Submandibular Mass Research Articles

Infantile Myofibroma or Lymphatic Malformation

A 5-day-old male neonate was referred to our vascular anomalies center with a large cystic submandibular mass. History and physical examination and ultrasonographic results indicated the diagnosis to be macrocystic lymphatic malformation. Consequently, the child was treated with sclerotherapy and subtotal excision. The histopathological examination result showed that the lesion was infantile myofibroma. This diagnostic error was attributed to atypical features of infantile myofibroma in this child: unusually large cysts, rapid enlargement, and coagulopathy. This report expands the clinical spectrum of infantile myofibromatosis and suggests its consideration in the differential diagnosis of neonatal cystic cervicofacial lesions.

Primary tumors of the submandibular glands: A retrospective study based on 41 cases

We report our experience on the diagnostic approach, treatment, and follow-up of primary submandibular gland tumors. Retrospective review. Tertiary referral center. Forty-one adult patients, 22 male and 19 female, with primary submandibular gland tumors, 20 benign and 21 malignant. Age, gender, clinical findings, cyto- and histopathology, treatment and outcome were analyzed. Most tumors presented as a painless submandibular mass. Thirty three patients underwent a fine needle aspiration, the sensitivity, specificity and accuracy of which--in detecting malignant tumors--were 79%, 100% and 88%, respectively. Preoperative radiological imaging was performed in 30 cases. Patients with benign tumors were treated with surgery. Most malignant tumors were treated with a combined modality, including neck dissection and radiation therapy. Five patients developed a postoperative complication. Recurrent disease was encountered in 5 malignant tumors. The 2, 5 and 10 year disease-specific survival of patients with malignancy were 84%, 75% and 41%, respectively. The preoperative assessment of the nature of submandibular gland tumors remains challenging. Aggressive treatment of patients with malignant disease may help to avoid poor prognosis.

Lateral Ectopic Thyroid Goiter with a Normally Located Thyroid

Midline ectopic thyroid tissue is a common pathological entity. In contrast, laterally located ectopic thyroid tissue with a normally located thyroid gland is a very rare condition in head and neck surgery. We report the association of laterally located multinodular thyroid tissue and a normally located multinodular goiter. A woman was seen in the otolaryngology clinic with a right submandibular mass. Clinical examination and investigations confirmed the presence of a multinodular goiter in an ectopic thyroid gland. Laterally located ectopic thyroid tissue is a very rare condition. Ectopic thyroid tissues can undergo the same pathological changes as a normally located thyroid gland. Ectopic thyroid goiter together with a normally located multinodular goiter is a rare entity, and this is the first to be reported in North America.

Submandibular Mass Excision in an Asian Population: A 10-year Review

The objectives of this study were to compare the prevalence of submandibular gland neoplasia in an Asian population with a Western population and to evaluate the accuracy of fi ne needle aspiration cytology (FNAC) and computed tomography (CT) scan in the study of submandibular gland pathologies. We conducted a 10-year retrospective review of 101 submandibular mass excisions. Data on the demographic profi le of patients, clinical features, correlation of fi ne needle aspiration cytology, CT scans and histology and morbidities related to surgery were collected. The prevalence of submandibular gland neoplasia was 27.1%. Most (78.9%) of these were benign. FNAC and CT scans were accurate for benign neoplasia. However, the accuracy of FNAC and CT scans for sialadenitis without sialolithiasis was low. The prevalence of submandibular gland malignancy was lower in the Asian population. FNAC and CT scans were accurate for benign neoplasia of the submandibular gland.

Isolated submandibular metastasis from a contralateral thyroid papillary microcarcinoma: an unusual case

Papillary carcinoma is the most common form of thyroid cancer. It is a relatively indolent disease, which commonly remains clinically silent until its incidental histological diagnosis in surgical material or at autopsy. A tumour less than 10 mm in size is termed a papillary microcarcinoma. Papillary microcarcinoma may present with clinical symptoms, most commonly jugulodigastric and pretracheal lymphadenopathy with or without palpable thyroid nodules. Isolated submandibular metastases are rare. We present the case of a submandibular metastasis arising from a solitary 3 mm papillary microcarcinoma of the thyroid on the contralateral side in a 46-year-old woman. We describe the ultrasound and MRI characteristics of the submandibular mass. The ultrasound findings in particular were suggestive of a thyroid malignancy and prompted detailed examination of the thyroid gland. Clinical and radiological examination of the thyroid was normal. To the best of our knowledge, we present the first report of a papillary microcarcinoma of the thyroid presenting as a contralateral and isolated submandibular mass.

Fibromatosis of the Submandibular Region

Fibromatoses are benign tumors that can occur anywhere in the body. They are locally aggressive and tend to recur; they also cause considerable morbidity, particularly when they arise in the head and neck. Fibromatosis in the submandibular region is rare. Here, we present a case of fibromatosis of the submandibular region in a 42-year-old female who presented with a right submandibular mass. The patient underwent complete excision of the fibromatosis, showed negative margins and had no adjuvant therapy. Her recovery was uneventful, and there was no recurrence or neurological deficit 1 year after the operation.

A hidden cause of renal failure.

A frail 84-year-old man of SE Asian origin with a history of metastatic prostate cancer presented with non-specific deterioration. Initial investigations showed acute on chronic renal failure (creatine 648 μmol/L; baseline 153), hyperkalaemia (K+ 6.4 mmol/L) and a marked leucocytosis (WBC 46 × 109/L; neutrophils 33 × 109/L). The only significant features found on examination were mild supra-pubic tenderness and a diffuse tender left submandibular mass (Figure 2). This was thought to be a lymph node, but was difficult to assess due to a dense beard and communication limitation by language barriers. Immediate treatment was correction of the hyperkalaemia by haemodialysis. A renal ultrasound did not show any obstruction and the prostate-specific antigen was normal. Other investigations revealed a serum paraprotein IgG lambda 14.3 g/L with high concentrations of free lambda light chains in blood and urine. Bone marrow aspirate was consistent with myeloma and this was accepted as the cause of his renal failure. The patient's main complaint was ‘toothache’. Nothing was found on examination of the mouth, and the patient was advised to visit his dentist after discharge. After further requests from the patient, a maxillofacial review was sought. Unexpectedly, orthopantogram films showed a large destructive mass in the left mandible (Figure 3). A CT scan confirmed this (Figures 1 and 4), and a biopsy showed a plasmacytoma (now over a month since admission). The patient received local radiotherapy and chemotherapy with dexamethasone, cyclophosphamide and thalidomide. Conflict of interest statement. None declared. Fig. 1. CT scan and 3D reconstructed CT scan, as performed by UHL. Fig. 2. The patients jaw as photographed by UHL medical illustrations. Fig. 3. OPG radiograph performed by UHL. Fig. 4. CT scan and 3D reconstructed CT scan, as performed by UHL.

Oncocytic Carcinoma Arising in the Submandibular Gland with Disseminated Bone Metastases

Oncocytic carcinoma of the head and neck is a very rare neoplasm. It usually occurs in the parotid glands. Only 11 cases of oncocytic carcinoma of the submandibular gland have been reported, and no cases have shown distant bone metastasis. A 67-year-old man presented with a tingling sensation in both hands due to a herniated cervical disc. A whole body bone scan and PET-CT showed disseminated bone metastases. Neck CT revealed a 1.7 cm calcified left submandibular mass. The submandibular gland and bone marrow biopsies were consistent with oncocytic carcinoma. Our case is the first report of oncocytic carcinoma of the submandibular gland with disseminated bone metastases.

Tortuous common carotid artery presenting as a pediatric submandibular neck mass

Tortuous common carotid artery presenting as a pediatric submandibular neck mass

Tortuous common carotid artery presenting as a pediatric submandibular neck mass

Summary Aberrant common carotid artery anatomy is a rare cause of a lateral neck mass in pediatric patients. In this article we present the first reported case of a young boy presenting with a pulsatile submandibular mass due to an aberrant common carotid artery. Although there are many causes of carotid artery tortuosity, no specific syndrome or specific diagnosis associated with the arterial changes was ultimately assigned to this child. Though only conservative medical management was elected, this case illustrates the importance of considering abnormal carotid artery anatomy as a rare, but potential cause of neck mass in the pediatric patient.

A Case of Recurrent Ectopic Parathyroid Adenoma Developed 22 Years after the Initial Parathyroidectomy

Persistent or recurrent primary hyperparathyroidism after initi al parathyroid surgery occurs at rates of 1.5~10%. A single missed parathyroid adenoma accounts for the majority of persistent hyperparathyroidism, whereas metachronous parathyroid adenoma is a rare cause of recurrent hypercalcemia. We report a case of a 39-year-old female who presented with recurrent pancreatitis. She had symptoms of hyperparathyroidism such as hypercalcemia, hypophosphatemia, hypercalciuria, nephrocalcinosis, and osteoporosis. She had a 2-cm firm neck mass under the right sub mandibular area. She was diagnosed with primary hyperparathyroidism 22 years ago. At that time, the right upper and lower parathyroid glands were removed after exploration of all parathyroid glands, and a right upper parathyroid adenoma was diagnosed. Now, she had a second surgery to remove the right submandibular mass with intraoperative PTH monitoring, which was diagnosed as a parathyroid adenoma in an ectopic supernumerary parathyroid gland. Because of hungry bone syndrome, she received calcium carbonate replacement therapy and has no evidence of recurrence. Here, we report a recurrent parathyroid adenoma in the undescended, supernumerary parathyroid gland after a long interval from the initial surgery. (J Korean Endocr Soc 24:260~264, 2009)

Thyroid tissue as a submandibular mass: a case report

Ectopic thyroid gland is a rare phenomenon that appears most frequently in the cervical midline and very rarely in other sites such as the submandibular region. This article reports a painless, non-tender and firm thyroid tissue mass in the right submandibular region of a 29-year-old female patient. Physicians should consider ectopic thyroid tissue as a differential diagnosis of submandibular swellings and be aware that this ectopic gland is the only functional thyroid tissue in 70% of cases.

Oncocytic sialolipoma of the submandibular gland with sebaceous differentiation: A new pathological entity

We report the case of an oncocytic sialolipoma of the submandibular gland with sebaceous differentiation in a 73-year-old man. The initial symptom was a right submandibular painless mass. Ultrasonography showed a hypoechoic oval mass posterior to the submandibular gland. The tumorectomy was performed with preservation of the salivary gland. The tumor was composed of mature adipose tissue surrounded by a thin fibrous capsule, multiple nodules of oncocytes, normal ductal-acinar units with focal ductal sebaceous differentiation. We reviewed literature of the reported cases of mixed tumors of the salivary glands composed of mature adipose tissue with oncocytosis, salivary ducts, and acini with sebaceous differentiation. Sialolipoma and lipoadenoma with or without oncocytosis and/or sebaceous differentiation should be considered organ-specific tumors with a distinct histological appearance and specific terminology.

顎下リンパ節腫脹が診断の契機となったサルコイドーシスの1例

Sarcoidosis is a systemic disease of unknown etiology characterized pathologically by noncaseatingepithelioid cell granulomas affecting any organ. It is often associated with bilateral hilar lymphadenopathy, pulmonaryinfiltration, and skin or eye lesions. We report a case of sarcoidosis with submandibular lymphadenopathy.The patient was a 28-year-old woman who presented with a left submandibular painless nodule. Magnetic resonanceimaging（MRI）revealed a submandibular mass with low intensity on T-1 weighted images and high intensityon T-2 weighted images. Under general anesthesia, resection of the lesion was performed. Histologically thespecimen consisted of numerous noncaseating epithelioid cell granulomas. The histological diagnosis was sarcoidosis,and additional examinations were conducted. Ga-67 scintigraphy bilaterally showed increased uptake inthe lacrimal glands, parotid glands, and hilar lymp nodes. Bilateral hilar lymphadenopathy was confirmed on thoracicCT scanning, and bilateral swelling of the parotid gland was confirmed on MRI. The results of the tuberculinskin test were negative, and the serum levels of angiotensin-converting enzymes and γ-globulin were increased.These findings supported the histological diagnosis. Because the patient was asymptomatic, only careful follow-upwas continued, without treatment. During 24 months of follow-up, bilateral swelling of the parotid gland graduallydecreased, and abnormal uptake of Ga-67 disappeared in all lesions.

Nodal and Extranodal Soft Tissue Polymorphous Hemangioendothelioma: A Case Report and Review of the Literature

Polymorphous hemangioendothelioma is a rare vascular tumor of borderline malignant potential and only 10 cases have been described in the literature so far. We report a case of nodal and extranodal polymorphous hemangioendothelioma and review the literature. A 66-year-old man with an unremarkable past medical history was admitted for a left submandibular mass. The main nodule was composed of angiosarcoma-like areas mixed with angiomatous features; some vascular spaces with hobnail endothelium were seen. The tumor involved two adjacent lymph nodes. Immunohistochemically, the neoplastic cells were strongly positive for CD31 and vimentin. After a few months the tumor recurred and the patient was treated with radiation therapy. Polymorphous hemangioendothelioma is a distinct entity in the hemangioendothelioma group with its own clinical and histological features.

An Unusual Case of Luetic Lymphadenitis as a Solitary Submandibular Mass

Syphilis, a chronic systemic infection caused by Treponema pallidum, is usually sexually transmitted and characterized by episodes of active disease interrupted by a latent period. After an incubation period of averaging 2-6 weeks, a primary syphilis with a firm, non-tender chancre appears, often associated with regional lymphadenopathy. Secondary syphilis shows localized or diffuse mucocutaneous lesions and generalized non-tender lymphadenopathy. In about one-third of untreated cases, the tertiary stage appears, characterized by progressive destructive mucocutaneous, musculoskeletal or parenchymal lesions, aortitis or symptomatic central nervous system diseases. Without serological testing, the precise diagnosis is very difficult. In fact, it has often been called the great imitator because it was often confused with other disease. We report a very unusual case of luetic lymphadenitis presented as a solitary submandibular mass.

Idiotype Vaccination of Untreated Indolent B-Cell Lymphoma: Durable Objective Remissions and Association of Superior Outcome with Cellular Immune Responses

Idiotype vaccination refers to active immunization of B-cell lymphoma (B-NHL) patients with the clonal immunoglobulin (Ig) expressed by the tumor cells. After systemic cytoreductive therapy, idiotype vaccination has been shown to induce specific cellular and humoral immune responses; and humoral responses in particular are associated with prolonged remission and encouraging survival rates. Conventional idiotype vaccines are composed of the entire lymphoma-derived Ig coupled to the immunogenic carrier KLH and are administered subcutaneously with adjuvant. We have developed a idiotype production strategy based on bacterial expression of the lymphoma-derived idiotype as a recombinant Fab fragment (Bertinetti et al., EJH 2006). Intradermal administration of this antigen with lipid-based adjuvant and subcutaneous coadministration of GM-CSF had excellent immunogenicity in a phase I trial of advanced, heavily pretreated B-NHL patients (Bertinetti et al., Cancer Research 2006). In a subsequent phase II trial, 20 patients with untreated indolent B-NHL (14 follicular [FL], 3 nodal marginal zone [nMZL], 3 mantle cell [MCL]) and without immediate need for cytoreduction received at least 6 monthly idiotype vaccinations. No grade IV toxicities were seen, and the sole case of grade III toxicity, generalized erythema, did not preclude completion of the vaccination schedule. Prior to vaccination, 5/19 patients (26%) had decreased CD4+ and 6/19 patients (31%) low CD8+ T cells counts. Furthermore, 10/12 anti-HbS-negative patients (83%) failed to mount a detectable immune response to a conventional hepatitis B vaccine administered concomitantly to idiotype vaccination. Despite this functional immunodeficiency, 12/18 analyzed patients (66%) developed a cellular immune response to idiotype as detected by enumeration of IFNgamma-secreting cells by DC-ELISpot. The ELISpot protocol was validated by blinded interlaboratory testing (www.cimt.de). The frequency of idiotype-responding T cells increased from the 2nd to the 6th vaccination and could be effectively boostered by maintenance immunization in 3-monthly intervals. In vitro stimulation of PBMC from responding patients with idiotype induced specific proliferation of CD4+ T-cells and a shift towards a Th1 response in post-vaccination samples. In addition, 8/18 analyzed patients (44%) developed anti-idiotype IgG or IgM antibodies as assessed by ELISA, and the combined immune response rate was 85%. After a median follow-up of 34 months, 8 patients (40%) are progression-free, and 10 (50%) did not require cytoreductive therapy. Cellular immune responses were associated with superior PFS (p<0.05), and 5 of 6 non-responders eventually required cytoreductive therapy. Humoral immune responses were not related to PFS. Six patients (30%; only FL or nMZL) achieved an objective partial remission, including near-complete disappearance of a large submandibular mass and one subcutaneous lymphoma mass. All objective responders developed specific cellular immunity, but only 4 anti-idiotype antibodies. Five patients are in continuing remission for 12–49 months. Intradermal immunization with the chosen idiotype formulation has excellent immunogenicity despite a severely impaired immune function in untreated B-NHL patients. Furthermore, this is the first active immunotherapy trial showing objective and durable lymphoma responses in first-line therapy at a higher rate than expected for spontaneous remissions. In this setting, and in contrast to conventional idiotype vaccination schedules, cellular anti-idiotype immunity may play a crucial role for a favorable clinical outcome. Since passive humoral anti-lymphoma immunity can be easily transferred by infusions of commercially available monoclonal antibodies, synergistic benefit may be envisioned for an initial vaccination course aimed to prime anti-idiotype T-cells combined with subsequent passive immunotherapy.

Acute Kidney Injury and Proteinuria in a Patient With Diabetes and a Submandibular Mass

Acute Kidney Injury and Proteinuria in a Patient With Diabetes and a Submandibular Mass

Nodular fasciitis in the submandibular gland

Nodular fasciitis (NF) is a benign, proliferative lesion of myofibroblasts. The most common site of the lesion is in the upper extremities or trunk. NF in the head and neck is next in frequency and is the most common site in infants and children. In adults, NF in the submandibular region is very rare. We report a case of a 53-year-old man with a submandibular mass, rapidly growing for 10 days. Cytologic findings revealed a few sheets or clusters of small, uniform spindle cells. The uniform spindle cells had centrally located round to ovoid nucleus, but there were no nuclear atypia and atypical mitosis. There were focally loose stroma but we could not find chondroid or myxoid components. A partial parotidectomy was performed. We describe a case of NF in the submandibular region, adjacent to the parotid gland and review the clinical, cytologic, histologic, and immunohistochemical features of NF.

Aplasia of the submandibular gland with compensatory sublingual gland hypertrophy: A case report

Aplasia of the submandibular gland is an uncommon disorder which has been previously reported in the literature in few case reports. We report a 14-year-old boy presenting with submandibular mass. He was later diagnosed as having aplasia of the submandibular gland and a compensatory ipsilateral sublingual hypertrophy. The diagnosis was made by imaging (CT-scan and MR). The case is presented with the emphasis that aplasia of the submandibular gland followed by a presumably compensatory sublingual gland enlargement is a rare phenomenon but still should be considered in the differential diagnosis of a neck mass.