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Competing Interests: Competing interests: None declared.

Aplasia of salivary glands is rare and usually involves several major salivary glands. Isolated unilateral submandibular gland aplasia or aplasia accompanied by hypertrophy of contralateral submandibular gland or sublingual glands is a rare entity. The majority of cases are incidentally detected findings in asymptomatic patients; however, imaging findings may mimic mass-like lesions, posing diagnostic challenges in cancer patients. We presented 2 cases, a case with melanoma and an asymptomatic case, who had asymmetric lesion at left submandibular space, demonstrated to represent left submandibular gland accompanied by right submandibular gland aplasia. We aimed to highlight the imaging features for correct identification of these mass-like lesions as normal contralateral salivary gland accompanied by unilateral submandibular gland aplasia.

Background: Apert syndrome is characterized by craniosynostosis and bony syndactyly of the hands and feet. The cause of Apert syndrome is a single nucleotide substitution mutation (S252W or P253R) in fibroblast growth factor receptor 2 (FGFR2). Clinical experience suggests increased production of saliva by Apert syndrome patients, but this has not been formally investigated. FGFR2 signaling is known to regulate branching morphogenesis of the submandibular glands (SMGs). With the Apert syndrome mouse model (Ap mouse), we investigated the role of FGFR2 in SMGs and analyzed the SMG pathology of Apert syndrome., Results: Ap mice demonstrated significantly greater SMG and sublingual gland (SMG/SLG complex) mass/body weight and percentage of parenchyma per unit area of the SMG compared with control mice. Furthermore, gene expression of Fgf1, Fgf2, Fgf3, Pdgfra, Pdgfrb, Mmp2, Bmp4, Lama5, Etv5, and Dusp6 was significantly higher in the SMG/SLG complex of Ap mice. FGF3 and BMP4 exhibited altered detection patterns. The numbers of macrophages were significantly greater in SMGs of Ap mice than in controls. Regarding functional evaluations of the salivary glands, no significant differences were observed., Conclusions: These results suggest that the gain-of-function mutation in FGFR2 in the SMGs of Ap mice enhances branching morphogenesis. Developmental Dynamics 247:1175-1185, 2018. © 2018 Wiley Periodicals, Inc., (© 2018 Wiley Periodicals, Inc.)

Development and maturation of submandibular salivary glands are influenced by intrauterine diabetic environment. Several studies investigated the effects of diabetes on the salivary glands. However, the effects of maternal diabetes on the submandibular glands of the offspring was not properly examined. Therefore, the present study was designed to describe the changes in the development of the submandibular glands of the offspring of diabetic mothers. The submandibular glands of the offspring of Streptozotocin (STZ)-induced diabetic female rats were examined at two and four weeks after birth. Detection of mRNA demonstrated that maternal diabetes affects the level of different indicators. The reduction of expression of epidermal growth factor (EGF); a protein mitogen, cytokeratin 5 (CK5); an epithelial cell progenitor, CK7 and aquaporin 5 (AQP5); differentiation markers and B cell lymphoma 2 (Bcl2); an antiapoptotic marker were found. Increase in Bcl2-associated X protein (Bax); an apoptotic marker was detected. These changes indicate their effects on saliva secretion, glands tumorigenesis, growth of normal oral flora and oral microbes, with decreased protein synthesis and production of xerostomia and dental caries. Loss of normal glandular architecture, significant increase in fibrosis, by the detection of collagen fibers, and stagnation of secretory granules were found with atrophic changes in the acinar cells. Marked defect of polysaccharides in the acinar cells, denoting functional changes, was manifested by significant reduction of the intensity of periodic acid-Schiff (PAS) reaction. The positive immunoreactivity of caspase-3, denoting cellular apoptosis, and minimal reaction of alpha-smooth muscle actin (α SMA) and proliferating cell nuclear antigen (PCNA) were evident in the offspring of diabetic mothers. We conclude that maternal diabetes produces degenerative effects in the structure and function of the submandibular salivary glands of the offspring, reflecting possible influences on their secretory activity affecting oral and digestive health., Competing Interests: The authors have declared that no competing interests exist.

Congenital dilatation of the submandibular duct also known as imperforate submandibular duct is a rare condition of unknown etiology, clinically characterized as a well-defined, fluctuant swelling in the floor of the mouth, and microscopically defined as a cystic cavity covered with pseudostratified columnar epithelium consistent with a dilated salivary duct. A 1-month-old female presented with a translucent, smooth cystic swelling in the floor of the mouth causing breastfeeding difficulty. The lesion was surgically excised and microscopically, a cystic cavity lined with pseudostratified columnar epithelium was observed. The final diagnosis was congenital dilatation of the submandibular duct. Dentists and otolaryngologists should consider congenital dilatation of the submandibular duct when evaluating fluctuant swelling in the floor of the month of infants., (Copyright © 2018 Elsevier B.V. All rights reserved.)

Lingual nerve (LN) is one among the 3 branches of posterior division of mandibular nerve. It might get entrapped in the infratemporal fossa by lateral pterygoid muscle, pterygospinous ligament, or lateral pterygoid plate. Its entrapment in the submandibular region has not been reported yet. A unique entrapment of LN in the fused submandibular and sublingual salivary glands in a cadaver was reported. The deep parts of the submandibular and sublingual salivary glands were completely fused with each other. The LN passed through the center of the fused part. Histologically both submandibular and sublingual salivary glands had both mucous and serous acini. Though this entrapment might not cause any functional problems, it might get injured during various surgical procedures of the submandibular region.

We describe a unique case of a 62-year-old patient with recurrent right submandibular sialadenitis. He initially appeared to have extensive sialolithiasis of the right submandibular duct on computed tomography imaging and examination, but sialendoscopy demonstrated a normal-appearing right submandibular duct. An accessory duct posterior and parallel to the normal duct was identified at the time of sialendoscopy and was found to have extensive sialolithiasis, which required removal by both an endoscopic and intraoral technique. To the best of our knowledge, this is the first report of sialolithiasis of an accessory submandibular duct identified at the time of sialendoscopy; the other few reported cases in the literature are based primarily on findings from traditional sialography or magnetic resonance sialography. Identification of accessory salivary ducts requires intraoperative consideration of this entity during sialendoscopy. Open approaches to sialolithiasis, however, may be aided by appropriate preoperative imaging.

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.

Purpose: Sialolithiasis is the most common cause of chronic sialadenitis. In this case report, intraoperative finding of an accessory submandibular duct, obstructed with stone, originating from the same gland nearby the main Warthon's duct, is presented., Case Report: A 22-year-old male patient, suffering from eating-related pain and swelling in his left submandibular region, was diagnosed with left sublandibular gland sialadenitis with radiologically manifested sialolithiasis, and gland excision was advised. Surgery was performed under general anesthesia. When the full anatomical scenery was delineated before excision of the gland, we surprisingly encountered two submandibular ducts originating from ipsilateral gland, one of them was obstructed with stone. After two ducts were ligated, the gland with sialolith was excised. According to histopathologic examination, the duct obstructed with stone was identified as the accessory duct and the other one was the main Wharton's duct. Postoperative days were uneventful; no neurologic complication was observed., Conclusions: Otolaryngologists should be aware of anatomic variations of the submandibular duct(s) to avoid possible complications, especially intraoperatively, because rutine preoperative radiologic preparation does not include investigation of possible accessory ducts.

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

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.

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., (Copyright © 2014 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

Congenital agenesis of the submandibular gland is uncommon. We report dynamic sonography and CT findings of the unilateral submandibular gland agenesis associated with herniated hypertrophic sublingual gland tissue through the mylohyoid gap. The dynamic sonography examination applied at rest and during the modified Valsalva maneuver demonstrated hypertrophied sublingual gland prolapsing to the anterior part through the mylohyoid gap. This may be the first reported case describing dynamic sonography findings of this entity. CT confirmed the agenesis of the left submandibular gland and compensatory hypertrophy of the ipsilateral sublingual gland., (Copyright © 2013 Wiley Periodicals, Inc.)

Agenesis of a submandibular gland with compensatory hypertrophy of the contralateral gland is an extremely rare disorder. Most reported cases of unilateral submandibular agenesis have been asymptomatic without any associated facial anomalies. We report a case of submandibular gland hypertrophy mimicking a mass that was detected incidentally. The absence of the contralateral submandibular gland and associated contralateral sublingual gland hypertrophy was confirmed by typical imaging findings., (Copyright © 2012 Wiley Periodicals, Inc., A Wiley Company.)

Sjogren's syndrome (SS) is a complex autoimmune disease that primarily affects salivary and lacrimal glands and is associated with high morbidity. Although the prevailing dogma is that immune system pathology drives SS, increasing evidence points to structural defects, including defective E-cadherin adhesion, to be involved in its etiology. We have shown that E-cadherin has pivotal roles in the development of the mouse salivary submandibular gland (SMG) by organizing apical-basal polarity in acinar and ductal progenitors and by signaling survival for differentiating duct cells. Recently, E-cadherin junctions have been shown to interact with effectors of the Hippo signaling pathway, a core pathway regulating the organ size, cell proliferation, and differentiation. We now show that Hippo signaling is required for SMG-branching morphogenesis and is involved in the pathophysiology of SS. During SMG development, a Hippo pathway effector, TAZ, becomes increasingly phosphorylated and associated with E-cadherin and α-catenin, consistent with the activation of Hippo signaling. Inhibition of Lats2, an upstream kinase that promotes TAZ phosphorylation, results in dysmorphogenesis of the SMG and impaired duct formation. SMGs from non-obese diabetic mice, a mouse model for SS, phenocopy the Lats2-inhibited SMGs and exhibit a reduction in E-cadherin junctional components, including TAZ. Importantly, labial specimens from human SS patients display mislocalization of TAZ from junctional regions to the nucleus, coincident with accumulation of extracellular matrix components, fibronectin and connective tissue growth factor, known downstream targets of TAZ. Our studies show that Hippo signaling has a crucial role in SMG-branching morphogenesis and provide evidence that defects in this pathway are associated with SS in humans.

A 4 year-old female patient was treated for persistent right-sided dacryocystitis and xerostomia. MRI was performed to screen for a dry syndrome; which resulted in the diagnosis of agenesis of the parotid and submandibular glands as well as lacrimal duct malformation. An MRI of each parent was normal. The mother's history revealed 4 days of pyrexia during the 8th week of amenorrhea. This was an isolated case, with no family history, characterized by a febrile episode during pregnancy at the period of main salivary gland genesis. Epigenetic mechanisms could be implicated., (Copyright © 2013 Elsevier Ireland Ltd. All rights reserved.)

Aplasia of the major salivary glands is very uncommon, whereas isolated aplasia of unilateral submandibular gland is even rarer. In this article, we report a 55-year-old female case in whom unilateral aplasia of the left submandibular gland was detected incidentally by ultrasonography with no other congenital comorbidities in the light of literature data.

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., (Copyright © 2012 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.)

There are very limited reports of aplasia of bilateral submandibular glands associated with bilateral sublingual gland hypertrophy. We report the case of a 13-year-old girl with bilateral submandibular gland aplasia associated with symmetrical sublingual gland hypertrophy that herniated from bilateral mylohyoid defects and presented as bilateral masses on CT and MRI images. Prolapsed bilateral sublingual gland hypertrophy should be considered in patients with bilateral submandibular masses to avoid unnecessary biopsy or surgery. We suggest that radiological evaluation of these cases should be performed before any intervention.

The congenital absence of the major salivary glands is a very infrequent disorder, in which several glands are usually involved at the same time. Sometimes this disorder can be associated with other developmental anomalies. The unilateral aplasia of the submandibular gland is an extremely rare finding with only 14 cases reported in the literature. Clinically, this kind of patients may complain of dryness of the mouth, difficulties in chewing and swallowing, severe periodontal disease or multiple caries, but usually they follow an asymptomatic course. Salivary gland aplasia can be diagnosed with a large variety of imaging techniques, which include computer tomography (CT), magnetic resonance imaging (MR), ultrasonography (US), sialography, or scintigraphy. In this paper we report a case of a patient referred to our department with a long term and progressive growing neck mass, who has an unilateral submandibular gland aplasia associated to an ipsilateral hypertrophy of the sublingual gland.

Congenital aplasia of the major salivary glands is rare, and there have been few cases of the condition reported to date. In many cases, absence of the salivary glands is associated with syndromes involving the ectodermal tissues. The xerostomia encountered in affected children is usually associated with increased risk for caries and infections of the soft tissues. The purpose of this paper was to describe the case of a 10-year-old boy with bilateral aplasia of the submandibular and parotid salivary glands and the preventive and restorative treatment rendered. This case study demonstrates the importance of timely diagnosis of this condition in order to prevent the serious oral complications from xerostomia.

Congenital aplasia of the major salivary gland is a rare condition. We report on a case of bilateral aplasia of the submandibular glands associated with a left submandibular hemangioma. A 62-year-old woman came to our department complaining of a 3-year history of left submandibular swelling. She had no notable family or personal medical history. On physical examination of the head and neck region, a 30 mm, non-tender mass was palpated in the left submandibular area. On imaging examinations including ultrasonography and CT, lack of the right submandibular gland and a left submandibular mass with calcification were demonstrated. Fine needle aspiration cytology resulted in blood elements only. Functioning tissue could not be observed in the bilateral submandibular glands on technetium pertechnetate scintigraphy. We performed a left submandibular tumor extirpation. Intraoperatively, the left submandibular gland and duct were missing. On pathologic examination, the tumor was found to be a hemangioma. The parotid and sublingual glands were recognized bilaterally on postoperative MRI. Her postoperative condition was satisfactory. This condition may be due to the dysfunction of several factors, such as fibroblast growth factors, related to gland differentiation.

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 anomalies is 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 of the complete acinar atrophy secondary to an early obstruction of Wharton's duct.

Aplasia of the major salivary glands is a very uncommon anomaly, with isolated aplasia of only 1 submandibular gland being even rarer. We report a case of a 60-year-old woman in whom unilateral aplasia of the left submandibular gland was detected incidentally on computerized tomography with no other associated congenital abnormalities., (Copyright 2010 Mosby, Inc. All rights reserved.)

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.

Objective: To better understand the diagnosis, treatments, and outcomes of congenitally imperforate salivary ducts., Methods: A systematic literature review was performed using the following terms: imperforate submandibular duct, imperforate Wharton's duct, congenital atresia submandibular duct, congenital atresia sublingual duct, and congenital ranula. Only papers focusing on congenital floor of mouth cysts arising from imperforate salivary ducts were included. Two additional patients from our own experience are also discussed., Results: Seventeen papers published since 1950 met inclusion criteria. A total of 26 patients underwent therapy ranging from mere observation to complete excision of the cyst with the sublingual gland. Both of our 2 additional patients both underwent simple sialodochostomy (excision of the imperforate sublingual caruncle) with cyst decompression. For all 28 patients, there have been no reported recurrences, with mean follow-up of 24.8 months and median follow-up of 14.8 months., Conclusions: Congenital floor of mouth cysts arising from imperforate salivary ducts are rare, but they may mimic the appearance of a ranula. Unlike ranulas, these cysts may be safely and effectively cured by simple sialodochostomy. To date, 26 other cases have been described in the literature, and nearly all have been successfully treated with simple incision or marsupialization without recurrence. Care should be taken to exclude ranula as a diagnosis, as incomplete excision may likely result in recurrence.

Aplasia of major salivary glands is very rare. Compensatory hypertrophy of the rest of the glands can result in clinico-radiological masses. We present a report of a rare case of non-syndromic bilateral submandibular gland aplasia with hypertrophied sublingual salivary tissue, the latter herniating through mylohyoid boutonnière to present as a palpable mass on the left side with corresponding CT findings. Multiplanar evaluation is emphasised by utilizing multidetector CT.

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.

Authors based upon multispiral computer tomography with building up multiplanar 3D-reconstructions disclosed earlier not described developmental anomaly of salivary glands: rudimentary ducts running beyond capsule of parotid and submandibular glands.

Objective: Five reports have examined unilateral submandibular gland aplasia. The purposes of this report are to demonstrate submandibular gland aplasia leading to contralateral gland hypertrophy in the setting of oral cavity cancer and to discuss the corresponding diagnostic and management challenges., Study Design: This study is a case report of a 60-year-old male who presented with pain on the right side of the mobile tongue., Method: This report uses literature review., Results: A 60-year-old male presented with pain on the right side of the mobile tongue. Subsequent results of punch biopsy revealed squamous cell carcinoma in situ with foci of microinvasion of the tongue. Head and neck examination revealed no abnormalities. The patient underwent a wide-local excision of the tongue lesion. Postoperative computed tomographic (CT) scan showed an asymmetric mass on the ipsilateral side of the cancer in the region of the submandibular gland. The gland was noted to be abnormally large. A diagnosis of contralateral submandibular gland aplasia was made. The patient is cancer-free at 2 years postlocal excision. Salivary gland aplasia is an extremely rare disorder and is often associated with various congenital syndromes. Unilateral submandibular gland aplasia is even rarer with ours representing the sixth reported case. Aplasia is believed to stem from a regional disturbance in early fetal development. Common symptoms can include dysphagia, dry mouth, decreased taste, and tooth decay. In the presence of a history of oral cavity cancer, unilateral submandibular gland aplasia poses a challenge during postoperative cancer follow-up., Conclusions: Unilateral submandibular gland aplasia in the setting of oral cavity cancer poses a unique challenge for cancer follow-up. Hypertrophy of the submandibular gland on the other side can masquerade as nodal metastasis. Head and neck examination as well as CT scan can be inconclusive. Regular confirmatory tests such as fine needle aspiration biopsy and positron emission tomography/CT for cancer detection is extremely useful for detecting recurrence.

Atresia of the submandibular ducts (particularly the duct orifice) is a rare occurrence that is mainly observed in newborns or infants; moreover, its differential diagnosis with other anomalies of the oral floor is sometimes difficult. Given the lack of data concerning the differences between atresia of the orifice of Wharton's duct and other anomalies of the submandibular duct system, we describe here the findings of videosialoendoscopic assessment of the salivary duct system in an infant who underwent traditional surgery for bilateral atresia of the orifice of Wharton's duct. During sialoendoscopy, a trifurcation of the main duct, a normal variant of the more common bifurcation of the secondary branches of the salivary duct system, was observed bilaterally. No other ductal anomalies were encountered. Complete resolution of the symptoms and complete patency of both ducts at 19-month follow-up evaluation were observed. In conclusion, although spontaneous resolution of the disease during feeding is possible, minimal incision of the orifice of the imperforated submandibular duct together with diagnostic videosialoendoscopy of the main salivary duct system may be considered as part of the modern approach to salivary obstructive disorders in pediatric age as is currently the case for sialolithiasis and recurrent juvenile parotitis.

In the sublingual/submandibular gland complex (L/M complex hereafter), the submandibular gland process adjoins the sublingual gland. Therefore, it is speculated that the L/M complex in the fused state is due to excessive development of the submandibular gland process. However, we encountered a case of the L/M complex formed due to supernumerary glandular lobes (lobes A, B, and C) developing in the posterior 1/3 portion of the submandibular duct. Lobes A, B, and C showed the following macroscopic morphologies, constitutions, ducts, and histological morphologies. Lobe A was disc-shaped, and this lobe together with the major sublingual gland was wrapped with the minor sublingual gland. Lobe B was attached to lobe C, forming a cingulate lobe. Lobe B had an irregular shape and was attached to the superior area of lobe A, while lobe C was rod-shaped and attached to the posterior area of lobe B. The posterior area of lobe C was attached to the submandibular gland at the base of the submandibular duct. The excretory ducts of lobes A, B, and C joined, forming one duct, which joined the posterior 1/3 portion of the submandibular duct. The histological morphologies of lobes A, B, and C showed a predominance of serous cells over mucous cells, which was the same as the morphology of the submandibular gland. In this case, the L/M complex may have formed by the following mechanism. Supernumerary lobes A, B, and C of the submandibular gland formed in the posterior 1/3 portion of the submandibular duct, and lobe A together with the major sublingual gland was wrapped with the minor sublingual gland. Lobes B and C formed a cingulate lobe, and its posterior portion (posterior portion of lobe C) attached to the submandibular gland, resulting in a fused state.

Varried conditions such as ranula, epidermal/dermal inclusion cyst, lymphatic cyst, thyroglossal cyst, sialolithiasis, branchial cleft cyst are known to produce swelling in the floor of mouth. Rarely imperforate or duplication anomaly of submandiblar duct may produce cystic lesion in the floor of mouth. We present a case of congenital imperforate submandibular duct with cyst formation in a newborn. We also review the literature regarding management.

Atresia of the submandibular duct orifice is a rare developmental anomaly. Early identification and treatment of atresia of the submandibular duct orifice prevent feeding and breathing difficulties, dilatation of the submandibular duct, submandibular gland infection, or pressure atrophy. Imperforate submandibular duct should be distinguished from duplication abnormalities of the duct and other cystic lesions in the floor of the mouth. To date, magnetic resonance imaging findings of the atresia of the submandibular duct orifice have not been reported. The aim of this study was to describe clinical and radiologic evaluation of an infant with atresia of the bilateral submandibular duct orifices.

Congenital absence of the major salivary glands is an infrequent disorder. Clinically, patients may be asymptomatic or may present with dryness of the mouth, difficulty in chewing and swallowing, and dental caries. The absence of the submandibular gland may be associated with hypertrophy of the contralateral submandibular gland. We report a case of ipsilateral sublingual hypertrophy associated with unilateral submandibular aplasia.

Background: Dental destruction can develop from numerous causes. Major salivary gland aplasia is an uncommon causative factor. The resulting xerostomia can lead to extensive dental demineralization., Case Description: The author examined a 19-year-old man because of the patient's concern regarding decreased salivary volume and his dental condition. There was extensive loss of tooth structure and an astonishing pattern of dental destruction most notable on the palatal portions of the maxillary molars and premolars that is best described as "chipping." It was only after taking the patient's history, clinically examining the patient and conducting a radioisotope study that the author was able to make a confident diagnosis of the absence of four major salivary glands., Clinical Implications: Dentists should be aware that salivary gland aplasia is an uncommon cause of dental deterioration. It may manifest itself not by extensive caries but by a dental chipping effect. Early recognition and a therapeutic strategy can prevent progressive dental damage.