Salivary Gland Disorders: Rapid Evidence Review.

The major salivary glands are the paired parotid, submandibular, and sublingual glands. Salivary gland disorders can affect the glandular tissue or its excretory system. The parotid glands are the largest and produce aqueous serous secretions that are less immunogenic. They are more susceptible to infections and neoplasms. The submandibular glands produce mucinous secretions that are high in calcium and phosphate salts through a long submandibular duct that flows against gravity. The submandibular glands are responsible for more than 80% of salivary stones. Sialadenitis can be acute or chronic and caused by bacterial, viral, and obstructive etiologies; the most common bacteria is Staphylococcus aureus. The most common viral etiologies in children are mumps (globally) and juvenile recurrent parotitis (in vaccinated populations). Sialadenosis is a chronic asymptomatic enlargement of the salivary glands due to systemic disease. Sialolithiasis causes up to 50% of salivary gland disorders. It is associated with salivary stasis and inflammation caused by dehydration, malnutrition, medications, or chronic illness. Obstruction is also caused by trauma, stenosis, and mucoceles. Neoplasms are rare and typically benign, but they warrant referral and imaging with ultrasonography, computed tomography, or magnetic resonance sialography. Most disorders are managed with conservative measures by treating the underlying etiology, optimizing predisposing factors, controlling pain, and increasing salivary flow with sialagogues, hydration, massage, warm compresses, oral hygiene, and medication adjustment. Sialendoscopy is a gland-sparing technique that can treat obstructive and nonobstructive disorders. (Am Fam Physician. 2024;109(6):550-559.

Chronic Phenotypes Underlying Radiation-Induced Salivary Gland Dysfunction.

Head and neck cancer (HNC) is the sixth most diagnosed cancer, and treatment typically consists of surgical removal of the tumor followed by ionizing radiation (IR). While excellent at controlling tumor growth, IR often damages salivary glands due to their proximity to common tumor sites. Radiation damage to salivary glands results in loss of secretory function, causing severe and chronic reductions in salivary flow. This leads to the patient-reported sensation of dry mouth, termed xerostomia, which significantly reduces quality of life for HNC patients and survivors. The mechanisms underlying salivary gland damage remain elusive, and therefore, treatment options are scarce. Available therapies provide temporary symptom relief, but there is no standard of care for permanent restoration of function. There is a significant gap in understanding the chronic mechanistic responses to radiation as well as treatments that can be given in the months to years following cessation of treatment. HNC cases are steadily rising; particularly, the number of young patients diagnosed with nonfatal human papillomavirus + HNC continues to increase. The growing number of HNC diagnoses and improved prognoses results in more people living with xerostomia, which highlights the mounting need for restorative treatments. Mechanisms underlying chronic damage include decreases in acinar differentiation markers, increases in acinar cell proliferation, immune and inflammatory dysregulation, and metabolic changes including increases in amino acids and reductions in glycolysis and oxidative phosphorylation, fibrosis, and dysregulated neuronal responses. Currently, promising treatment options include adenoviral gene transfers and stem cell therapy. Thus, this review describes in depth known mechanisms contributing to chronic damage and discusses therapeutic advances in treating chronically damaged glands. Understanding the chronic response to radiation offers potential in development of new therapeutics to reverse salivary gland damage and improve the quality of life of HNC survivors.

Research status and prospects of acupuncture for prevention and treatment of chemo- and radiotherapy-induced salivary gland dysfunction in head and neck cancer.

Salivary gland dysfunction (SGD) induced by chemo- and radiotherapy for head and neck cancer (HNC) has always been a difficult problem in modern medicine. The quality of life of a large number of HNC patients is severely impaired by SGD such as xerostomia and dysphagia. In recent years, several studies have found that acupuncture can improve patients' salivary secretion, but it has not yet been approved as an alternative therapy for SGD. For this reason, we collected the clinical study reports on acupuncture in the treatment of SGD induced by chemo- and radiotherapy in HNC patients in the past 20 years, and analyzed and discussed the advantages and disadvantages of these studies with respect to tumor types, group setting, intervention modality, acupoints selection, outcome evaluation, and safety. We believed that acupuncture is beneficial for SGD, but the existing objective evidence is insufficient to support its effectiveness. Therefore, improving the Standards for Reporting Interventions in Clinical Trials of Acupuncture, selecting the optimal combination of acupoints through scientific and rigorous study design, and exploring the potential mechanism of acupuncture in the treatment of diseases combined with the meridian theory may be effective ways to promote the acceptance of acupuncture as an alternative therapy for SGD in future. The significance of this review is to provide a reference for researchers to carry out high-quality clinical trials of acupuncture in the treatment of SGD in future from the perspective of the combination of modern medicine and traditional Chinese medicine.

Salivary Gland Hypofunction and/or Xerostomia Induced by Nonsurgical Cancer Therapies: ISOO/MASCC/ASCO Guideline.

PURPOSE: To provide evidence-based recommendations for prevention and management of salivary gland hypofunction and xerostomia induced by nonsurgical cancer therapies. METHODS: Multinational Association of Supportive Care in Cancer/International Society of Oral Oncology (MASCC/ISOO) and ASCO convened a multidisciplinary Expert Panel to evaluate the evidence and formulate recommendations. PubMed, EMBASE, and Cochrane Library were searched for randomized controlled trials published between January 2009 and June 2020. The guideline also incorporated two previous systematic reviews conducted by MASCC/ISOO, which included studies published from 1990 through 2008. RESULTS: A total of 58 publications were identified: 46 addressed preventive interventions and 12 addressed therapeutic interventions. A majority of the evidence focused on the setting of radiation therapy for head and neck cancer. For the prevention of salivary gland hypofunction and/or xerostomia in patients with head and neck cancer, there is high-quality evidence for tissue-sparing radiation modalities. Evidence is weaker or insufficient for other interventions. For the management of salivary gland hypofunction and/or xerostomia, intermediate-quality evidence supports the use of topical mucosal lubricants, saliva substitutes, and agents that stimulate the salivary reflex. RECOMMENDATIONS: For patients who receive radiation therapy for head and neck cancer, tissue-sparing radiation modalities should be used when possible to reduce the risk of salivary gland hypofunction and xerostomia. Other risk-reducing interventions that may be offered during radiation therapy for head and neck cancer include bethanechol and acupuncture. For patients who develop salivary gland hypofunction and/or xerostomia, interventions include topical mucosal lubricants, saliva substitutes, and sugar-free lozenges or chewing gum. For patients with head and neck cancer, oral pilocarpine and oral cevimeline, acupuncture, or transcutaneous electrostimulation may be offered after radiation therapy.Additional information can be found at www.asco.org/supportive-care-guidelines.

Oral and ocular late effects in head and neck cancer patients treated with radiotherapy.

A broader understanding of oral and ocular late effects in head and neck cancer (HNC) patients who underwent intensity-modulated radiotherapy (IMRT) may provide valuable information in follow-up and improve quality of life. Twenty-nine HNC patients treated at least 6 months earlier and 30 age-matched controls were recruited. After completing several questionnaires: Oral Health Impact Profile-14 (OHIP-14), Shortened Xerostomia Inventory (SXI), Ocular Surface Disease Index (OSDI) and McMonnies Dry Eye questionnaire (MDEQ), participants underwent oral and ocular examinations. Oral examination included clinical oral dryness score (CODS) and secretion rates of unstimulated and stimulated saliva (UWS, SWS). Ocular examination included tear film break-up time, Schirmer test and ocular surface staining. The patients had more problems related to dry mouth than controls based on CODS and SXI, and more complaints of dry eye disease based on OSDI and MDEQ. UWS and SWS rates and oral health related quality of life were significantly lower in the patient group. Subjective oral dryness (SXI) correlated significantly with subjective ocular dryness (OSDI and MDEQ). Our study demonstrates that HNC patients treated with IMRT experience late effects in terms of xerostomia and ocular dryness underlining the importance of interdisciplinary approach in the evaluation and follow-up of HNC patients.

Treatment of oral ranula in HIV-positive patient.

HIV-associated salivary gland disease refers to the pathology in head and neck lesions such as ranula, salivary gland swelling, xerostomia, and benign lymphoepithelial cysts in the parotid gland. Here, we present a unique case of the ranula patient with HIV infection treated with OK-423 sclerotherapy. Case report: The patient was a 42-year-old Japanese male with a few months history of oral floor swelling. Computed tomography (CT) showed a low-density area limited within the right floor of the mouth. Magnetic resonance imaging (MRI) revealed a distinct T2-high intensity area localized on the same location. The puncture fluid was bloody mucus, and the cytology was no malignancy. We diagnosed a simple ranula. He was, however, found to be HIV-antibody positive at the examination before treatment by chance. He was referred to the department of infectious diseases and definitively diagnosed HIV infection by western blot. We chose OK-432 sclerotherapy because of its minimally invasive and the risk of HIV infecting medical staff. Two times OK-432 injection made the lesion disappear. Conclusion: The case indicated that OK-432 sclerotherapy could be effective for ranula related to HIV.

Comparison of Clinical Characteristics and Magnetic Resonance Imaging of Salivary Glands With Magnetic Resonance Sialography in Sjögren's Syndrome.

OBJECTIVES/HYPOTHESIS: To compare the results of magnetic resonance imaging with magnetic resonance sialography (MRSIAL) and the clinical and laboratory characteristics in a well-characterized cohort of patients with primary or secondary Sjögren's syndrome (SS) meeting the American-European Consensus Group criteria. STUDY DESIGN: Retrospective, observational, monocentric study. METHODS: Thirty-six patients (81% female, mean age = 48 ± 35 years) with primary or secondary SS who underwent MRSIAL were included in the study. RESULTS: MRSIAL revealed characteristic radiological signs in the parotid, sublingual, and submandibular salivary glands in 35/36 patients (97%). Patients presenting with anti-Sjögren's syndrome-related antigen A (SSA) autoantibodies showed more often fatty infiltration, a "pepper-and-salt" appearance, ductal stenosis, and/or ductal dilation of the parotid gland (88%, 88%, and 72% respectively) than patients negative for anti-SSA (12%, 4%, and 28% respectively). MRSIAL demonstrated signs characteristic of SS in all 11 patients with negative minor salivary gland biopsy. For 15 patients undergoing ultrasound examination only, 11 (73%) had SS findings, but all 15 had SS findings on MRSIAL. Two cases of parotid lymphoma were detected by MRSIAL (6%). CONCLUSIONS: MRSIAL is a reliable technique to detect glandular anomalies in patients with SS, and seems to provide a valuable aid in the diagnosis of SS. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E83-E89, 2021.

Experimental Animal Model Systems for Understanding Salivary Secretory Disorders.

Salivary secretory disorders are life-disrupting pathologic conditions with a high prevalence, especially in the geriatric population. Both patients and clinicians frequently feel helpless and get frustrated by the currently available therapeutic strategies, which consist mainly of palliative managements. Accordingly, to unravel the underlying mechanisms and to develop effective and curative strategies, several animal models have been developed and introduced. Experimental findings from these models have contributed to answer biological and biomedical questions. This review aims to provide various methodological considerations used for the examination of pathological fundamentals in salivary disorders using animal models and to summarize the obtained findings. The information provided in this review could provide plausible solutions for overcoming salivary disorders and also suggest purpose-specific experimental animal systems.

Long Term Delta-9-tetrahydrocannabinol Administration Inhibits Proinflammatory Responses in Minor Salivary Glands of Chronically Simian Immunodeficieny Virus Infected Rhesus Macaques.

HIV/SIV-associated oral mucosal disease/dysfunction (HAOMD) (gingivitis/periodontitis/salivary adenitis) represents a major comorbidity affecting HIV patients on anti-retroviral therapy. Using a systems biology approach, we investigated molecular changes (mRNA/microRNA) underlying HAOMD and its modulation by phytocannabinoids (delta-9-tetrahydrocannabinol (∆9-THC)) in uninfected (n = 5) and SIV-infected rhesus macaques untreated (VEH-untreated/SIV; n = 7) or treated with vehicle (VEH/SIV; n = 3) or ∆9-THC (THC/SIV; n = 3). Relative to controls, fewer mRNAs were upregulated in THC/SIV compared to VEH-untreated/SIV macaques. Gene enrichment analysis showed differential enrichment of biological functions involved in anti-viral defense, Type-I interferon, Toll-like receptor, RIG-1 and IL1R signaling in VEH-untreated/SIV macaques. We focused on the anti-ER-stress anterior gradient-2 (AGR2), epithelial barrier protecting and anti-dysbiotic WAP Four-Disulfide Core Domain-2 (WFDC2) and glucocorticoid-induced anti-inflammatory TSC22D3 (TSC22-domain family member-3) that were significantly downregulated in oropharyngeal mucosa (OPM) of VEH-untreated/SIV macaques. All three proteins localized to minor salivary gland acini and secretory ducts and showed enhanced and reduced expression in OPM of THC/SIV and VEH/SIV macaques, respectively. Additionally, inflammation associated miR-21, miR-142-3p and miR-29b showed significantly higher expression in OPM of VEH-untreated/SIV macaques. TSC22D3 was validated as a target of miR-29b. These preliminary translational findings suggest that phytocannabinoids may safely and effectively reduce oral inflammatory responses in HIV/SIV and other (autoimmune) diseases.

Photobiomodulation Therapy for Cancer Treatment-Related Salivary Gland Dysfunction: A Systematic Review.

Objective: Symptoms and clinical signs of decreased saliva secretion are a common after cancer therapy. The goal of this research is to systematically review the evidence about the efficacy of photobiomodulation therapy (PBMT) for the management of cancer treatment-related xerostomia or salivary hypofunction. Methods: PubMed was searched for articles investigating the clinical effects of PBMT on cancer therapy-related xerostomia or hyposalivation. The publications that met the eligibility criteria were evaluated for the quality of the study design, physical parameter setting reproducibility, specifics of the treatment protocol, clinical outcomes, and adverse effects. The strongest evidence was given a heavier weight in the overall conclusions. Results: A total of 314 articles were identified, and 5 controlled trials were included in this systematic review. Most of the studies were in head and neck cancer patients treated with radiotherapy (RT) or radiochemotherapy (RT-CT), and one study was in dry mouth associated with hematopoietic stem cell transplantation (HSCT). Data showed conflicting results for either prevention or treatment of RT- or RT-CT-induced dry mouth or hyposalivation. The data for HSCT-related dry mouth were positive. Conclusions: Despite positive preliminary outcomes in most of the trials, it is too early to confidently determine the efficacy of PBM for cancer therapy-related hyposalivation or xerostomia.

IgG4-related lymphadenopathy masquerading as Hodgkin lymphoma: lessons from a pathologist's desk.

IgG4-related disease (IgG4-RD) is a systemic fibroinflammatory disorder affecting multiple organ systems. The awareness of this disease has tremendously increased over the last decade leading to effective treatment and decreased morbidity to the patients. Histopathology plays an important role in the diagnosis of IgG4-RD, and definite histologic criteria are proposed in clinically suspected patients. We report a patient with multiple organ system involvements of the salivary gland, lymph node and kidney. IgG4-related lymphadenopathy (IgG4-RL) in this patient was misdiagnosed as nodular lymphocyte predominant Hodgkin's lymphoma (NLPHL). Refractoriness to treatment for NLPHL and subsequent manifestations of renal involvement lead us to the correct diagnosis of this potentially treatable condition. IgG4-RL can mimic reactive proliferation as well as lymphomas. We report the clinical presentation and discuss the problems faced by pathologists in diagnosing IgG4-RL. We believe that awareness of this rare presentation will enhance the knowledge in diagnosing IgG4-RD.

Salivary Gland Hypofunction and Xerostomia in Head and Neck Radiation Patients.

BACKGROUND: The most manifest long-term consequences of radiation therapy in the head and neck cancer patient are salivary gland hypofunction and a sensation of oral dryness (xerostomia). METHODS: This critical review addresses the consequences of radiation injury to salivary gland tissue, the clinical management of salivary gland hypofunction and xerostomia, and current and potential strategies to prevent or reduce radiation injury to salivary gland tissue or restore the function of radiation-injured salivary gland tissue. RESULTS: Salivary gland hypofunction and xerostomia have severe implications for oral functioning, maintenance of oral and general health, and quality of life. Significant progress has been made to spare salivary gland function chiefly due to advances in radiation techniques. Other strategies have also been developed, e.g., radioprotectors, identification and preservation/expansion of salivary stem cells by stimulation with cholinergic muscarinic agonists, and application of new lubricating or stimulatory agents, surgical transfer of submandibular glands, and acupuncture. CONCLUSION: Many advances to manage salivary gland hypofunction and xerostomia induced by radiation therapy still only offer partial protection since they are often of short duration, lack the protective effects of saliva, or potentially have significant adverse effects. Intensity-modulated radiation therapy (IMRT), and its next step, proton therapy, have the greatest potential as a management strategy for permanently preserving salivary gland function in head and neck cancer patients.Presently, gene transfer to supplement fluid formation and stem cell transfer to increase the regenerative potential in radiation-damaged salivary glands are promising approaches for regaining function and/or regeneration of radiation-damaged salivary gland tissue.

Sjögren's syndrome and ranula development.

OBJECTIVES: We hypothesized that the extravasation of saliva from damaged ducts with lymphocytic infiltration in patients with Sjögren's syndrome causes ranulas. There are too many uncertainties to support this hypothesis. The aim of this study was to investigate whether there is an association between Sjögren's syndrome and ranulas. MATERIALS AND METHODS: We observed three cases of patients with ranulas who were also diagnosed with Sjögren's syndrome at the same facility. These cases led to the question of whether there are other such cases, and thus, an exhaustive literature search was conducted. RESULTS: Three cases in two case reports of mucocele of the floor of the mouth associated with adult Sjögren's syndrome were noted. Including our cases, until now, there have been six cases of ranula with adult Sjögren's syndrome. CONCLUSIONS: It could be useful to investigate whether patients presenting with a ranula are also affected by Sjögren's syndrome and, conversely, investigate patients with Sjögren's syndrome longitudinally to see whether they develop ranulas.

Parotid ductal stenosis after facial cosmetic surgery (2 case reports).

RATIONALE: Facial cosmetic surgery sometimes causes complications involving surrounding tissues. In particular, parotid damage may cause fibrous tissue formation, ductal stenosis, and obstruct salivary flow. Affected patients usually complain of symptoms such as pain and swelling. PATIENT CONCERNS: The authors report 2 cases of obstructive parotitis, arising as a complication of facial cosmetic surgery that were treated successfully using sialendoscopic procedures. DIAGNOSIS: A diagnosis of parotid ductal stenosis was made based on medical history, clinical manifestations, imaging studies, and sialendoscopic findings for the 2 patients. INTERVENTIONS: The successful sialendoscopic procedures was given to the 2 patients. OUTCOMES: The 2 patients have shown no recurrent symptoms during follow-up. LESSONS: Surgeons that perform facial cosmetic procedures should attain a thorough knowledge of the surgical anatomy of the parotid region and sialendoscopic procedure may be helpful for the management of obstructive parotitis due to complication of cosmetic surgery.

Obesity, salivary glands and oral pathology / Obesidad, glándulas salivales y patología oral

Abstract Obesity has reached pandemic proportions in recent years. Not only adults suffer from the disease, but increasingly children and young people. One of the main causes of overweight and obesity is excessive food intake, in particular heavily processed carbohydrates. Obesity alters multiple organs, including the salivary glands, bringing functional alterations with it. Among researchers, the relation between obesity and tooth decay, periodontal disease and xerostomia is being debated. More and more scientific reports are drawing attention to the changes in the microflora of the oral cavity during obesity. All changes are closely related to the morphological and functional alterations of the salivary glands. This article review presents the current points of view regarding the impact of obesity on the health of the salivary glands, and how changes their functions influence other structures in the oral cavity.

Resumen La obesidad ha alcanzado proporciones pandémicas en los últimos años. No solo los adultos padecen la enfermedad, sino también cada vez más niños y jóvenes. Una de las principales causas del sobrepeso y la obesidad es la ingesta excesiva de alimentos, en particular los carbohidratos altamente procesados. La obesidad altera múltiples órganos, incluidas las glándulas salivales, y trae consigo alteraciones funcionales. Entre los investigadores, se está debatiendo la relación entre la obesidad y la caries dental, la enfermedad periodontal y la xerostomía. Cada vez más informes científicos están llamando la atención sobre los cambios en la microflora de la cavidad oral durante la obesidad. Todos los cambios están estrechamente relacionados con las alteraciones morfológicas y funcionales de las glándulas salivales. Esta revisión del artículo presenta los puntos de vista actuales sobre el impacto de la obesidad en la salud de las glándulas salivales, y cómo los cambios en sus funciones influyen en otras estructuras de la cavidad oral.

Management of duct obstruction in transplanted submandibular glands.

BACKGROUND: Submandibular gland (SMG) transplantation is a successful treatment approach for patients with severe dry eye. However, duct obstruction can occur post-transplant. METHODS: We studied nineteen patients with duct obstruction of transplanted SMGs, including five interventional modalities: stone removal; secretory stimulation (to mimic "internal irrigation" with substantial secretory flow); irrigation; surgical opening of stenosis and orifice reconstruction; cephalic vein bypass and Wharton's duct reconstruction. RESULTS: A solitary stone was found and removed in one patient. Duct blockages like mucus plug were cleared by secretory stimulation in three patients, and by normal saline irrigation in two grafts. In the remaining 13 patients, irrigation failed and surgical opening was performed. Orifice reconstruction succeeded in six of the eight patients, whose stenosis was near the orifice. Wharton's duct reconstruction was successful in two of the five cases where stenosis was located in the middle segment of the duct. CONCLUSION: Transplanted SMGs obstruct for various reasons. Stone, which is easy to diagnose and treat, should be excluded first. Non-organic blockage and stenosis were semblable in clinic. Therefore, subsequent steps should be a diagnostic/therapeutic trial of secretory stimulation, followed by irrigation; failure of these interventions suggests the diagnosis of duct stenosis, necessitating surgical recanalization.

Radioactive iodine: An unappreciated threat to salivary gland function.

Thyroid cancer is an endocrine malignancy whose prevalence is increasing in the United States. Nearly 57,000 new cases of thyroid cancer are estimated to be diagnosed in 2017. The standard of care for differentiated thyroid cancer is thyroidectomy followed by ablation of thyroid remnants with high-dose radioactive iodine (131 I). Apart from thyroid glands, 131 I accumulates in cells of salivary glands and compromises its function. Xerostomia is, therefore, a frequent and often persistent complaint of patients. Despite adoption of standard preventive measures, parenchymal damage and chronic salivary dysfunction are observed in a substantial number of patients. Saliva is important for oral homeostasis, and its reduction increases the risk of oral morbidity. As differentiated thyroid cancer patients have an excellent survival rate, preservation of salivary gland function carries added significance. A focus on treatments that preserve or restore long-term salivary flow can significantly improve the quality of life of thyroid cancer survivors.

Head and Neck Cancer Patients ­ Information for the General Dental Practitioner.

Salivary gland damage is the most common adverse effect associated with radiation therapy to the head and neck. A combination of hyposalivation and dietary changes, with a reduced emphasis on oral hygiene practices can contribute to a massive increase in a person's caries risk status. This can be further complicated by limited mouth opening. To enable optimal dental care for head and neck cancer patients before, during and after radiation therapy, patients must be informed and educated about the potential risks of dental caries and the preventive strategies available. All patients should receive a pre-radiotherapy dental assessment by a Restorative Dentistry Consultant. This information will be delivered to the patient, often at an emotionally charged time, and can be lost amongst all the information related to other aspects of his/her cancer management. General Dental Practitioners (GDPs) are therefore in a pivotal position to reiterate this information post radiation therapy and ensure compliance with preventive strategies, with the overall aim to improve quality of life and avoid the need for future extractions and the resulting risk of osteoradionecrosis. Clinical relevance: This article highlights the GDP's role in the shared management of head and neck cancer patients who have received radiotherapy as part of their cancer treatment. The critical issue of dental caries, one of the late effects of radiation-induced hyposalivation, will be focused upon. Other side-effects, such as trismus and osteoradionecrosis, will also be discussed. This article aims to supply GDPs with accurate information to provide to their patients with post radiation therapy, whilst highlighting what treatment is within their remit and when it may be appropriate to refer.

Pediatric primary Sjögren syndrome presenting with bilateral ranulas: A case report and systematic review of the literature.

CONTEXT: Primary Sjögren syndrome is uncommon in children, and the standard clinical criteria used in diagnosis of adult Sjögren syndrome will miss many children with the disease. Floor of mouth ranulas have not been described in Sjögren syndrome. OBJECTIVE: This study aims to describe a novel presentation of juvenile primary Sjögren syndrome, and to present a comprehensive systematic review of the literature regarding the presentation and diagnosis of Sjögren syndrome in children. DATA SOURCES: Ovid MEDLINE. STUDY SELECTION: A MEDLINE literature search was performed using the following search terms: primary, Sjögren, disease, and children. Results were limited to human subjects and articles written in English between 1981 and 2014. Applicable articles were reviewed and qualitatively summarized. DATA EXTRACTION: Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines (PRIMA). RESULTS: Initial MEDLINE search yielded 146 articles, 80 of which were excluded as not clinically pertaining to Sjögren syndrome. An additional 25 were excluded due to lack of pediatric-specific data. Systematic review of the literature revealed no reports of ranula in association with Sjögren syndrome. 6 papers were manually included from review of reference lists of included articles. Our review indicated that recurrent parotitis is the most commonly reported presenting symptom in children, followed by ocular and oral symptoms, musculoskeletal, and renal symptoms. Compared to adults, children are less likely to present with dry eyes and mouth. LIMITATIONS: All studies were retrospective chart reviews, case series or case reports. CONCLUSION: This is the first report of a child presenting with floor of mouth ranulas in association with Sjögren syndrome. While recurrent parotitis is the most common presentation in children, other salivary gland and extra-salivary manifestations may be seen, and the clinician must maintain a high index of suspicion for underlying Sjögren syndrome.

Clinical Outcomes of Several IMRT Techniques for Patients With Head and Neck Cancer: A Propensity Score-Weighted Analysis.

PURPOSE: The Advanced Radiotherapy Oto-Rhino-Laryngologie (ART-ORL) study (NCT02024035) was performed to prospectively evaluate the clinical and economic aspects of helical TomoTherapy and volumetric modulated arc therapy (RapidArc, Varian Medical Systems, Palo Alto, CA) for patients with head and neck cancer. METHODS AND MATERIALS: Fourteen centers participated in this prospective comparative study. Randomization was not possible based on the availability of equipment. Patients with epidermoid or undifferentiated nasopharyngeal carcinoma or epidermoid carcinoma of the oropharynx and oral cavity (T1-T4, M0, N0-N3) were included between February 2010 and February 2012. Only the results of the clinical study are presented in this report, as the results of the economic assessment have been published previously. Inverse probability of treatment weighting using the propensity score analysis was undertaken in an effort to adjust for potential bias due to nonrandomization. Locoregional control, cancer-specific survival, and overall survival assessed 18 months after treatment, as well as long-term toxicity and salivary function, were evaluated. RESULTS: The analysis included 166 patients. The following results are given after inverse probability of treatment weighting adjustment. The locoregional control rate at 18 months was significantly better in the TomoTherapy group: 83.3% (95% confidence interval [CI], 72.5%-90.2%) versus 72.7% (95% CI, 62.1%-80.8%) in the RapidArc group (P=.025). The cancer-specific survival rate was better in the TomoTherapy group: 97.2% (95% CI, 89.3%-99.3%) versus 85.5% (95% CI, 75.8%-91.5%) in the RapidArc group (P=.014). No significant difference was shown in progression-free or overall survival. TomoTherapy induced fewer acute salivary disorders (P=.012). Posttreatment salivary function degradation was worse in the RapidArc group (P=.012). CONCLUSIONS: TomoTherapy provided better locoregional control and cancer-specific survival than RapidArc treatment, with fewer salivary disorders. No significant difference was shown in progression-free and overall survival. These results should be explored in a randomized trial.