J Bakshi, PJ Venables, S Abraham
Kennedy Institute of Rheumatology, Charing Cross Hospital, 65 Aspenlea Road, Hammersmith, London, W6 8LH, United Kingdom
Kennedy Institute of Rheumatology, Charing Cross Hospital, 65 Aspenlea Road, Hammersmith, London, W6 8LH
|How to cite this article:|
Bakshi J, Venables P J, Abraham S. Sinonasal sarcoid.J Postgrad Med 2009;55:43-44
|How to cite this URL:|
Bakshi J, Venables P J, Abraham S. Sinonasal sarcoid. J Postgrad Med [serial online] 2009 [cited 2021 Oct 18 ];55:43-44
Available from: https://www.jpgmonline.com/text.asp?2009/55/1/43/48441
Parotid swelling is commonly seen by the general internal physician but rarely in combination with anosmia. Olfactory dysfunction is a recognised complication of sarcoidosis in 1-4% of patients.  The incidence of sinonasal sarcoid (SNS) is difficult to estimate and believed to be underreported. In a study of 20 patients with biopsy-proven SNS, SN involvement preceded disease diagnosis in 12 patients. Only a ξ/3 of these patients presented with strictly isolated SNS and anosmia was commonly reported in 70% of cases. 
A 60-year-old male presented to our clinic with a six-month history of bilateral parotid and submandibular gland swelling, dry mouth and complete anosmia. On examination, the salivary glands were bilaterally smoothly enlarged, non-tender with no palpable lymphadenopathy. Schirmer's test was positive in the right eye and unstimulated salivary flow rate was less than 0.5 ml in 5 min. Rigid nasoendocopic examination was normal.
Full blood count, C-reactive protein, serum angiotensin converting enzyme (ACE) and calcium were in the normal range. Erythrocyte sedimentation rate was raised at 32 mm/h (0-10 mm/h) with a polyclonal increase of IgG= 20g/l (8.5-15.0). Anti-nuclear antibodies, anti-neutrophil cytoplasmic antibodies, rheumatoid factor, Anti-Ro and anti-La antibodies were negative. Viral screening was negative for mumps and HIV. Tests for Mycobacterium tuberculosis were negative.
Submandibular gland histology revealed an abnormal architecture with a mixed population of lymphoid cells and occasional blasts. Multinucleate giant cells were present, suggesting the diagnosis of a chronic granulomatous condition and there was no evidence of lymphoma. High-resolution computer tomography (CT) of the thorax showed no features of sarcoid. Magnetic resonance of the brain [Figure 1a] excluded neurosarcoid but did show bilaterally enlarged submandibular and lacrimal glands. There was an extensive abnormal signal within the ethmoid air cells, right frontal sinus and the sphenoid sinus. Additionally, there was mucosal thickening and signal abnormality involving the paranasal sinuses indicative of rhinosinusitis. Gallium scintigraphy demonstrated increased parotid uptake [Figure 1b].
Bilateral parotid gland involvement commonly presents in Sjogren's syndrome but has been reported in 6% of sarcoid patients.  A diagnosis of "seronegative" Sjogren's was unlikely given the lack of ocular symptoms, and histological findings of granulomatous disease. Anosmia occurs in 70% of patients with SNS.  Wegener's granulomatosis can also present with anosmia, but typically presents with a triad of upper and lower airways and renal involvement with c-ANCA positivity in 80% of cases. 
The diagnosis of SNS was made on the basis of clinical, pathological and radiological findings, fulfilling many of the criteria proposed by Braun et al .  Due to normal ear, nose and throat findings we were unable to confirm SNS histologically. Diagnosis from nasal sites can be technically challenging as reported by Djabazov et al .  Blindly performed biopsies are usually negative whereas biopsies from abnormal mucosa gives a higher diagnostic yield.  Specific CT changes in SNS include multiple mucosal nodules and granulomas and accurate recognition can often be challenging. Other CT findings such as opacification of the paranasal sinuses are nonspecific and need to be interpreted in the clinical context. The potential use of [18F] fluorodeoxyglucose positron emission tomography/CT as an imaging modality for accurate diagnosis has been reported by Braun et al., who demonstrated a 100% sensitivity for SNS and may help to identify future cases.  The patient's symptoms improved after a single depo-medrone injection with olfactory function returning within two days. At five months follow-up, the parotid swelling had subsided on a reducing dose of prednisolone and hydroxychloroquine. When bilateral parotid swelling is associated with anosmia, sarcoidosis is likely to be the primary diagnosis.
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