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Dentomaxillofacial Radiology (2007) 36, 428-433
© 2007 British Institute of Radiology
doi: 10.1259/dmfr/39858276

CASE REPORTS

Rosai–Dorfman disease of the parotid and submandibular glands: salivary gland scintigraphy and oral findings in two siblings

G Güven*,1, S Ilgan2, C Altun1, M Gerek3 and O Gunhan4

1 Department of Pedodontics, Center of Dental Sciences, Gulhane Military Medical Academy, Ankara, Turkey; 2 Department of Nuclear Medicine, Gulhane Military Medical Academy and Medical Faculty, Ankara, Turkey; 3 Department of Otolaryngology, Head and Neck Surgery, Gulhane Military Medical Academy and Medical Faculty, Ankara, Turkey; 4 Department of Pathology, Gulhane Military Medical Academy, Ankara, Turkey

*Correspondence to: Dr Günseli Güven, Gülhane Askeri Tip Akademisi, Dishekimligi Bilimleri Merkezi, Pedodonti Anabilim Dali, Etlik-Ankara, Türkiye; Email: guvengunseli{at}yahoo.com

Received 7 August 2006; revised 29 November 2006; accepted 19 December 2006

Rosai–Dorfman disease (RDD) is an unusual clinical entity characterized by benign pseudolymphomatous proliferation with significant histiocytic infiltration. In the present paper, extranodal RDD of the major salivary glands causing salivary hypofunction and the results of salivary gland scintigraphy and ultrasound are presented in two siblings. Case 1: a 10-year-old boy with bilateral painless masses around the parotid and submandibular glands was referred. Ultrasound examination showed bilateral, well-defined, hypoechoic solid mass lesions within both parotid glands with minimal normal parenchyma in the upper poles. Both submandibular glands were markedly hypoechoic and heterogeneous. Mass lesions within the parotid glands appeared as cold lesions with regular contours on scintigraphy. Dynamic images showed normal uptake and normal response to secretion in the upper poles of the parotid glands, corresponding with ultrasonographically normal parenchyma. Both submandibular glands showed markedly diminished uptake and secretion. Case 2: a 9-year-old boy presented with mass lesions around the submandibular glands. Ultrasound examination showed normal parotid glands and markedly hypoechoic and heterogeneous submandibular glands. Salivary gland scintigraphy showed normal uptake and secretion of parotid glands with markedly diminished uptake and secretion in both submandibular glands. There were severe carious lesions in both patients due to salivary hypofunction. Treatments of the two patients' teeth were performed. Major salivary gland involvement of RDD is important for dentists as it may cause xerostomia and can mimic dental abscess. Functional evaluation of salivary glands with scintigraphy, besides radiological and pathological techniques, will help to explain whether salivary glands are affected or not and improve the diagnostic effectiveness.

Keywords: Rosai–Dorfman disease; salivary glands; scintigraphy; ultrasound






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