OBJECTIVES

To report a case of triple primary malignant neoplasms in a 70-year-old woman diagnosed with follicular and papillary thyroid carcinoma and diffuse B-cell lymphoma of the right submandibular gland and cervical lymph nodes.

METHODS

Design:Case Report

Setting:Tertiary Government Training Hospital

Patient: One

RESULTS

A 70- year-old woman presented with a four-year history of gradually enlarging anterior neck mass, associated with a right submandibular mass and neck nodes for one year. The gradual progression of her symptoms made the patient think that it was a benign condition. This led to a delay in medical consultation. The patient underwent total thyroidectomy with functional neck dissection of the ipsilateral right neck. Histopathology revealed simultaneous follicular and papillary thyroid carcinoma, and diffuse B-cell lymphoma of the cervical lymph nodes. The patient was referred to medical oncology and nuclear medicine for further management.

CONCLUSION

Our patient was incidentally diagnosed with follicular and papillary thyroid carcinoma and diffuse B cell lymphoma of the cervical lymph nodes after surgery. Such triple primary malignant neoplasms in a single individual are rare, and as in our case, may only be diagnosed in hindsight.

Human ; Female ; Aged: 65-79 Yrs Old ; Carcinoma ; B-lymphocytes ; Adenocarcinoma, Follicular ; Neoplasms ; Submandibular Gland ; Lymph Nodes ; Lymphoma, B-cell ; Thyroid Gland ; Thyroid Cancer, Papillary ; Thyroidectomy

OBJECTIVE: To evaluate the clinical outcomes and explore the application of partial sialoadenectomy for the treatment of benign tumors in the submandibular gland (SMG). METHODS: Patients with pleomorphic adenoma of the SMG who underwent surgical treatment in the Department of Oral and Maxillofacial Surgery, Peking University Shenzhen Hospital, from October 2017 to February 2021, were enrolled and assessed in the follow-up. Fifteen patients underwent partial sialoadenectomy (PS group), and 18 patients underwent total sialoadenectomy (TS group). Postoperative salivary secretion, degree of dry mouth, appearance changes of the face and neck, nerve damage, and tumor recurrence were compared between the groups. The volume of the glands on the operated and contralateral sides of the patients in the PS group, the saliva flow rate, and their correlations, were also analyzed. RESULTS: There was no recurrence during the follow-up period. The whole saliva flow rate at rest in the PS group was higher than that in the TS group [(2.15±1.10) g/5 min vs. (1.35±0.97) g/5 min, t=2.208, P=0.035)], while the stimulated saliva flow rate was not significantly different. The objective feeling of dry mouth, evaluated by visual analogue scale (VAS) score, was more obvious in the TS group than in the PS group (Z=-2.244, P=0.025). In the PS group, the resting saliva flow rate of the SMG on the operated side was lower than that on the contralateral side of the same patient [(0.92±0.40) g/5 min vs. (1.18±0.40) g/5 min, t=-2.821, P=0.014], however, in the cases whose remaining SMG was more than 80% of the contralateral side, the saliva flow rate of both sides was not significantly different (t=-0.027, P=0.980). There was no significant difference in the saliva flow rate per unit volume of the gland on either side (t=-0.015, P=0.989), and the saliva flow rate of the operated SMG was positively correlated with the volume of the remaining gland (r=0.750, P=0.012). The VAS scores for neck deformity were not significantly different between the two groups (t=-0.997, P=0.319). No symptoms of nerve injury occurred in either group. CONCLUSION Partial sialoadenectomy in the SMG can safely remove benign tumors while preserving glandular secretory function, with fewer complications and improved quality of life.
Humans ; Submandibular Gland/pathology* ; Male ; Female ; Middle Aged ; Adenoma, Pleomorphic/surgery* ; Adult ; Treatment Outcome ; Submandibular Gland Neoplasms/surgery* ; Saliva/metabolism* ; Aged

Endoscopy ; Humans ; Neoplasms/surgery* ; Submandibular Gland/surgery* ; Thyroidectomy

The saliva secreted from submandibular gland (SMG) accounts for 60%-65%. It plays an important role in maintaining the human function of swallow, digestion, testing, speech, protection of oral mucosa, and prevention from dental carries. The SMG is frequently resected during the treatment for various kinds of oral and maxillofacial diseases, resulting in xerostomia and decreased quality of life. During the past 15 years, Research Center of Salivary Gland Diseases in Peking University School and Hospital of Stomatology conducted a series of studies on new techniques for preservation of SMG and achieved remarkable results. The clinicopathologic and imaging characteristics of IgG4-related sialadenitis (IgG4-RS) were clarified based on systematic studies. The results of studies on the pathogenesis of IgG4-RS showed that unbalance of inflammatory factors mediated the abnormality of secretion of SMG. IL-4 participates in occurring and development of glandular fibrosis of SMG. Regulation of tumor necrosis factor α (TNF-α) and cleaning of senescent cells might be taken as the targets for treatment of IgG4-RS. The combination of glucocorticoid and steroid-sparing agents showed effective results for treating IgG4-RS, clinical remission was achieved in all the patients, serum IgG4 levels decreased, and salivary gland secretion significantly increased. Sialoendoscopy-assisted sialolithectomy was applied in the treatment of about 1 000 cases with submandibular hilar calculi with a success rate of more than 90%. Transfer of SMG was used for prevention from radiation-induced xerostomia in the patients with head and neck carcinoma. SMG was transferred to the submental region before radiotherapy and was kept away from the ra-diation field. The results of prospective clinical controlled study showed this technique could effectively preserve the function of SMG and prevent from xerostomia. Based on the micro-anatomical study on the blood vessels and ducts of SMG, partial sialoadenectomy was applied for treatment of benign tumors in the SMG. A clinical controlled study confirmed its safety for control of the tumors and its advantage of preservation of SMG function. The studies on the involvement of SMG in oral squamous cell carcinoma (OSCC) provided the anatomical and histopathological basis for preservation of SMG during neck dissection for early cases with OSCC. A innovated surgical modality "four preservations including SMG" was used during the neck dissection for the early cases with OSCC. A prospective randomized clinical controlled study confirmed its safety, feasibility, effectiveness for control of the carcinoma, and advantages of preservation of SMG function.
Humans ; Carcinoma, Squamous Cell/pathology* ; Glucocorticoids ; Immunoglobulin G ; Interleukin-4 ; Mouth Neoplasms/pathology* ; Prospective Studies ; Quality of Life ; Sialadenitis/surgery* ; Submandibular Gland/surgery* ; Tumor Necrosis Factor-alpha ; Xerostomia/prevention & control* ; Randomized Controlled Trials as Topic

OBJECTIVE: To measure the weight and volume of normal submandibular gland by in vitro detection of glandular body, to compare with the measurement values of CT volume rendering to evaluate the consistency of the two methods, and to explore the related factors affecting the weight and volume of the submandibular gland. METHODS: The patients who underwent neck dissection due to oral cancer and were confirmed with normal submandibular gland by postoperative histopathology in Peking University School and Hospital of Stomatology from May 2019 to January 2020 were collected in this study and divi-ded into 4 groups according to the age standards raised by the Society of Geriatrics Chinese Medical Association and WHO (2000) age standards. The submandibular gland was carefully dissected along with the capsule immediately after neck dissection. The weight and volume of submandibular gland were accurately measured by an integrated aseptic measuring device. The volume of submandibular gland was measured by CT volume rendering before operation and the consistency of the two methods was evaluated. The 95% confidence interval of the weight and volume of submandibular gland in the different groups of Chinese people, which were divided by gender and age, were calculated, and the correlation with age, gender, height, weight, body mass index (BMI) and other factors was analyzed. RESULTS: The weight and vo-lume of submandibular gland were measured in 220 subjects. The average weight and volume of submandibular gland were (11.69±2.45) g and (11.55±2.41) cm³ respectively. The volume of submandibular gland measured by CT volume rendering ranged from 70% to 82% of that measured by in vitro detection of glandular body, and the correlation coefficient between the two methods was 0.976 (P < 0.05). The gender difference of weight and volume of submandibular gland existed in the groups of youth, middle-aged, young elderly and elderly groups, the values of males were obviously higher than those of females (P < 0.05). There was no gender difference of weight and volume of submandibular gland in the adolescent group (P > 0.05). The weight and volume of submandibular gland had a strong positive correlation with body height (P < 0.05), a weak positive correlation with body weight (P < 0.05), and no correlation with BMI (P > 0.05). The female's weight and volume of submandibular gland in the young elderly and elderly groups were significantly lower than those in the other three groups (P < 0.05). CONCLUSION The normal reference range of the weight and volume of submandibular gland in vitro were established in different age and gender groups of Chinese people. The volume of submandibular gland mea-sured by CT volume rendering was 70%-82% of that measured by in vitro detection of glandular body. The results of the two methods had a high degree of consistency. The weight and volume of submandibular gland were related to age, gender, body height and body weight.
Adolescent ; Adult ; Aged ; Body Mass Index ; Body Weight ; Female ; Humans ; Male ; Middle Aged ; Mouth Neoplasms ; Neck Dissection ; Submandibular Gland/diagnostic imaging*

Immunoglobulin G4-related sialadenitis (IgG4-RS) is a newly recognized immune-mediated disease and one of immunoglobulin G4-related diseases (IgG4-RD). Our multidisciplinary research group investigated the clinicopathological characteristics and diagnosis of IgG4-RS during the past 10 years. Clinically, it showed multiple bilateral enlargement of major salivary glands (including sublingual and accessory parotid glands) and lacrimal glands. The comorbid diseases of head and neck region including rhinosinusitis, allergic rhinitis, and lymphadenopathy were commonly seen, which could occur more early than enlargement of major salivary glands. Internal organ involvements, such as autoimmune pancreatitis, sclerosing cholangitis, and interstitial pneumonia could also be seen. Thirty-five (38.5%) patients had the symptom of xerostomia. Saliva flow at rest was lower than normal. Secretory function was reduced more severely in the submandibular glands than in the parotid glands. Serum levels of IgG4 were elevated in almost all the cases and the majority of the patients had increased IgE levels. CT, ultrasonography, and sialography showed their imaging characteristics. Histologically it showed marked lymphoplasmacytic inflammation, large irregular lymphoid follicles with expanded germinal centers, prominent cellular interlobular fibrosis, eosinophil infiltration, and obliterative phlebitis. Their immunohistological examination showed marked IgG-positive and IgG4-positive plasma cell infiltration and high IgG4/IgG ratio. The disease could be divided into three stages according to severity of glandular fibrosis. The serum IgG4 level was higher and the saliva secretion lower as glandular fibrosis increased. IgG4-RS should be differentiated from other diseases with enlargement of major salivary gland and lacrimal gland, such as primary Sjögren syndrome, chronic obstructive submandibular sialadenitis, and eosinophilic hyperplastic lymphogranuloma.
Autoimmune Diseases ; Humans ; Immunoglobulin G ; Sialadenitis ; Sjogren's Syndrome ; Submandibular Gland

Necrobiotic xanthogranulomatous reaction is a multiorgan, non-Langerhans cell histiocytosis with an unknown etiology. Occurrence in the salivary gland is extremely rare. We recently identified a case of necrobiotic xanthogranulomatous sialadenitis in a 73-year-old Korean woman who presented with a painless palpable lesion in the chin. There was no accompanying cutaneous lesion. Partial resection and subsequent wide excision with neck dissection were performed. Pathological examination showed a severe inflammatory lesion that included foamy macrophages centrally admixed with neutrophils, eosinophils, lymphocytes, plasma cells, and scattered giant cells, as well as necrobiosis. During the 12-month postoperative period, no grossly remarkable change in size was noted. Necrobiotic xanthogranulomatous inflammation may be preceded by or combined with hematologic malignancy. Although rare, clinicians and radiologists should be aware that an adhesive necrobiotic xanthogranuloma in the salivary gland may present with a mass-like lesion. Further evaluation for hematologic disease and close follow-up are needed when a pathologic diagnosis is made.
Adhesives ; Aged ; Chin ; Diagnosis ; Eosinophils ; Female ; Follow-Up Studies ; Giant Cells ; Hematologic Diseases ; Hematologic Neoplasms ; Histiocytosis ; Humans ; Inflammation ; Lymphocytes ; Macrophages ; Neck Dissection ; Necrobiotic Disorders ; Necrobiotic Xanthogranuloma ; Neutrophils ; Plasma Cells ; Postoperative Period ; Salivary Glands ; Sialadenitis ; Skin ; Submandibular Gland

Severe dry eye is a refractory ophthalmologic disease. Our multidisciplinary research group treated severe dry eye by microvascular autologous transplantation of submandibular gland (SMG) during the past 20 years. The SMG, with its blood vessels and Wharton's duct, was harvested from the submandibular triangle and transferred to the temporal area. The blood vessels in the SMG were anastomosed with the temporal blood vessels using a microsurgical technique. Then, the distal end of Wharton's duct was sutured to form an opening in the upper lateral conjunctival fold. The tear was replaced by the secretion of the transplanted SMG to lubricate the ocular surface. In our study, the surgical techniques of blood vessel management were continuously modified to increase the survival rate of the transplanted SMG. A novel surgical modality of partial transplantation of SMG was established to prevent postoperative epiphora. A clinical study with the largest case number in the world was conducted and the effectiveness of transplantation of SMG for severe dry eye was fully confirmed. In order to resolve two main clinical problems including ductal obstruction resulted from low secretion rate during the latent period, and epiphora due to over secretion of the transplanted SMG in the later term of transplantation, the regulation of the secretion mechanism of the normal and transplanted SMG were investigated. New opinions on mechanisms of saliva secretion were provided. Based on the priniciple of translational medicine, the results of related basic research were applied in the clinic. The clinical guidelines for secretion regulation of transplanted SMG were established. A concept of chronic obstructive sialadenitis of transplanted SMG was provided and its diagnostic criteria, diagnostic technique of sialography, and therapeutic regimen were established. As a result, the surgical success rate was obviously elevated, the surgical complications were decreased, and life quality of the patients was greatly improved.
Humans ; Lacrimal Apparatus Diseases/therapy* ; Salivary Ducts ; Submandibular Gland/transplantation* ; Tears ; Transplantation, Autologous

Sebaceous carcinoma of the salivary gland is an extremely unordinary malignancy occurring mainly in the parotid gland. Sebaceous carcinoma arising in the submandibular gland is exceptional and only three cases have been reported in the literature. We herein present a unique case of sebaceous carcinoma of the submandibular gland, the first to be reported in literature in South Korea. An 85-year-old man visited our hospital complaining of a progressively enlarging submandibular mass with tenderness and ipsilateral facial palsy. We performed a wide excision of the mass with superficial parotidectomy and modified neck dissection. Microscopic and immunohistochemical examinations demonstrated sebaceous carcinoma of low grade differentiation. Further adjuvant radiotherapy was rejected and the patient was followed up for five months. The optimal treatment of sebaceous carcinoma of the salivary gland is still not established due to the rarity of the disease. When more cases are reported, the clinicopathologic characteristics will be better understood.
Aged, 80 and over ; Facial Paralysis ; Humans ; Korea ; Neck Dissection ; Parotid Gland ; Radiotherapy, Adjuvant ; Salivary Gland Neoplasms ; Salivary Glands ; Submandibular Gland Neoplasms ; Submandibular Gland*

OBJECTIVE: To investigate the value of diffusion-weighted magnetic resonance imaging (DW-MRI) as a noninvasive tool to assess salivary gland function for follow-up of patients with radiation-induced xerostomia. MATERIALS AND METHODS: This study included 23 patients with nasopharyngeal carcinoma who had been treated with parotid-sparing radiotherapy (RT). Salivary function was assessed by DW-MRI pre-treatment and one week and one year post-RT, respectively. The maximum apparent diffusion coefficient (ADC) of parotid glands (pADCmax) and the time to peak ADC of parotid glands (pTmax) during stimulation were obtained. Multivariate analysis was used to analyze factors correlated with the severity of radiation-induced xerostomia. RESULTS: The ADCs of parotid and submandibular glands (1.26 ± 0.10 × 10−3 mm2/s and 1.32 ± 0.07 × 10−3 mm2/s pre-RT, respectively) both showed an increase in all patients at one week post-RT (1.75 ± 0.16 × 10−3 mm2/s, p < 0.001 and 1.70 ± 0.16 × 10−3 mm2/s, p < 0.001, respectively), followed by a decrease in parotid glands at one year post-RT(1.57 ± 0.15 × 10−3 mm2/s, p < 0.001) but not in submandibular glands (1.69 ± 0.18 × 10−3 mm2/s, p = 0.581). An improvement in xerostomia was found in 13 patients at one year post-RT. Multivariate analysis revealed 4 significant predictors for the improvement of xerostomia, including dose to parotid glands (p = 0.009, odds ratio [OR] = 0.639), the ADC of submandibular glands (p = 0.013, OR = 3.295), pADCmax (p = 0.024, OR = 0.474), and pTmax (p = 0.017, OR = 0.729) at one week post-RT. CONCLUSION: The ADC value is a sensitive indicator for salivary gland dysfunction. DW-MRI is potentially useful for noninvasively predicting the severity of radiation-induced xerostomia.
Diffusion ; Follow-Up Studies* ; Head and Neck Neoplasms ; Humans ; Magnetic Resonance Imaging* ; Multivariate Analysis ; Odds Ratio ; Parotid Gland ; Radiotherapy ; Salivary Glands* ; Submandibular Gland ; Xerostomia*