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Ungprasert P, Crowson CS, Matteson EL. Clinical Characteristics of Parotid Gland Sarcoidosis: A Population-Based Study. JAMA Otolaryngol Head Neck Surg. 2016;142(5):503–504. doi:10.1001/jamaoto.2016.0061
Parotid gland involvement is a well-recognized extrathoracic feature of sarcoidosis. However, data on the epidemiologic and clinical characteristics of parotid gland disease in sarcoidosis are limited. This study describes the epidemiologic features of parotid gland involvement in sarcoidosis, with an emphasis on clinical characteristics, in a geographically well-defined population.
A cohort of Olmsted County, Minnesota, residents diagnosed with sarcoidosis between January 1, 1976, and December 31, 2013, was identified using the resources of the Rochester Epidemiology Project.1 Potential cases were screened from diagnostic codes related to sarcoidosis. Diagnoses of sarcoidosis and parotid gland involvement were then confirmed by review of individual medical records. Cases included were pulmonary sarcoidosis with parotid gland involvement and isolated parotid gland sarcoidosis without intrathoracic disease. Diagnosis of pulmonary sarcoidosis required physician diagnosis supported by histopathologic evidence of noncaseating granuloma and/or radiographic findings of intrathoracic sarcoidosis and compatible clinical presentations, without evidence of other granulomatous diseases. The only exception was stage I pulmonary sarcoidosis, which required only radiographic evidence of symmetric bilateral hilar adenopathy. Diagnosis of parotid gland involvement required signs and symptoms of parotid gland infiltration, such as parotid gland enlargement. Biopsy confirmation was not required if the patient had pulmonary sarcoidosis. If the patient did not have pulmonary involvement, parotid gland biopsy with demonstration of noncaseating granuloma was required. In cases without intrathoracic involvement, biopsy-proven isolated granulomatous disease of other specific organs except for the skin was also included if there was no better alternative diagnosis. Patients with a diagnosis of sarcoidosis before residency in Olmsted County were excluded. Approval for this study was obtained from the Mayo Clinic and Olmsted Medical Center Institutional Review Boards and the need for patient written informed consent was waived. Data analysis was performed from January 1 to June 30, 2015.
A total of 345 incident cases of sarcoidosis were identified. Of those, only 7 patients had parotid gland involvement (mean age, 44.2 years; 3 female [42.9%], 6 white [85.7%], and 1 African American [14.3%]). Most patients had parotid gland disease in association with intrathoracic sarcoidosis; isolated parotid sarcoidosis was observed in only 1 patient. Parotid gland disease was usually painless and unilateral and was the initial presentation in 4 patients. The angiotensin-converting enzyme level was elevated in 3 patients (42.9%) and no patients had hypercalcemia. Gland swelling regressed after treatment with corticosteroids in all patients, although 1 relapse was seen. The Table describes the clinical characteristics of these patients.
Only 7 patients (2.0%) with sarcoidosis in this population-based cohort developed parotid gland involvement, a frequency considerably lower than previous reports of 5% to 30%.2-4 The difference could be owing to the diversity in ethnic background of the cohorts, as our cohort was predominantly white.5 Another possible explanation was related to the study design. As this study was a population-based study, it might capture a more complete spectrum of the disease, in contrast to the referral-based cohort design used in previous studies.2-4
The most common presentation of parotid gland disease was unilateral painless gland swelling, which was also different from previous studies that found bilateral involvement in more than 70% of their cohorts.3,4 More important, parotid gland disease was often the initial manifestation in our study, highlighting the importance of the otolaryngologic assessment in the diagnosis of systemic sarcoidosis.
The demographics of patients with parotid gland involvement were similar to those of the complete cohort of 345 patients (mean age, 45.6 years; 50% female and 95% white). The angiotensin-converting enzyme level was elevated in 3 of 6 tested patients, which was also not significantly different from the complete cohort (104 of 251 tested patients [41.4%]).
In conclusion, the prevalence of parotid gland involvement in this population-based cohort of patients with sarcoidosis was lower than in previous reports. Prognosis was favorable for patients with parotid gland involvement.
Corresponding Author: Patompong Ungprasert, MD, Division of Rheumatology, Department of Internal Medicine, Mayo Clinic College of Medicine, 200 First St SW, Rochester, MN 55905 (email@example.com).
Published Online: March 17, 2016. doi:10.1001/jamaoto.2016.0061.
Author Contributions: Dr Ungprasert and Ms Crowson had full access to all the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis.
Study concept and design: Ungprasert, Matteson.
Acquisition, analysis, or interpretation of data: All authors.
Drafting of the manuscript: Ungprasert, Matteson.
Critical revision of the manuscript for important intellectual content: All authors.
Statistical analysis: Ungprasert, Crowson.
Administrative, technical, or material support: Ungprasert,
Study supervision: Matteson.
Conflict of Interest Disclosures: None reported.
Funding/Support: This study was funded by award R01 AG034676 from the National Institute on Aging of the National Institutes of Health via the Rochester Epidemiology Project, and Clinical and Translational Science Awards grant UL1 TR000135 from the National Center for Advancing Translational Sciences, a component of the National Institutes of Health.
Role of the Funder/Sponsor: The funding sources had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication.
Disclaimer: The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.