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An Unusual Case Report Of Unilateral Parotid Gland Sarcoidosis With Spontaneous Remission

Diamantopoulos, P. 2019Other/Unknown

Diamantopoulos, Panagiotis T. MD, PhDa,∗; Charakopoulos, Emmanouil MDa; Viniou, Nora-Athina MD, PhDa; Diamantopoulou, Lydia BSca; Gaggadi, Maria MDb. An unusual case report of unilateral parotid gland sarcoidosis with spontaneous remission. Medicine 98(49):p e18172, December 2019. | DOI: 10.1097/MD.0000000000018172

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Abstract

Rationale: Parotid gland sarcoidosis is a well-recognized, but uncommon disease entity. Parotidectomy is most commonly performed to establish the diagnosis and most patients are treated with corticosteroids. Patient concerns: A young female patient presented with right parotid enlargement and developed symptoms of facial nerve palsy during diagnostic investigation. Diagnoses: A fine-needle aspiration cytology showed granulomatous inflammation. The diagnosis of sarcoidosis was eventually established based on the demonstration of the characteristic lambda and panda signs by a Gallium-67 scintigraphy. Interventions: No specific pharmacologic therapy was initiated. Outcomes: The patient's symptoms regressed completely over a period of 3 months. Additionally, she remains asymptomatic 2 years later. Lessons: This case underlines the importance of not initiating corticosteroids in all patients with parotid gland sarcoidosis and suggests that parotidectomy can be avoided in the presence of characteristic for sarcoidosis imaging findings.

Case Details

Disease Location

Left parotid gland

Personal Characteristics

28-year-old woman, hypothyroidism and b-thalassemia heterozygosity.

Clinical Characteristics

Fifteen days before presentation, the patient developed enlargement of the right parotid gland and ipsilateral subman- dibular area, which was accompanied by a diffuse headache of constant intensity without neurologic symptoms the patient had been diagnosed with left trigeminal neuralgia (tn) 10 weeks earlier. Physical examination: 2-cm large, hard, round, irregularly demarcated, painless, and nonmobile mass in the right parotid region. Furthermore, 2 oval-shaped, up to 2cm large, hard, painless, mobile lymph nodes could be palpated in the right submandibular area. Ultrasound demonstrated an enlarged right parotid gland of lobed margin and inhomogeneous echotexture. Endoparotid lymph nodes were also detected. In addition, 2 enlarged right submandibular lymph nodes (22 and 24 mm) were revealed. Fine-needle aspiration (fna) cytology revealed a granulomatous parotitis and lymphadenitis. 24-hour urinary calcium was low (31 mg/24 h) and the patient was vitamin d deficient. A week later, signs of right facial palsy with concomitant increase in the parotid’s size became apparent. Additionally, persistent frontal headache, low grade fever, malaise, lower limb arthralgias and an annular rash in the right knee and gluteal region were added. Chest CT showed enlargement of right paratracheal and hilar nodes bilaterally. In a subsequent gallium-67 (ga-67) scintigraphy, the typical uptake patterns of both lambda and panda signs were observed. The diagnosis of sarcoidosis was established

Remission Characteristics

Over a period of 3 months, lymph node and parotid enlargement regressed completely, the rash resolved, and the patient was free of headache, fever, and symptoms of facial palsy. Two years later, the patient remains free of symptoms.

Treatment & Mechanisms

Proposed Remission Mechanisms

"we are among the few authors that suggest that parotid gland sarcoidosis is a self-limited disease"