J Korean Thyroid Assoc.  2013 Nov;6(2):126-130. 10.11106/jkta.2013.6.2.126.

Concurrent Presence of Sjogren's Syndrome, Warthin Tumor, and MALT Lymphoma in a Parotid Gland and Hashimoto's Thyroiditis

• Kim HS
• Hahm JR
• Jung TS
• Kim SK
• Kim S
• Kim KY
• Jung JH

• KMID: 1845385
• DOI: http://doi.org/10.11106/jkta.2013.6.2.126

Abstract

A 54-year-old female patient was referred due to a mass in the left salivary gland. A neck CT was performed and surgery was agreed due to a suspected Warthin tumor. The patient was also diagnosed with Sjogren's syndrome and Hashimoto's thyroiditis and treated. Warthin tumor and extranodal marginal zone B-cell lymphoma were also diagnosed after parotidectomy. The coexistence of the two autoimmune diseases, Hashimoto's thyroiditis and Sjogren's syndrome, has been reported, as has the coexistence of Warthin tumor and malignant tumor within a single salivary gland. However, these four diseases have not previously been reported in an individual patient. The authors treated a patient who was first diagnosed with Sjogren's syndrome and Hashimoto's thyroiditis, and subsequently also with Warthin tumor and extranodal marginal zone B-cell lymphoma after superficial parotidectomy. Therefore, this case is reported together with a related literature review.

Keyword

Hashimoto's thyroiditis; Sjogren's syndrome; Warthin tumor; MALT lymphoma

MeSH Terms

Adenolymphoma*
Autoimmune Diseases
Female
Humans
Lymphoma, B-Cell, Marginal Zone*
Middle Aged
Neck
Parotid Gland*
Salivary Glands
Sjogren's Syndrome*
Thyroid Gland*
Thyroiditis*

Figure

• 

  Fig. 1. Neck CT scan shows multiple masses with internal cystic changes and focal calcification in both parotid glands.

• 

  Fig. 2. Microscopic findings. (A, B) Warthin tumor. The tumor is composed of a mixture of ductal epithelium and a lymphoid stroma (H&E stain, X40, X100). (C, D) MALT lymphoma. (C) Histological examination of surgical specimens shows diffuse proliferation of atypical small lymphocytes in the mucosa and submucosal layer (H&E stain, X40). (D) Infiltrative lymphocytes are seen within the glandular epithelium (lymphoepithelial lesion) and the gland structures are disrupted in a high power filed (H&E stain, X200).

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  Fig. 3. (A) Coronal PET image shows a focal hot lesion on a right neck level II node and an incidental finding of thyroiditis. (B, C) Transaxial PET and fused PET/CT images show a hypermetabolic lesion on right parotid area (SUVmax=5.94).

Reference

References

1. D’Arbonneau F, Ansart S, Le Berre R, Dueymes M, Youinou P, Pennec YL. Thyroid dysfunction in primary Sjogren's syndrome: a long-term followup study. Arthritis Rheum. 2003; 49(6):804–9.

2. Lazarus MN, Isenberg DA. Development of additional autoimmune diseases in a population of patients with primary Sjogren's syndrome. Ann Rheum Dis. 2005; 64(7):1062–4.

3. Williamson JA. Blue Cross advertising is a crown of thorns for hospitals. Hosp Prog. 1975; 56(5):6–8.

4. Saxena A, Memauri B, Hasegawa W. Initial diagnosis of small lymphocytic lymphoma in parotidectomy for Warthin tumour, a rare collision tumour. J Clin Pathol. 2005; 58(3):331–3.

5. Asmussen K, Andersen V, Bendixen G, Schiodt M, Oxholm P. A new model for classification of disease manifestations in primary Sjogren's syndrome: evaluation in a retrospective long-term study. J Intern Med. 1996; 239(6):475–82.

6. Ramos-Casals M, Garcia-Carrasco M, Cervera R, Gaya J, Halperin I, Ubieto I, et al. Thyroid disease in primary Sjogren syndrome. Study in a series of 160 patients. Medicine (Baltimore). 2000; 79(2):103–8.

7. Tapinos NI, Polihronis M, Moutsopoulos HM. Lymphoma development in Sjogren's syndrome: novel p53 mutations. Arthritis Rheum. 1999; 42(7):1466–72.

8. Theander E, Henriksson G, Ljungberg O, Mandl T, Manthorpe R, Jacobsson LT. Lymphoma and other malignancies in primary Sjogren's syndrome: a cohort study on cancer incidence and lymphoma predictors. Ann Rheum Dis. 2006; 65(6):796–803.

9. Lazarus MN, Robinson D, Mak V, Moller H, Isenberg DA. Incidence of cancer in a cohort of patients with primary Sjogren's syndrome. Rheumatology (Oxford). 2006; 45(8):1012–5.