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Radiology Quiz Case 1: Diagnosis

Radiology Quiz Case 1: Diagnosis Diagnosis: Chronic sclerosing sialadenitis of the submandibular gland Chronic sclerosing sialadenitis, or Küttner tumor, which was first reported by Küttner in 1896, is a benign chronic inflammatory condition of the submandibular glands. It occurs almost exclusively in the submandibular gland of middle-aged adults unilaterally or bilaterally, but rare involvement of the parotid gland, minor salivary gland, and lacrimal gland has been reported.1-3It is histologically characterized by periductal fibrosis, dense lymphocytic infiltration with lymphoid follicle formation, loss of acini, and, eventually, marked sclerosis of the salivary gland.4It usually manifests as a firm, enlarged gland, with recurring tenderness, unrelated to mastication, which might raise strong clinical suspicion of a malignant neoplasm. However, chronic sclerosing sialadenitis has been underrecognized by otorhinolaryngologists and radiologists, even though it is one of the most common causes of chronic sialadenitis of the submandibular gland. The cause of the disease is still not well established. Postulated mechanisms included sialolithiasis (demonstrated in 29%-83% of the affected glands) or dysfunctional secretory function leading to ductal inspissation and consequently chronic inflammation.4,5However, some authors think that the sialoliths are just a secondary finding of the disease.6Recently, others have detected dense infiltration of IgG4-positive plasma cells in the salivary glands and extrasalivary glandular manifestations in patients with chronic sclerosing sialadenitis and have suggested a link or an overlap between chronic sclerosing sialadenitis and IgG4-related sclerosing disease, which has been recently proposed as a novel clinicopathological entity.7,8IgG4-related sclerosing disease is characterized by extensive infiltration of IgG4-positive plasma cells and T lymphocytes in various organs, including the pancreas, bile duct, gallbladder, salivary gland, retroperitoneum, kidneys, lungs, and prostate gland. Because the submandibular glands are superficial, sonography is often used for the initial evaluation of salivary glands. Characteristic sonographic features include bilateral diffuse involvement with multiple hypoechoic lesions on a heterogeneous background, resembling a “cirrhotic” liver.9Glandular vascularity is prominent, with no mass effects or displacement of these vessels as they course through the parenchyma. The disease might affect the whole gland, showing a parenchymal, nonneoplastic nature, but the degree of involvement may vary. Computed tomography and magnetic resonance imaging may show nonspecific diffuse enlargement of the submandibular glands, and the magnetic resonance signal characteristics are variable.1,9 Patients with chronic sclerosing sialadenitis often present with diffuse, firm enlargement of the submandibular glands, which may clinically mimic malignancy. Therefore, the differential diagnoses include simple chronic sialadenitis, inflammatory pseudotumors, Sjögren syndrome, Mikulicz disease, Kimura disease, sarcoidosis, lymphoma, and a variety of benign and malignant salivary gland tumors.2,9 Chronic sclerosing sialadenitis is a benign disease, and no additional treatment is warranted after diagnosis. While Cheuk and Chan10argue that cytologic findings under the appropriate clinical setting may suggest the diagnosis of chronic sclerosing sialadenitis, and clinical observation afterward is feasible, other authors usually recommend biopsy or excision of the affected glands for diagnosis and treatment. The results of an aspiration cytologic examination may be inconclusive because of low cellularity or the potential overlap in cytologic features with other lymphoproliferative disorders.5,6Therefore, we suggest that IgG assay as well as needle aspiration biopsy or core biopsy would be helpful when chronic sclerosing sialadenitis is strongly suspected on sonography. In summary, chronic sclerosing sialadenitis is a benign inflammatory condition of the submandibular gland that clinically mimics a malignant neoplasm because of its presentation as a hard mass. It shows characteristic imaging findings and should be carefully considered in the differential diagnosis of submandibular masses in clinical practice. Return to . References 1. Roh JLKim JM Küttner's tumor: unusual presentation with bilateral involvement of the lacrimal and submandibular glands. Acta Otolaryngol 2005;125 (7) 792- 796PubMedGoogle ScholarCrossref 2. Blanco MMesko TCura MCabello-Inchausti B Chronic sclerosing sialadenitis (Kuttner's tumor): unusual presentation with bilateral involvement of major and minor salivary glands. Ann Diagn Pathol 2003;7 (1) 25- 30PubMedGoogle ScholarCrossref 3. Williams HKConnor REdmondson H Chronic sclerosing sialadenitis of the submandibular and parotid glands: a report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89 (6) 720- 723PubMedGoogle ScholarCrossref 4. Ellis GLAuclair PL Tumors of the Salivary Glands. Washington, DC Armed Forces Institute of Pathology1996;419- 421 5. Huang CDamrose EBhuta SAbemayor E Kuttner tumor (chronic sclerosing sialadenitis). Am J Otolaryngol 2002;23 (6) 394- 397PubMedGoogle ScholarCrossref 6. Teymoortash ATiemann MSchrader CWerner JA Chronic sclerosing sialadenitis of the submandibular gland is not just a severe chronic sialadenitis. Am J Otolaryngol 2003;24 (4) 278- 279PubMedGoogle ScholarCrossref 7. Kitagawa SZen YHarada K et al. Abundant IgG4-positive plasma cell infiltration characterizes chronic sclerosing sialadenitis (Küttner's tumor). Am J Surg Pathol 2005;29 (6) 783- 791PubMedGoogle ScholarCrossref 8. Kamisawa TOkamoto A IgG4-related sclerosing disease. World J Gastroenterol 2008;14 (25) 3948- 3955PubMedGoogle ScholarCrossref 9. Ahuja ATRichards PSWong KT et al. Kuttner tumour (chronic sclerosing sialadenitis) of the submandibular gland: sonographic appearances. Ultrasound Med Biol 2003;29 (7) 913- 919PubMedGoogle ScholarCrossref 10. Cheuk WChan JK Kuttner tumor of the submandibular gland: fine-needle aspiration cytologic findings of seven cases. Am J Clin Pathol 2002;117 (1) 103- 108PubMedGoogle ScholarCrossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Otolaryngology - Head & Neck Surgery American Medical Association

Radiology Quiz Case 1: Diagnosis

Archives of Otolaryngology - Head & Neck Surgery , Volume 136 (10) – Oct 18, 2010

Radiology Quiz Case 1: Diagnosis

Abstract

Diagnosis: Chronic sclerosing sialadenitis of the submandibular gland Chronic sclerosing sialadenitis, or Küttner tumor, which was first reported by Küttner in 1896, is a benign chronic inflammatory condition of the submandibular glands. It occurs almost exclusively in the submandibular gland of middle-aged adults unilaterally or bilaterally, but rare involvement of the parotid gland, minor salivary gland, and lacrimal gland has been reported.1-3It is histologically characterized by...
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Publisher
American Medical Association
Copyright
Copyright © 2010 American Medical Association. All Rights Reserved.
ISSN
0886-4470
eISSN
1538-361X
DOI
10.1001/archoto.2010.157-b
Publisher site
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Abstract

Diagnosis: Chronic sclerosing sialadenitis of the submandibular gland Chronic sclerosing sialadenitis, or Küttner tumor, which was first reported by Küttner in 1896, is a benign chronic inflammatory condition of the submandibular glands. It occurs almost exclusively in the submandibular gland of middle-aged adults unilaterally or bilaterally, but rare involvement of the parotid gland, minor salivary gland, and lacrimal gland has been reported.1-3It is histologically characterized by periductal fibrosis, dense lymphocytic infiltration with lymphoid follicle formation, loss of acini, and, eventually, marked sclerosis of the salivary gland.4It usually manifests as a firm, enlarged gland, with recurring tenderness, unrelated to mastication, which might raise strong clinical suspicion of a malignant neoplasm. However, chronic sclerosing sialadenitis has been underrecognized by otorhinolaryngologists and radiologists, even though it is one of the most common causes of chronic sialadenitis of the submandibular gland. The cause of the disease is still not well established. Postulated mechanisms included sialolithiasis (demonstrated in 29%-83% of the affected glands) or dysfunctional secretory function leading to ductal inspissation and consequently chronic inflammation.4,5However, some authors think that the sialoliths are just a secondary finding of the disease.6Recently, others have detected dense infiltration of IgG4-positive plasma cells in the salivary glands and extrasalivary glandular manifestations in patients with chronic sclerosing sialadenitis and have suggested a link or an overlap between chronic sclerosing sialadenitis and IgG4-related sclerosing disease, which has been recently proposed as a novel clinicopathological entity.7,8IgG4-related sclerosing disease is characterized by extensive infiltration of IgG4-positive plasma cells and T lymphocytes in various organs, including the pancreas, bile duct, gallbladder, salivary gland, retroperitoneum, kidneys, lungs, and prostate gland. Because the submandibular glands are superficial, sonography is often used for the initial evaluation of salivary glands. Characteristic sonographic features include bilateral diffuse involvement with multiple hypoechoic lesions on a heterogeneous background, resembling a “cirrhotic” liver.9Glandular vascularity is prominent, with no mass effects or displacement of these vessels as they course through the parenchyma. The disease might affect the whole gland, showing a parenchymal, nonneoplastic nature, but the degree of involvement may vary. Computed tomography and magnetic resonance imaging may show nonspecific diffuse enlargement of the submandibular glands, and the magnetic resonance signal characteristics are variable.1,9 Patients with chronic sclerosing sialadenitis often present with diffuse, firm enlargement of the submandibular glands, which may clinically mimic malignancy. Therefore, the differential diagnoses include simple chronic sialadenitis, inflammatory pseudotumors, Sjögren syndrome, Mikulicz disease, Kimura disease, sarcoidosis, lymphoma, and a variety of benign and malignant salivary gland tumors.2,9 Chronic sclerosing sialadenitis is a benign disease, and no additional treatment is warranted after diagnosis. While Cheuk and Chan10argue that cytologic findings under the appropriate clinical setting may suggest the diagnosis of chronic sclerosing sialadenitis, and clinical observation afterward is feasible, other authors usually recommend biopsy or excision of the affected glands for diagnosis and treatment. The results of an aspiration cytologic examination may be inconclusive because of low cellularity or the potential overlap in cytologic features with other lymphoproliferative disorders.5,6Therefore, we suggest that IgG assay as well as needle aspiration biopsy or core biopsy would be helpful when chronic sclerosing sialadenitis is strongly suspected on sonography. In summary, chronic sclerosing sialadenitis is a benign inflammatory condition of the submandibular gland that clinically mimics a malignant neoplasm because of its presentation as a hard mass. It shows characteristic imaging findings and should be carefully considered in the differential diagnosis of submandibular masses in clinical practice. Return to . References 1. Roh JLKim JM Küttner's tumor: unusual presentation with bilateral involvement of the lacrimal and submandibular glands. Acta Otolaryngol 2005;125 (7) 792- 796PubMedGoogle ScholarCrossref 2. Blanco MMesko TCura MCabello-Inchausti B Chronic sclerosing sialadenitis (Kuttner's tumor): unusual presentation with bilateral involvement of major and minor salivary glands. Ann Diagn Pathol 2003;7 (1) 25- 30PubMedGoogle ScholarCrossref 3. Williams HKConnor REdmondson H Chronic sclerosing sialadenitis of the submandibular and parotid glands: a report of a case and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89 (6) 720- 723PubMedGoogle ScholarCrossref 4. Ellis GLAuclair PL Tumors of the Salivary Glands. Washington, DC Armed Forces Institute of Pathology1996;419- 421 5. Huang CDamrose EBhuta SAbemayor E Kuttner tumor (chronic sclerosing sialadenitis). Am J Otolaryngol 2002;23 (6) 394- 397PubMedGoogle ScholarCrossref 6. Teymoortash ATiemann MSchrader CWerner JA Chronic sclerosing sialadenitis of the submandibular gland is not just a severe chronic sialadenitis. Am J Otolaryngol 2003;24 (4) 278- 279PubMedGoogle ScholarCrossref 7. Kitagawa SZen YHarada K et al. Abundant IgG4-positive plasma cell infiltration characterizes chronic sclerosing sialadenitis (Küttner's tumor). Am J Surg Pathol 2005;29 (6) 783- 791PubMedGoogle ScholarCrossref 8. Kamisawa TOkamoto A IgG4-related sclerosing disease. World J Gastroenterol 2008;14 (25) 3948- 3955PubMedGoogle ScholarCrossref 9. Ahuja ATRichards PSWong KT et al. Kuttner tumour (chronic sclerosing sialadenitis) of the submandibular gland: sonographic appearances. Ultrasound Med Biol 2003;29 (7) 913- 919PubMedGoogle ScholarCrossref 10. Cheuk WChan JK Kuttner tumor of the submandibular gland: fine-needle aspiration cytologic findings of seven cases. Am J Clin Pathol 2002;117 (1) 103- 108PubMedGoogle ScholarCrossref

Journal

Archives of Otolaryngology - Head & Neck SurgeryAmerican Medical Association

Published: Oct 18, 2010

Keywords: radiology specialty,sialadenitis,submandibular gland

References