Bull Tokyo Dent Coll (2018) 59(1): 53–58
Case Report doi:10.2209/tdcpublication.2017-0013
Difficulties in Diagnosis of Sialolithiasis: A Case Series
Svitlana Veniaminivna Kolomiiets, Kristina Oleksandrivna Udaltsova, Tetiana Andriivna Khmil, Alina Mykolaiivna Yelinska, Olena Anatoliivna Pisarenko and Viktoriya Ihorivna Shynkevych Department of Postgraduate Education for Dentists, Higher State Educational Establishment of Ukraine “Ukrainian Medical Stomatological Academy”, Poltava, Ukraine
Received 13 April, 2017/Accepted for publication 8 July, 2017
Abstract Sialolithiasis is one of the most common and extensively obstructive disorders of the major salivary glands. Here, we report 3 cases of sialolithiasis in the submandibular sali- vary gland showing symptomatic similarities to other dental and non-dental disorders of the maxillofacial area. How the various clinical features of this condition and findings on 3D-CT may lead to a misdiagnosis are also discussed. In the first case, that of a 45-year-old woman, a final diagnosis of a non-radiopaque submandibular sialolith allowed the ini- tially indicated surgical extraction of a malerupted and semi-impacted right mandibular 3rd molar to be abandoned. In the second case, that of a 57-year-old woman, radiographic findings had previously led to a diagnosis of ameloblastoma, which had masked the pres- ence of sialolithiasis for at least 9 years, despite the radiopacity of the sialolith. Meanwhile, exacerbation of sialolithiasis was mistaken for lymphadenitis. In the third case, that of a 40-year-old woman, sialolithiasis was diagnosed in a timely manner, despite the fact that the dentists’ attention had initially been focused on odontopathological symptoms. One feature of the present report is the concurrence of dental and non-dental pathologies affecting the same sextant as the sialolithiasis. Despite recent advances in imaging tech- nology and diagnostics, cases of sialolithiasis being misdiagnosed continue to occur in clinical practice. Key words: Diagnosis — Sialolithiasis — Submandibular salivary gland
Introduction per 20,000 (individuals)8). The incidence rate of sialolithiasis has been reported to be Sialolithiasis is one of the most prevalent between 7.27 and 14.10 per one hundred and extensively obstructive disorders of the thousand (person-years), with sex showing no major salivary glands. In one recent study, it significant effect10). was estimated that the annual symptomatic Most sialoliths develop in the submandibu- incidence of sialoliths was 1 per 10,000 and 1 lar salivary gland or its ducts (80 to 92%) and
53 54 Kolomiiets SV et al. can cause acute and chronic inflammation7). How the various clinical features of this condi- The predominant prevalence of sialoliths in tion and findings on 3D-CT may lead to a the submandibular salivary glands is due to misdiagnosis are also discussed. The authors various anatomical and chemical factors, such obtained written informed consent from the as differences in the composition of the saliva. patients concerned to present materials from Patients most often present with a tingling their case histories in this paper in compli- sensation and swelling of the glands after ance with the ethical approval protocol of this meals. The course of the disease is character- institute. ized by relapse and remission until final definitive treatment is required, usually in the form of a surgical intervention. Case Presentations Despite the fact that the patient will gener- ally require hospitalization for such treat- 1. Case no.1 ment, the diagnosis itself is usually made by a This patient was a healthy 45-year-old dentist working in a private or municipal set- woman attending our clinic with a diagnosis ting depending on the type of facilities avail- of a malerupted and semi-impacted right able in the post-Soviet area in which they live. mandibular 3rd molar. She had been com- A number of tools and research methods plaining of non-localized recurrent pain in are commonly used to diagnose sialolithiasis. the distal area of the right mandible for Both panoramic and occlusal radiographic approximately 2 months. Swelling in the right techniques have been demonstrated to dis- submandibular area had caused asymmetry of play satisfactory diagnostic screening perfor- the face. After visiting her local dentist, the mance6). Approximately 20% of sialoliths are patient had been referred to our hospital for poorly calcified, and therefore not visible on an orthopantomogram (OPG) (Fig. 1) and 2D radiographs5). Moreover, the deposition of then to a dental surgeon for extraction of the a radiopaque sialolith on the body of the man- right mandibular 3rd molar. dible may render the former barely visible A physical examination revealed that the radiographically. right submandibular lymph node was High-frequency ultrasound examination enlarged to approximately 0.5 cm in diameter. 【版面】W：396 pt（片段 192 pt） H：588 pt 【本文】行数不明（手組み） 10pt 12pt 送り (US) has been reported【図】●図番号・タイトル・説明：11.3Q 12.7H New to be effective in con- It was slightly Baskerville painful ITC onStd 図タイトルと説明のアキ 9Q ●タイトル折り返し：番号の後（続 palpation and was く説明の先頭は字下げ不要） ●図説の幅 片段：片段固定 全段：図幅 1) firming a diagnosis of【表】●番号・タイトル・説明：11.3Q 12.7H New sialolithiasis . Recon- Baskerville ITC Std タイトルと表のアキ 10.5Q ●罫線 表はじめのみ双罫 表中の structed conventional CT罫の太さ 1.411mm and volume render●表中：11.3Q 12.7H New- Baskerville ITC Std ●脚注 11.3Q 12.7H New Baskerville ITC Std 字下げなし ing or 3D computed radiography斜体は New Baskerville (3D-CT) ITC Std Italic（タグは
ing to the right ear and swelling in the same submandibular area. These symptoms had arisen the previous day. The patient felt dis- comfort in the root of the tongue on the right when swallowing. Anamnesis revealed submandibular lymph- adenitis, which had been treated with partial success 6 months earlier. She mentioned that Fig. 2 Patient no.1: Fragments of 3D-CT scan of showed clearly visible sialolith when not super- the right mandibular 1st and 2nd molars had imposed on mandible been extracted after unsuccessful conserva- tive treatment approximately 32 years earlier. She also told us that she had been given a diagnosis of a suppurated ameloblastoma of the right mandibular ramus at that time, and movable. that this has been surgically treated with Intraoral examination revealed that the curettage and simultaneous extraction of the right mandibular 3rd molar was covered with right 3rd mandibular molar. Since then, the pale-pink oral mucosa. Palpation of the retro- patient had periodically felt the taste of pus, molar trigone and transitory fold elicited no which resulted in frequent visits to her pain response. Therefore, removal of the dentist. right mandibular 3rd molar was not indicated. Examination of the right submandibular In contrast to the left one, palpation of the area revealed a dense, non-elastic swelling right sublingual fold caused discomfort. The 2 cm in diameter, which was mildly painful, right Wharton’s duct was smooth and had an slightly movable, and was not connected with intermittent structure. Only poor secretion the surrounding tissue. This swelling was cov- was observed from the right sublingual car- ered in healthy epithelium. Intraoral palpa- uncle. Taken together, this led us to suspect tion of the right sublingual fold caused dis- dysfunction of the salivary gland, despite the comfort, and no secretion was released from absence of any sign of a sialolith on OPG. the right paramedian sublingual caruncle. Subsequently, US of the submandibular The patient had also undergone dental gland was performed together with 3D-CT of OPG 4 times within the previous 9 years, the mandible, with the latter revealing a sialo- revealing multilocular radiolucent lesions of lith (Fig. 2). The US allowed us to determine the right mandibular ramus corresponding to the size of the sialolith, its localization in an ameloblastoma, and clearly visible radi- Wharton’s duct, and the isoechogenic struc- opaque calculus, which was superimposed on ture of the glandular parenchyma. the mandible, but undiagnosed. The patient was then referred to maxillofa- The preliminary diagnosis was sialolithiasis cial surgery, where she successfully underwent in the right submandibular salivary glands, intraoral surgical removal of the sialolith. At a which was confirmed by 3D-CT (Fig. 3). High follow-up examination at 7 days postopera- frequency ultrasound revealed a sialolith in tively, the patient presented with a complete the parenchyma of the glands. resolution of symptoms. The prognosis in Since ameloblastoma is a painless disor- regard to sialolithiasis is favorable, due to the der3), it was determined that the sialolithiasis low recurrence rate7). was responsible for the patient’s reported discomfort over the years. The patient was 2. Case no.2 referred to maxillofacial surgery for gland This patient was a healthy 57-year-old removal, but surgical intervention was post- woman who attended the university dental poned due to increased total bilirubin in her clinic with the chief complaint of pain radiat- blood. Radical resection of the jaw ramus was 【版面】W：396 pt（片段 192 pt） H：588 pt 【本文】行数不明（手組み） 10pt 12pt【版面】W：396 pt（片段 192 送り pt） H：588 pt 【本文】行数不明（手組み） 10pt 12pt 送り 【図】●図番号・タイトル・説明：11.3Q 12.7H New Baskerville ITC 【図】●図番号・タイトル・説明：11.3Q 12.7H NewStd 図タイトルと説明のアキ 9Q ●タイトル折り返し：番号の後（続 Baskerville ITC Std 図タイトルと説明のアキ 9Q ●タイトル折り返し：番号の後（続 く説明の先頭は字下げ不要） ●図説の幅 片段：片段固定 全段：図幅 く説明の先頭は字下げ不要） ●図説の幅 片段：片段固定 全段：図幅 【表】●番号・タイトル・説明：11.3Q 12.7H New Baskerville ITC Std タイトルと表のアキ 10.5Q ●罫線 表はじめのみ双罫 表中の【表】●番号・タイトル・説明：11.3Q 12.7H New Baskerville ITC Std タイトルと表のアキ 10.5Q ●罫線 表はじめのみ双罫 表中の 罫の太さ 1.411mm ●表中：11.3Q 12.7H New Baskerville ITC Std ●脚注 11.3Q 12.7H New罫の太さ 1.411mm ●表中：11.3Q 12.7H New Baskerville ITC Std Baskerville 字下げなし ITC Std ●脚注 11.3Q 12.7H New Baskerville ITC Std 字下げなし 斜体は New Baskerville ITC Std Italic（タグは56
Fig. 4 Patient no.3: Periapical radiography of left Fig. 3 Patient no.2: Fragment of 3D-CT scan showed mandibular molars showed focus of bone indu- sialolith (outlined) in right submandibular ration of approximately 5 mm in diameter with area; right mandibular ramus showed defor- clear border, probably osteoma, localized close 【版面】W：396mation pt（片段 192 (arrows) pt） H：588by neoplastic pt 【本文】行数不明（手組み） 10pt 12pt (benign) pro- to apical 送り third of distal root of left 2nd man- 【図】●図番号・タイトル・説明：11.3Q 12.7H Newcess Baskerville ITC Std 図タイトルと説明のアキ 9Q ●タイトル折り返し：番号の後（続dibular molar く説明の先頭は字下げ不要） ●図説の幅 片段：片段固定 全段：図幅 【表】●番号・タイトル・説明：11.3Q 12.7H New Baskerville ITC Std タイトルと表のアキ 10.5Q ●罫線 表はじめのみ双罫 表中の 罫の太さ 1.411mm ●表中：11.3Q 12.7H New Baskerville ITC Std ●脚注 11.3Q 12.7H New Baskerville ITC Std 字下げなし 斜体は New Baskerville ITC Std Italic（タグは
Fig. 5 Patient no.3: 3D-CT revealed sialolith in submandibular region
indicated for the ameloblastoma3), but was discomfort during swallowing. These symp- postponed as the patient refused. At present, toms had appeared 4 days earlier, along with the patient’s condition is satisfactory, and she an increase in toothache when biting. This is aware of a possible unfavorable prognosis. toothache resolved, however, the next day, while pain in the submandibular area 3. Case no.3 sharpened. This patient was a healthy 40-year-old Extraoral examination revealed no signifi- woman who attended the university dental cant swelling or lymphadenopathy. Intraoral clinic for consultation on a radiographically examination revealed extensive cavitation in detected osteoma adjacent to the distal root the left mandibular 2nd molar and an of the left mandibular 2nd molar (Fig. 4). enlarged orifice of Wharton’s duct on the left The patient complained of dull pain in the side. The duct itself was deformed, and left submandibular area in the evenings and bimanual palpation of the floor of the mouth Misdiagnosed Sialolithiasis 57 caused pain but no saliva secretion. Conse- vary glands here was mostly due to the non- quently, sialolithiasis was suspected as the radiopacity of the sialolith. Fortunately, an most likely cause. appropriate and relatively timely diagnosis High frequency ultrasound revealed a sialo- allowed for the surgical extraction of a mal- lith approximately 1.0 mm in diameter in the erupted and semi-impacted right mandibular dilated left Wharton’s duct. The glandular 3rd molar, which had been initially indicated, tissue was isoechogenic in structure. A diag- and which is rather traumatic and can incur nosis of sialolithiasis of the left submandibu- the risk of complications due to its interrela- lar salivary gland was confirmed by 3D-CT tionship with the mandibular canal, to be (Fig. 5). abandoned (Fig. 1). An osteoma and endodontic problems with In case no.2, a previously diagnosed amelo- the left mandibular 2nd molar were the pref- blastoma, which was the preferential radio- erential initial clinical findings. However, a graphic finding, rendered sialolithiasis imper- more thorough clinical examination sug- ceptible for at least 9 years, despite the radi- gested submandibular sialolithiasis. opacity of the sialolith. Here, exacerbation of Subsequently, endodontic treatment of the sialolithiasis was mistaken for lymphadenitis, molar was carried out first. The sialolith, was again, despite the radiopacity of sialolith. This treated conservatively as it was small. An oste- also entailed a loss of time for resection of the oma is considered to be an inactive neoplasm, salivary gland. and only a single case of malignant transfor- In case no.3 sialolithiasis was accompanied mation of an osteoid osteoma into low grade by endodontic problems and a minor endos- aggressive osteoblastoma has been reported teal osteoma. Here, the sialolithiasis had to be in the literature9). Therefore, it was decided to treated taking into account the other condi- conduct regular follow-up of the neoplasm. tion. The sialolithiasis in this case was diag- To date, the patient has made no further com- nosed in a timely manner, despite the dentists’ plaints at subsequent follow-up examinations. attention initially being focused on odonto- As to the prognosis, according to the litera- pathological symptoms. ture, the recurrence rate of sialolithiasis is Diagnostic algorithms in dentistry are approximately 8.9%7). The prognosis in based on the odontogenic causes of the prob- regard to osteoma is in generally favorable. lem. This being the case, a chronic process, such as sialolithiasis in the submandibular salivary gland in the presence of affected Discussion teeth in the same quadrant, has almost no chance of being immediately identified by a Despite being one of the most prevalent dentist. In such a case, the diagnostic search disorders of the submandibular salivary can take years, as shown in case no.2 here. glands, the diagnosis of sialolithiasis can still Only after exclusion of all other possible den- be a challenge, especially in dentistry2). Cases tal diagnoses, and probably only after treat- of sialolithiasis, and methods for its visualiza- ment or extraction of teeth, will persistent tion and removal, have frequently been clinical presentation be likely to lead to reported2,4,6). One feature of the present examination of the salivary glands and a report is the combination of dental and non- 3D-CT study of the jaws, which is the only way dental pathologies, with sialolithiasis of the that a sialolith, even one that was radiopaque, submandibular salivary glands affecting the would be identified. same sextant. In all the present cases, 3D-CT was used as Case no.1 showed signs common to two dif- the method of choice for visualization of the ferent diseases: pericoronitis and subman- sialoliths. Meanwhile, one study has reported dibular sialolithiasis. The difficulty in diag- that US is the first choice for such examina- nosing sialolithiasis of the submandibular sali- tion in order to screen for and exclude paren- 58 Kolomiiets SV et al. chymal and intraductal morphological References changes1). 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The authors declare that they have no Correspondence: financial interests, direct or indirect, that Dr. Viktoriya Shynkevych exist or might be perceived to exist in connec- Department of Postgraduate Education tion with the contents of this paper. for Dentists, Higher State Educational Establishment Source of financial support or funding: Nil. of Ukraine “Ukrainian Medical Stomatological Academy”, Poltava, Ukraine E-mail: [email protected]