 20. Rankow RM, Polayes IM: Diseases of the salivary glands, Philadelphia, 1976, W.B. Saunders Company, p. 162. 21. Moore KL: Clinically oriented anatomy, Baltimore, 1980, Williams & Wilkins Company, p. 1015. 22. Bhaskar SN, Bolden TE, Weinmann JP: Experimental obstructive adenitis in the mouse, J Dent Res 35: 852, 1956. 23. Patey DH: In Irvine WT (editor): Modern trends in surgery, ed. 3, London, 1971, Butterworth & Co., p. 261. Reprint requests to:
Dr. Sidney L. Bronstein Division of Oral and Maxillofacial Surgery School of Dentistry University of Colorado Health Science Center Campus Box C284 4200 East Ninth Ave. Denver, CO 80261