Accessory Submandibular Salivary Gland Forming a "Horseshoe" With the Main Submandibular Salivary Gland: A Unique Variation.

Presence of accessory submandibular salivary gland (ASSG) is an extremely rare variation. Knowledge of its relations could be very useful to oral and maxillofacial surgeons, head and neck surgeons, and radiologists. During dissection classes, an ASSG was noted between the mylohyoid and hyoglossus muscles. The main submandibular salivary gland had superficial and deep parts. The deep part was narrow and measured about 5 cm. The lingual nerve passed between the superficial and deep parts. The accessory submandibular gland was situated below and parallel to the deep part of SSG. It also measured 5 cm. The ASSG had its own duct, which joined the duct of main gland. The ASSG and the deep part of the SSG were united at the lateral border of geniohyoid muscle to give a characteristic "horseshoe" appearance. The ASSG overlapped both lingual and hypoglossal nerves.

Are the patients with anatomic variation of the sublingual/Wharton's duct system predisposed to ranula formation?

OBJECTIVE: To evaluate ranula development according to anatomic variation of the ductal system of sublingual gland (SLG), especially the presence of Bartholin's duct. METHODS: The anatomic variation of SLG duct was prospectively investigated and compared between 55 consecutive patients with ranulas treated by SLG excision (group 1) and another 15 consecutive patients undergoing similar surgeries for other conditions (group 2). The ductal structures of SLGs and submandibular glands (SMG) were also compared between the pediatric patients and adult patients with ranulas. RESULTS: In 32 of 55 patients with ranulas (58.2%) and 1 of 15 patients without ranulas (6.7%), the SLG showed an anatomic variation of the main duct called Bartholin's duct structure (P<0.01). Seventeen of 22 (77.3%) pediatric patients with ranulas had Bartholin's ducts and 15 of 33 (45.5%) adult patients with ranulas had Bartholin's ducts (0.01

Congenital dilatation of Stensen's duct in siblings.

Congenital dilatation of the Stensen's duct is an extremely rare disease, which may have a hereditary background. However, there are no reports about the familial occurrence of congenital Stensen's duct dilatation. Herein, we present the first case of congenital dilatation of Stensen's duct in siblings. Therefore, we suggest that congenital dilatation of the Stensen's duct is an extremely rare hereditary disorder.

Large tonsillolith associated with the accessory duct of the ipsilateral submandibular gland: support for saliva stasis hypothesis.

Tonsillolith is a calcified mass in the tonsil and/or its surrounding tissue, which is considered to be caused by chronic tonsillitis. However, here we hypothesized that a tonsillolith can also be formed by chronic saliva stasis in the tonsillar tissue, without any signs of chronic inflammation. We present the case of a 32-year-old male patient with a large tonsillolith. We reviewed his medical files, pre-operative imaging and intraoperative findings. During a standard tonsillectomy, we encountered a large tonsillolith measuring 3.1 × 2.6 cm. Additionally, a careful dissection of the lower pole of the remaining tonsillar tissue revealed a large fistulous tract filled with saliva. Post-operative examination of the pre-operative CT scan found a hypodense fistulous tract extending from the lower tonsillar pole towards the left submandibular gland, measuring 36 mm in length, which was diagnosed as an accessory duct of the submandibular gland. To our knowledge, this is the first case of a large tonsillolith associated with the accessory duct of the ipsilateral major salivary gland. Furthermore, from the aetiopathological view, this finding supports the saliva stasis hypothesis for formation of the tonsillolith. However, larger studies, including a detailed radiological analysis as in our case, are needed to further investigate this possible aetiology of tonsilloliths.

Recurrent sublingual ranula or saliva leakage from the submandibular gland? Anatomical consideration of the ductal system of the sublingual gland.

PURPOSE: Some believe that the recurrence of sublingual ranula results from incomplete removal of the sublingual gland (SLG), but recurrence remains in some patients who undergo repeated excision of the remnant SLG, and the final solution to the recurrence is to remove the ipsilateral submandibular gland (SMG). In the authors' experience, preoperative aspirate from a sublingual ranula was a thick mucus-like fluid resembling egg white, whereas that from recurrent cyst after removal of the SLG was thin serous fluid. Based on the difference of the aspirated fluids, the authors speculated that the recurrent cystic mass might not be a ranula, but rather iatrogenic saliva leakage from the SMG through the previous surgically damaged excretory duct of the SLG (Bartholin duct) that opens into the SMG duct (Wharton duct). MATERIALS AND METHODS: A gross anatomic study was performed of the ductal system of the SLG and the anatomic communication between the Bartholin duct and Wharton duct. RESULTS: Four anatomic SLG duct variants were found. 1) The SLG has 1 Bartholin duct that seems to fuse with the Wharton duct but does not join the Wharton duct, running parallel to the Wharton duct and opening at its own orifice adjacent to and a short distance from the orifice of the Wharton duct (35.8%). 2) The SLG has 1 Bartholin duct that empties into the middle section of the Wharton duct (32.1%). 3) The SLG has 2 Bartholin ducts, one of which joins the Wharton duct and the other opens at its own orifice near that of the Wharton duct on the floor of the mouth (7.1%). 4) The SLG has many fine ducts (Rivinus ducts) that open at the floor of the mouth with no relation to the Wharton duct (25.0%). Of the 4 anatomic SLG duct variations, types 2 and 3 form immediate anatomic communication between the Bartholin duct and Wharton duct (39.2%). CONCLUSIONS: Several conclusions can be made from the present anatomic findings. 1) A certain proportion of Bartholin ducts open into the Wharton duct, and "recurrent ranula" after removal of the SLG can result from iatrogenic saliva leakage from the SMG through the surgically severed Bartholin duct if the aspirated fluid from the "recurrent cyst" is a thin seriflux. 2) The Bartholin duct emptying into the Wharton duct should be ligated during removal of the SLG to prevent local saliva accumulation from the SMG or even caudal "recurrence" as plunging ranula. 3) The surgical incision for SLG removal should be sutured loosely so that the surgically severed Bartholin duct with communication to the Wharton duct can reopen at the floor of mouth as an outlet for possible saliva leakage from the SMG and saliva accumulation can be avoided.

Unilateral duplication of the parotid duct, its embryological basis and clinical significance: a rare cadaveric case report.

The parotid gland is the largest salivary gland in humans, and produces mainly serous secretions. The secretion of this gland reaches the oral cavity through single parotid duct (Stensen's duct). The parotid duct begins at the anterior border of the gland, crosses the masseter, and then pierces the buccinator and open the mouth. The present study reports a rare variation of a unilateral double parotid duct that was observed in the right side of the face of a 75-year-old male cadaver during dissection. The embryological basis of this variation can be explained as follows. A sprout originates from epithelium lining the primitive oral cavity, grows dorsally, and bifurcates. The end parts of these branches invaginate into the adjacent mesenchyme separately, proliferate, and make the parotid gland, while their proximal parts later form communicating ducts (D1 and D2) connecting the gland to the mouth. Furthermore, the clinical importance of this variation was discussed.

[Pneumoparotid: a rare cause of recurrent parotid swelling]. / Pneumoparotide : une étiologie rare de tuméfaction parotidienne récidivante.

INTRODUCTION: Pneumoparotid is a rare cause of recurrent parotid swelling. It is often fortuitous, caused by an action inducing intraoral pressure, increased or self-induced. OBSERVATION: An 8-year-old boy presented with recurrent right unilateral parotid swelling for 2 years. A CT-scan was performed, outside of the acute phase, which revealed the presence of intraparotid air bubbles. DISCUSSION: Pneumoparotitis results from air flowing back into Stensen's duct, due to an anatomical abnormality and a significant increase of intraoral pressure. The diagnosis is made on clinical examination, often completed by a morphological examination (cervicofacial CT-scan or parotid ultrasound investigation). The outcome is most often favorable, with symptomatic treatment, and sometimes antibiotic prophylaxis. Patient education is the best means to prevent recurrence.

A case of imperforate Wharton duct.

Congenital oral masses are rare entities. The establishment of formal fetal diagnostic teams has led to an increased antenatal detection of such lesions. The congenital ranula is a distinct entity from the more familiar variant presenting later in life. The congenital variant may result from an anomaly of the Wharton duct with subsequent dilation of the duct. The variant presenting later in life is the more familiar mucous extravasation phenomenon in the floor of the mouth. Management of the congenital ranula is distinct from its noncongenital counterpart and more conservative and is discussed in the present report.

Ductal variation of the sublingual gland: a predisposing factor for ranula formation.

BACKGROUND: The purpose of this study was to evaluate ranula development according to anatomic variation of the sublingual gland (SLG). METHODS: We conducted a prospective, consecutive case series considering other clinical conditions. Twenty-five cases treated by SLG excision were enrolled in this study. The ductal structures of the SLGs of another 11 patients undergoing similar surgeries for other conditions were compared. RESULTS: In 19 of a total of 25 ranulas (76.0%) and 16 of 18 oral nonplunging ranulas (88.9%), the SLG showed an anatomic variation of the main duct called Bartholin's duct structure. Meanwhile, only 3 of 7 plunging ranulas (42.9%) had Bartholin's ducts. Bartholin's duct structure of the SLG was not found in the 11 control cases. CONCLUSION: Anatomic variation of the ductal system of the SLG might be a possible cause of ranulas. Surgical resection of the SLG is a better treatment choice for ranulas than other, more conservative treatments.

[Treatment of significant parotid ducts deformities].

The paper summarizes authors experience in treatment of 14 patients with congenital parotid ducts deformities (megastenon type). The results showed reconstructive surgical treatment to be sufficiently effective in such cases. If restoration of parotid function is not possible one should consider interventions for secretion reduction and duct walls excision.

"Mustache sign" due to Stensen duct dilation.

An 80-year-old woman presented with a 5-year history of painless swellings of the left and right cheeks. The degree of swelling did not change with mastication. On palpation, the cheeks were soft, well defined, and movable. Compression and massage of the swollen areas caused increased salivary discharge from the orifices of the Stensen ducts. Three-dimensional computed tomography showed well-bordered, 15- to 20-mm wide, bilateral, tube-like dilatations of the ducts. The ductal origin of the swellings was explained to the patient, but she refused invasive procedures, thus no sialogram or surgical procedure was performed. We describe the clinical and radiographic features of a case of bilateral, congenital Stensen duct dilatation with bilateral swelling of the cheeks.

Diagnostic imaging for submandibular duct atresia: literature review and case report.

PURPOSE: The basic aim of this review was to retrospectively study the role of imaging, particularly magnetic resonance imaging, in the diagnosis of submandibular duct (SMD) orifice atresia. MATERIALS AND METHODS: From a literature review, data were collected from previously reported cases of imaged SMD atresia and from the present case. RESULTS: Five images, 4 from the literature and 1 magnetic resonance image from our case, were obtained to illustrate the singular appearance of SMD orifice atresia. CONCLUSIONS: To the authors' knowledge, this is the first study, using a review of the literature concerning SMD orifice atresia, to call specific attention to the role imaging plays in illustrating the pathognomonic presence of the uniquely dilated and segmented salivary duct seen in SMD orifice atresia.

Congenital salivary fistula of an accessory parotid gland in Goldenhar syndrome.

OBJECTIVES: We report two cases of congenital salivary fistula of an accessory parotid gland, and we discuss its occurrence in Goldenhar syndrome. METHODS: Two teenagers complained of a congenital cheek fistula with constant salivary discharge. Computed tomography fistulography and sialography were performed. The diagnosis of Goldenhar syndrome was established based on clinical and imaging findings. Previously reported cases are reviewed and the clinical and radiological features summarised. RESULTS: In these two patients, a salivary fistula of an accessory parotid gland was demonstrated on computed tomography fistulography, and did not communicate with Stensen's duct. Deformity of Stensen's duct and hypoplasia of the ipsilateral mandibular ramus were present. Tragal appendices have frequently been reported in such cases. CONCLUSION: A congenital cheek salivary fistula of an accessory parotid gland should be considered indicative of Goldenhar syndrome.

Diagnosis and treatment of congenital dilatation of Stensen's duct.

OBJECTIVES/HYPOTHESIS: This study aimed to describe the diagnosis and management of congenital dilation of Stensen's duct (CDSD) in seven cases. STUDY DESIGN: Retrospective study. METHODS: We collected data including medical records, radiology, and histopathology findings and follow-up for seven patients (four males) with CDSD. The mean age was 23.9 years (range, 2-72 years). RESULTS: The clinical features of CDSD were the primary presentation of painless swelling in the cheek without an obvious cause that was unilateral or bilateral and occurred at any age, and a swelling along Stensen's duct. In patients without a history of inflammation, aggressive massage of the swelling could produce abundant intraoral salivary flow. Parotid sialography demonstrated a dilated Stensen's duct with a smooth margin but no obvious obstruction. All seven patients underwent superficial parotidectomy including the intact Stensen's duct. No patient showed recurrent swelling after a follow-up of 6 to 65 months. CONCLUSIONS: CDSD is an uncommon congenital disorder of the parotid gland. Management with parotidectomy is effective.

Unilateral absence of submandibular gland secondary to stones. Aplasia versus early atrophy

Major salivary gland absence is a rare disorder. The cause of congenital absence of the salivary glands has not been determined, but it may be associated with ectodermal defects of the first and second branchial arches. Isolated absence of a unilateral submandibular gland is an unusual entity with less than ten cases reported in the literature.The etiopathogenesis of isolated absence of a major salivary gland without other developmental anomaliesis still unclear. The formation of a sialolith within the remaining Wharton’s duct, associated with isolated aplasia(versus atrophy) of a unilateral submandibular gland has been recently reported. We describe in this work two cases of sialolithiasis within the ipsilateral remaining Wharton´s duct in patients with isolated absence of a unilateral submandibular gland. In the cases reported, absence of the submandibular gland may have been the result ofthe complete acinar atrophy secondary to an early obstruction of Wharton´s duct (AU)

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Atresia of the submandibular duct orifices: an unusual cause of feeding problems and failure to thrive in an infant.

UNLABELLED: Atresia of the submandibular duct orifice is a rare developmental anomaly, which causes swelling of the duct by accumulation of saliva. The cystic mass in the floor of the mouth can cause feeding problems, which can be treated by surgical opening of the duct. We report the first Swedish case in a male infant, who had severe difficulties to feed because of bilateral swellings of the submandibular ducts caused by orifice atresia. CONCLUSION: This is the first case that has described failure to thrive because of this condition and catch up after treatment. It is important to remember that evaluation of feeding problem in an infant must include inspection of the oral cavity.

Gustatory otorrhoea: a rare case of congenital external ear salivary fistula.

OBJECTIVE: We present an extremely rare case of a 44-year-old woman with right gustatory otorrhoea and otalgia. CASE REPORT: The patient had been initially treated for otitis externa after Pseudomonas aeruginosa was grown from a microbiological swab. The otorrhoea fluid was collected and tested positive for amylase. Sialography and computed tomography imaging of the temporal bone confirmed a sialo-aural fistula from the right parotid gland to the bony external acoustic meatus. The defect was consistent with a patent foramen of Huschke. The fistula was identified surgically via a superficial parotidectomy approach, after contrast injection of Bonney's blue dye into the parotid duct, and then ligated and divided. The patient had immediate and sustained resolution of her otorrhoea. CONCLUSIONS: Sialo-aural fistulae are extremely rare, and usually arise as a complication of surgery or as an acquired disease process. To date, only four cases have been reported. This case demonstrates the use of sensitive investigation involving sialography and computed tomography, as well as successful surgical management, with complete resolution of symptoms.

Sialolithiasis in a stump of Wharton's duct of an aplastic unilateral submandibular gland.

Congenital absence of the salivary glands is an uncommon disorder. Isolated aplasia of a unilateral submandibular gland is rare and only 6 cases have been reported. The formation of a sialolith within the remaining Wharton's duct, associated with isolated aplasia of a unilateral submandibular gland, has not been reported previously. The authors report two cases of sialolithiasis within the ipsilateral remaining Wharton's duct in patients with isolated aplasia of a unilateral submandibular gland.