Plunging ranula is rare and manifests as the submandibular or submental cystic mass, without intra-oral abnormality. It usually causes misdiagnosis and malpractice. This article reported a case of plunging ranula that appeared as a massive mass located behind the left ear. A 6-year-old child presented with recurrent left retroauricular swelling over six months without obvious inducement. The mass showed repeatedly swelling and persistent skin ulcers. Physical examinations described a fluctuant, nontender mass behind the left ear whose size was about 5 cm×5 cm×3 cm. The skin upon the surface of the mass was thin and red, and overflowed yellow slimelike contents. Computed tomography (CT) showed an extensive cystic lesion in the left neck. After local incision and drainage the mass was shrink. With the primary clinical diagnosis of branchial cyst, the patient underwent mass resection through postauricular incision. During the operation, the fistula was traced to the area around the sublingual gland, and the postoperative pathology report demonstrated cystic spaces occurring in soft tissue without lymphoid tissue. One month postoperatively, the patient presented the "egg-white", wire-drawing transparent viscous fluid outflowing from the left external auditory canal, indicating that the fluid in the external auditory canal originated from the sublingual gland and the disease was the plunging ranula presenting as a giant left retroauricular mass. We readmitted the patient to the hospital and the ipsilateral sublingual gland was completely removed in the mouth under general anesthesia. No clinical evidence of recurrence was found at the end of 9 months follow-up. So far, there is no literature reporting plunging ranula behind ear. This case report describes the clinical manifestation, diagnosis, and differential diagnosis and treatment of a case of plunging ranula, and reveals that, other than the submandibular or submental, plunging ranula also could be found in retroauricular region. The plunging ranula is difficult to fully confirm through a clinical character, and usually easy to be misdiagnosed as other tumors, like lymphatic malformation, venous malformations, etc. Radiographic examination and/or puncture fluid observation are required for identification. "Egg-white", wire-drawing mucous secretion is the most important characteristic in clinical diagnosis of sublingual gland cyst. However, if sublingual gland resection, i.e. removal of the source of cyst fluid is performed, a good prognosis can be achieved.
Child ; Humans ; Neoplasm Recurrence, Local ; Ranula ; Salivary Gland Diseases ; Sublingual Gland ; Tomography, X-Ray Computed

OBJECTIVE: To evaluate the secretory function of parotid glands by dynamic magnetic resonance (MR) sialography and determine the clinical performance of this technique in diagnosing and evaluating Sjögren's syndrome (SS) patients. MATERIALS AND METHODS: This study enrolled 29 healthy volunteers (25 women and 4 men; mean age, 34.8 ± 6.3 years; age range, 26–47 years) and 25 primary SS (pSS) patients (23 women and 2 men; mean age, 37.7 ± 7.9 years; age range, 25–50 years) with decreased secretory function. The volume of the parotid gland ducts was precisely measured for both groups at single pre- and 6 post-gustatory-stimulated phases. Time-dependent volume change ratio curves were generated, four parameters were derived from the curves: the slope of the increase in the first post-stimulation phase (slope(1st)), the peak value, the time-to-peak, the total saliva secretion post-stimulation. All values were used to quantitatively evaluate the secretory function of the parotid gland. The repeated measurement analysis, Mann-Whitney U test and receiver operating characteristic curve were applied. RESULTS: Time-dependent volume change ratio curves demonstrated that there is a statistically significant difference between the two groups (F = 8.750; p = 0.005). A quickly increasing curve was shown in the volunteer group, whereas a slowly increasing curve was shown in the pSS patient group. The slope(1st), peak value and total saliva secretion post-stimulation of the patient group were significantly lower than those of the volunteer group (p = 0.005, p = 0.003, and p = 0.002, respectively). The time-to-peak between the two groups was not significantly different (p = 0.383). The slope(1st) can be used as a discriminator to diagnose SS patients (p = 0.015; odds ratio = 4.234; area under the curve = 0.726). CONCLUSION: Dynamic MR sialography is proven to be an effective method in evaluating salivary gland function and has a great potential in diagnosing and evaluating pSS patients.
Autoimmune Diseases ; Female ; Healthy Volunteers ; Humans ; Male ; Methods ; Odds Ratio ; Parotid Gland ; ROC Curve ; Saliva ; Salivary Glands ; Salivation ; Sialadenitis ; Sialography ; Volunteers

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

BACKGROUND: The conventional transcervical resection for submandibular gland disease has some risks and an unsatisfactory cosmetic result. Recently, robot-assisted surgery has been developed as a plausible substitute for conventional surgery which provides an excellent cosmetic outcome. CASE PRESENTATION: The authors performed robot-assisted sialadenectomy via modified facelift incision using the da Vinci Xi surgical system (Intuitive Surgical Inc., CA, USA) with two endowrist arms (monopolar curved scissors and Maryland bipolar forceps) successfully in a 44-year-old female patient who suffered from sialolith and severe atrophic submandibular gland. CONCLUSIONS: If similar studies are done in the future, this robot-assisted sialadenectomy may become established as an alternative to existing disadvantageous surgical methods.
Adult ; Arm ; Female ; Humans ; Maryland ; Rhytidoplasty* ; Salivary Gland Calculi ; Submandibular Gland Diseases ; Submandibular Gland*

PURPOSE: Although Sjögren's syndrome (SS) is the most common disease causing xerostomia, autoimmune thyroid diseases can also affect the salivary glands. The aim of our study was to estimate the prevalence of thyroid diseases (TD) in subjects with symptoms of xerostomia and evaluate the efficacy of salivary gland scintigraphy (SGS) in the detection of TD in patients with SS and without SS.METHODS: We retrospectively reviewed the SGS findings of 173 subjects (men:women, 29:144) with symptoms of xerostomia. Ejection fractions (EF) in the parotid and submandibular glands were calculated. Thyroid disease was diagnosed on the basis of the results of the visual assessment of tracer uptake in the thyroid gland on SGS images as well as serological thyroid function tests.RESULTS: Based on the American-European Criteria, 94 patients were diagnosed with SS. Hashimoto's thyroiditis was diagnosed in 63 patients, subacute thyroiditis in 23, subclinical hypothyroidism in five, and Graves' disease in one. There were significant differences in the EF values of the parotid and submandibular glands between patients with TD and those with undetermined diagnoses.CONCLUSIONS: More than half of patients with xerostomia exhibited TD. Thyroid assessment by SGS is feasible, and SGS appears to be useful for the patients with xerostomia caused by TD. SGS may be the first imaging modality capable of evaluating both salivary gland function and thyroid gland status in patients with xerostomia. This strategy would make the requirement for additional workup for thyroid disease.
Diagnosis ; Graves Disease ; Humans ; Hypothyroidism ; Prevalence ; Radionuclide Imaging ; Retrospective Studies ; Salivary Glands ; Submandibular Gland ; Thyroid Diseases ; Thyroid Function Tests ; Thyroid Gland ; Thyroiditis ; Thyroiditis, Subacute ; Xerostomia

PURPOSE: To investigate the difference of expression of autophagy and reactive oxygen species (ROS) related proteins in adenoid cystic carcinoma (ACC) of lacrimal gland in comparison with ACC of salivary gland. MATERIALS AND METHODS: Formalin-fixed, paraffin-embedded tissue samples from patients pathologically diagnosed as lacrimal gland ACC (n=11) and salivary gland ACC (n=64) were used. Immunochemistry was used to measure expression of autophagy related proteins [beclin-1, light chain (LC) 3A, LC3B, p62, and BCL2/adenovirus E1B 19 kDa protein-interacting protein 3 (BNIP3)] and ROS related proteins [catalase, thioredoxinreductase, glutathione S-transferasepi (GSTpi), thioredoxin interacting protein, and manganese superoxide dismutase (MnSOD)]. The prognostic factors related to disease-free and overall survival (OS) in lacrimal gland ACC by log-rank tests, were determined. RESULTS: GSTpi in stromal cells was more highly expressed in lacrimal gland ACC (p=0.006), however, MnSOD in epithelial cells was expressed more in salivary gland ACC (p=0.046). LC3B positivity and BNIP3 positivity in epithelial component were associated with shorter disease-free survival (both p=0.002), and LC3A positivity in stromal component was the factor related to shorter OS (p=0.005). CONCLUSION: This is the first study to demonstrate the expression of autophagy and ROS related proteins in lacrimal gland ACC in comparison with the salivary gland ACC, which would provide a basis for further study of autophagy and ROS mechanism as novel therapeutic targets in lacrimal gland ACC.
Adult ; Aged ; *Autophagy ; Beclin-1 ; Biomarkers, Tumor/metabolism ; Carcinoma, Adenoid Cystic/*metabolism/pathology ; Carrier Proteins ; Disease-Free Survival ; Female ; Gene Expression Regulation, Neoplastic ; Humans ; Lacrimal Apparatus/metabolism/pathology ; Lacrimal Apparatus Diseases/*metabolism/pathology ; Male ; Membrane Proteins ; Middle Aged ; Proto-Oncogene Proteins ; Reactive Oxygen Species/*metabolism ; Salivary Gland Neoplasms/metabolism/*pathology ; Salivary Glands/pathology

BACKGROUNDImmunoglobulin G4-related disease (IgG4-RD) is a newly recognized systemic disease that can involve multiple organs and various clinical phenotypes. The purpose of this study was to analyze different types of organ involvement in IgG4-RD patients in China.

METHODSWe conducted a prospective cohort study on IgG4-RD patients to analyze the clinical manifestations and rare features of IgG4-RD. Patients were grouped into different types according to organ involvement regarding organ number and organ site. The constituent ratio in different types was also analyzed.

RESULTSA total of 200 IgG4-RD patients, with a male:female ratio of 2.08:1, were grouped into different types. Cases having involvement of two or three organs were the most common whereas the fewest number of patients had multi-organ (≥4) involvement. Serum IgG4 and IgE levels, IgG4/IgG ratio, and percentage of eosinophils increased as the number of involved organs increased. In addition, constituent ratio analysis revealed that patients with salivary gland/lacrimal gland swelling, who also constituted the largest number of IgG4-RD patients, had higher serum IgG4 concentrations and IgG4/IgG values, had higher percentage of Eos, and were more likely to have had a history of allergies relative to patients with internal organ involvement.

CONCLUSIONSThe characteristic feature of IgG4-RD is multiple organ involvement with various clinical manifestations and different types. Although serum IgG4 levels increased with the number of involved organs, serum IgG4 levels were higher for those patients with salivary gland/lacrimal gland swelling compared with those with internal organ involvement. Thus, valuable clues to the differential diagnosis of IgG4-RD could be obtained by examining the clinical patterns of organ involvement.

Adolescent ; Adult ; Aged ; Aged, 80 and over ; Autoimmune Diseases ; complications ; pathology ; Eosinophils ; metabolism ; Female ; Humans ; Immunoglobulin E ; blood ; Immunoglobulin G ; blood ; Kidney ; pathology ; Lacrimal Apparatus ; pathology ; Lung ; pathology ; Male ; Middle Aged ; Pituitary Gland ; pathology ; Prospective Studies ; Prostate ; pathology ; Salivary Glands ; pathology ; Thyroid Gland ; pathology ; Uterus ; pathology ; Young Adult

Since IgG4-related pancreatitis was first reported in 2001, IgG4-related disease has been identified in other organs such as salivary gland, gallbladder, thyroid, retroperitoneum and kidney; but lung invasion is rare. A 63-year-old man presented with hemoptysis at the pulmonary clinic and chest computed tomography revealed about 4.1 cm irregular shaped mass with spiculated margin at the left upper lobe. Despite no elevation of serum IgG4 level, he was finally diagnosed as IgG4-related lung disease by transthoracic needle biopsy. After treatment with oral glucocorticoids, hemoptysis disappeared and the size of lung mass was decreased.
Biopsy, Needle ; Gallbladder ; Glucocorticoids ; Hemoptysis ; Humans ; Immunoglobulin G* ; Immunoglobulins ; Kidney ; Lung Diseases* ; Lung* ; Middle Aged ; Pancreatitis ; Salivary Glands ; Thorax ; Thyroid Gland

Mucosa-associated lymphoid tissue (MALT) lymphomas are low-grade extranodal B-cell lymphomas that may involve various sites in the head and neck including the thyroid, salivary, and lacrimal glands. In particular, primary salivary gland MALT lymphoma is rare, with sublingual gland tumors especially rarer and frequently malignant. In Korea, there has been no reported case of MALT lymphoma in sublingual glands. Here we report a case of a MALT lymphoma in the sublingual gland.
Autoimmune Diseases* ; Head ; Korea ; Lacrimal Apparatus ; Lymphoid Tissue ; Lymphoma ; Lymphoma, B-Cell ; Lymphoma, B-Cell, Marginal Zone* ; Neck ; Salivary Glands ; Sublingual Gland* ; Thyroid Gland