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Unusual case of a sialolith: a case report
Vol. 117 No. 1 January 2014
Unusual case of a sialolith: a case report Tyler T. Boynton, DMD,a and Stuart E. Lieblich, DMD,b Farmington, CR; Boston, MA; and Avon, CT University of Connecticut and Tufts University
Sialoliths are a common cause of salivary gland obstruction, usually affecting the submandibular gland. Although many theories are postulated for their formation (dehydration, medications that reduce salivary flow, increased salivary viscosity), no one aspect is clearly the primary etiology. This case report details an unusual case in which a facial hair of the patient apparently became entrapped in Wharton’s duct, causing a nidus for the formation of a sialolith. (Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:e9-e10)
Sialoliths are calcium-rich crystallized minerals that can occur in any of the salivary glands and are the most common cause of obstructive sialadenitis. They may be single or multiple but a majority of sialoliths occur as a single entity with no preference toward the left or right side.1 They are mainly found within the submandibular gland (80%-90%), to a lesser extent within the parotid gland (5%-20%), and rarely in the sublingual and minor salivary glands.1 This higher incidence in the submandibular gland is caused by a number of reasons relating to the anatomy and course of Wharton’s duct as well as the salivary composition. Anatomically, the duct is narrower in diameter and follows a longer and more tortuous course than Stenson’s duct. Specifically, there are two areas where the duct makes a sharp turn: the first is around the posterior edge of the mylohyoid muscle and the second occurs before the duct empties into the oral cavity. These particular areas are therefore more prone to salivary stone formation. Other contributing factors are that the saliva from the submandibular gland is twice as viscous as saliva from the parotid gland and also has a higher pH and calcium content.2 The etiology of sialoliths is not definitively proven, but salivary viscosity and salivary stasis contribute to stone formation. These can be caused by a change in morphology of the duct/gland as well as a change in salivary composition and flow rate caused by systemic factors such as dehydration of the patient and the antisialalog effects of certain medications (antihistamines, antipsychotic medications). A popular theory is that sialoliths develop from a
Resident, Division of Oral and Maxillofacial Surgery, University of Connecticut, Farmington, CT. b Associate Clinical Professor, University of Connecticut, Farmington, Connecticut; Visiting Assistant Professor, Tufts University, Boston, MA; and Private Practice, Avon Oral and Maxillofacial Surgery, Avon, CT. Received for publication Mar 7, 2012; accepted for publication Mar 10, 2012. © 2014 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter http://dx.doi.org/10.1016/j.oooo.2012.03.020
organic material such as inspissated mucous, ductal epithelial cells, salivary proteins, and foreign bodies that serve as a scaffold for the deposition of inorganic material (calcium salts).3 It is unlikely that stone formation occurs solely because of a high calcium concentration, but rather when a nidus serves as the starting point for apatite crystal formation. The following case represents the first mention in the literature of a facial hair serving as a nidus for sialolith formation.
CASE REPORT The patient is a 66-year-old male who noted discomfort and swelling under the left side of his jaw. He reported its presence for a 6-day duration and the swelling and pain was noted to be increased at mealtime. On clinical examination there was induration and tenderness of the left submandibular gland (Figure 1). The distal end of Wharton’s duct was noted to be inflamed and there was a notable submucosal mass consistent with a sialolith. No saliva could be expressed on massage of the gland. An uncomplicated transoral removal of the sialolith was achieved with local anesthesia. Of note was that on removal of the sialolith there was the presence of a hair, consistent with his facial hair, surrounded by the sialolith (Figure 2). On discussion with the patient he reported occasional facial hairs getting caught in his mouth.
DISCUSSION Sialolithiasis is the most common nonneoplastic disease of the salivary glands and autopsy reports show a 1% incidence across the population.4 Sialoliths occur most frequently in adults during the 4th, 5th, and 6th decades but can occur at any age. Diagnosis of sialoliths is both clinical and radiographic. Patients generally develop symptoms when the sialolith begins to obstruct salivary flow, leading to swelling and eventually pain that occurs before or during meals. Stasis of saliva can lead to retrograde flow of bacteria (usually staphylococcal organisms such as Staphylococcus aureus) into the parenchyma of the gland, in which the patient may present with purulence from the duct, leukocytosis, and fever. Submandibular stones may be felt in the floor of the mouth through e9
ORAL AND MAXILLOFACIAL SURGERY e10 Boynton and Lieblich
Fig. 1. Clinical examination revealed induration and tenderness of the left submandibular gland.
OOOO January 2014
ered in cases of small stones. This includes application of warm compresses, massaging the gland, using sialologs to increase salivary flow, ensuring that the patient is well hydrated, and prescribing antistaphylococcal antibiotics.6 Large sialoliths can be removed surgically either through a transoral approach or through a transfacial approach, which includes removal of the submandibular gland. In cases where a stone can be visualized on the floor of the mouth, an intraoral surgical approach is recommended. If the stone is located within the horizontal portion of the duct, the stone can be retrieved through a small mucosal incision and the duct can be left to marsupialize. Transfacial sialadenectomy is the preferred treatment when stones are located close to the hilum or in patients with persistent sialadenitis that is refractory to other methods of treatment.
CONCLUSIONS Although there is no consensus in the literature as to what causes sialoliths to form, it is generally agreed that a nidus is required. There are only a few cases reported in the literature in which a foreign body is suspected to have acted as a nidus for sialolith formation.7–9 However, this is the first known case report of a facial hair causing the initiation of stone formation. REFERENCES
Fig. 2. Sialolith surrounding a hair consistent with the patient’s facial hair.
bimanual palpation and plain-film radiographs can confirm their presence. In a majority of cases, the sialolith is located distal to the posterior edge of the mylohyoid muscle and can be easily visualized using intraoral occlusal films. In a smaller number of cases, the sialolith is located below the mylohyoid or at the hilum of the gland in which extraoral lateral oblique or panoramic films can be diagnostic. Plain films are usually sufficient to make a correct diagnosis because stones in the submandibular gland are radiopaque more than 85% of the time.5 Furthermore, conventional or cone beam computed tomography can be helpful in determining the exact location of the stone, especially if the practitioner cannot visualize or palpate the stone. The treatment of sialoliths is determined by the size and location of the stone as well as the history of the patient’s symptoms. Conservative management should be consid-
1. Lustmann J, Regev E, Melamed RE. Sialothiasis: a survey on 245 patients and a review of the literature. Int J Oral Maxillofac Surg 1990;19:135-38. 2. Baurmash HD. Submandibular salivary stones: current management modalities. J Oral Maxillofac Surg 2004;62:369-78. 3. Carr SJ. Sialolith of unusual size and configuration. Report of a case. Oral Surg Oral Med Oral Pathol 1965;20:709-12. 4. Rauch S, Gorlin RJ. Diseases of the salivary glands. In: Gorlin RJ, Goldman HM, editors. Thomas’ oral pathology, 6th ed. St. Louis: C. V. Mosby; 1970. p. 997-1003. 5. Fonseca RJ, Marciani RD, Turvey TA, Carlson ER, Braun TW. Oral and maxillofacial surgery. St. Louis: Saunders Elsevier; 2009. 543 pp. 6. Williams MF. Sialolithiasis. Otolaryngol Clin North Am 1999;32: 819-34. 7. Takeshita H, Ishihara A, Yamashita T, Itoh A, Yoshida K, Fukaya M. A case of a salivary calculus containing a limb of a shrimp: the structural analysis. Aichi-Gakuin Dent Sci 1990;3:49-58. 8. Watkins RM. Submandibular salivary duct calculus secondary to a foreign body. Br J Surg 1982;69:379-. 9. Sato K, Umeno H. Clinical photography. Fish bone–induced sialolith. Otolaryngol Head Neck Surg 2009;141:539-40. Reprint requests: Stuart E. Lieblich Avon Oral and Maxillofacial Surgery 34 Dale Road, Suite 105 Avon, Connecticut 06001 [email protected]
Unusual case of a sialolith: a case report Tyler T. Boynton, DMD,a and Stuart E. Lieblich, DMD,b Farmington, CR; Boston, MA; and Avon, CT University of Connecticut and Tufts University
Sialoliths are a common cause of salivary gland obstruction, usually affecting the submandibular gland. Although many theories are postulated for their formation (dehydration, medications that reduce salivary flow, increased salivary viscosity), no one aspect is clearly the primary etiology. This case report details an unusual case in which a facial hair of the patient apparently became entrapped in Wharton’s duct, causing a nidus for the formation of a sialolith. (Oral Surg Oral Med Oral Pathol Oral Radiol 2014;117:e9-e10)
Sialoliths are calcium-rich crystallized minerals that can occur in any of the salivary glands and are the most common cause of obstructive sialadenitis. They may be single or multiple but a majority of sialoliths occur as a single entity with no preference toward the left or right side.1 They are mainly found within the submandibular gland (80%-90%), to a lesser extent within the parotid gland (5%-20%), and rarely in the sublingual and minor salivary glands.1 This higher incidence in the submandibular gland is caused by a number of reasons relating to the anatomy and course of Wharton’s duct as well as the salivary composition. Anatomically, the duct is narrower in diameter and follows a longer and more tortuous course than Stenson’s duct. Specifically, there are two areas where the duct makes a sharp turn: the first is around the posterior edge of the mylohyoid muscle and the second occurs before the duct empties into the oral cavity. These particular areas are therefore more prone to salivary stone formation. Other contributing factors are that the saliva from the submandibular gland is twice as viscous as saliva from the parotid gland and also has a higher pH and calcium content.2 The etiology of sialoliths is not definitively proven, but salivary viscosity and salivary stasis contribute to stone formation. These can be caused by a change in morphology of the duct/gland as well as a change in salivary composition and flow rate caused by systemic factors such as dehydration of the patient and the antisialalog effects of certain medications (antihistamines, antipsychotic medications). A popular theory is that sialoliths develop from a
Resident, Division of Oral and Maxillofacial Surgery, University of Connecticut, Farmington, CT. b Associate Clinical Professor, University of Connecticut, Farmington, Connecticut; Visiting Assistant Professor, Tufts University, Boston, MA; and Private Practice, Avon Oral and Maxillofacial Surgery, Avon, CT. Received for publication Mar 7, 2012; accepted for publication Mar 10, 2012. © 2014 Elsevier Inc. All rights reserved. 2212-4403/$ - see front matter http://dx.doi.org/10.1016/j.oooo.2012.03.020
organic material such as inspissated mucous, ductal epithelial cells, salivary proteins, and foreign bodies that serve as a scaffold for the deposition of inorganic material (calcium salts).3 It is unlikely that stone formation occurs solely because of a high calcium concentration, but rather when a nidus serves as the starting point for apatite crystal formation. The following case represents the first mention in the literature of a facial hair serving as a nidus for sialolith formation.
CASE REPORT The patient is a 66-year-old male who noted discomfort and swelling under the left side of his jaw. He reported its presence for a 6-day duration and the swelling and pain was noted to be increased at mealtime. On clinical examination there was induration and tenderness of the left submandibular gland (Figure 1). The distal end of Wharton’s duct was noted to be inflamed and there was a notable submucosal mass consistent with a sialolith. No saliva could be expressed on massage of the gland. An uncomplicated transoral removal of the sialolith was achieved with local anesthesia. Of note was that on removal of the sialolith there was the presence of a hair, consistent with his facial hair, surrounded by the sialolith (Figure 2). On discussion with the patient he reported occasional facial hairs getting caught in his mouth.
DISCUSSION Sialolithiasis is the most common nonneoplastic disease of the salivary glands and autopsy reports show a 1% incidence across the population.4 Sialoliths occur most frequently in adults during the 4th, 5th, and 6th decades but can occur at any age. Diagnosis of sialoliths is both clinical and radiographic. Patients generally develop symptoms when the sialolith begins to obstruct salivary flow, leading to swelling and eventually pain that occurs before or during meals. Stasis of saliva can lead to retrograde flow of bacteria (usually staphylococcal organisms such as Staphylococcus aureus) into the parenchyma of the gland, in which the patient may present with purulence from the duct, leukocytosis, and fever. Submandibular stones may be felt in the floor of the mouth through e9
ORAL AND MAXILLOFACIAL SURGERY e10 Boynton and Lieblich
Fig. 1. Clinical examination revealed induration and tenderness of the left submandibular gland.
OOOO January 2014
ered in cases of small stones. This includes application of warm compresses, massaging the gland, using sialologs to increase salivary flow, ensuring that the patient is well hydrated, and prescribing antistaphylococcal antibiotics.6 Large sialoliths can be removed surgically either through a transoral approach or through a transfacial approach, which includes removal of the submandibular gland. In cases where a stone can be visualized on the floor of the mouth, an intraoral surgical approach is recommended. If the stone is located within the horizontal portion of the duct, the stone can be retrieved through a small mucosal incision and the duct can be left to marsupialize. Transfacial sialadenectomy is the preferred treatment when stones are located close to the hilum or in patients with persistent sialadenitis that is refractory to other methods of treatment.
CONCLUSIONS Although there is no consensus in the literature as to what causes sialoliths to form, it is generally agreed that a nidus is required. There are only a few cases reported in the literature in which a foreign body is suspected to have acted as a nidus for sialolith formation.7–9 However, this is the first known case report of a facial hair causing the initiation of stone formation. REFERENCES
Fig. 2. Sialolith surrounding a hair consistent with the patient’s facial hair.
bimanual palpation and plain-film radiographs can confirm their presence. In a majority of cases, the sialolith is located distal to the posterior edge of the mylohyoid muscle and can be easily visualized using intraoral occlusal films. In a smaller number of cases, the sialolith is located below the mylohyoid or at the hilum of the gland in which extraoral lateral oblique or panoramic films can be diagnostic. Plain films are usually sufficient to make a correct diagnosis because stones in the submandibular gland are radiopaque more than 85% of the time.5 Furthermore, conventional or cone beam computed tomography can be helpful in determining the exact location of the stone, especially if the practitioner cannot visualize or palpate the stone. The treatment of sialoliths is determined by the size and location of the stone as well as the history of the patient’s symptoms. Conservative management should be consid-
1. Lustmann J, Regev E, Melamed RE. Sialothiasis: a survey on 245 patients and a review of the literature. Int J Oral Maxillofac Surg 1990;19:135-38. 2. Baurmash HD. Submandibular salivary stones: current management modalities. J Oral Maxillofac Surg 2004;62:369-78. 3. Carr SJ. Sialolith of unusual size and configuration. Report of a case. Oral Surg Oral Med Oral Pathol 1965;20:709-12. 4. Rauch S, Gorlin RJ. Diseases of the salivary glands. In: Gorlin RJ, Goldman HM, editors. Thomas’ oral pathology, 6th ed. St. Louis: C. V. Mosby; 1970. p. 997-1003. 5. Fonseca RJ, Marciani RD, Turvey TA, Carlson ER, Braun TW. Oral and maxillofacial surgery. St. Louis: Saunders Elsevier; 2009. 543 pp. 6. Williams MF. Sialolithiasis. Otolaryngol Clin North Am 1999;32: 819-34. 7. Takeshita H, Ishihara A, Yamashita T, Itoh A, Yoshida K, Fukaya M. A case of a salivary calculus containing a limb of a shrimp: the structural analysis. Aichi-Gakuin Dent Sci 1990;3:49-58. 8. Watkins RM. Submandibular salivary duct calculus secondary to a foreign body. Br J Surg 1982;69:379-. 9. Sato K, Umeno H. Clinical photography. Fish bone–induced sialolith. Otolaryngol Head Neck Surg 2009;141:539-40. Reprint requests: Stuart E. Lieblich Avon Oral and Maxillofacial Surgery 34 Dale Road, Suite 105 Avon, Connecticut 06001 [email protected]