Chronic sclerosing sialadenitis

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Chronic sclerosing sialadenitis
Specialty Dentistry

Chronic sclerosing sialadenitis is a chronic (long-lasting) inflammatory condition affecting the salivary gland. Relatively rare in occurrence, this condition is benign, but presents as hard, indurated and enlarged masses that are clinically indistinguishable from salivary gland neoplasms or tumors. It is now regarded as a manifestation of IgG4-related disease. [1]

Contents

Involvement of the submandibular glands is also known as Küttner's tumor, named after Hermann Küttner (1870–1932), a German Oral and Maxillofacial Surgeon, who reported four cases of submandibular gland lesions for the first time in 1896. [2]

Presentation

The inflammatory lesions in Küttner's tumor may occur on one side (unilateral) or both sides (bilateral), predominantly involving the submandibular gland, [3] but is also known to occur in other major and minor salivary glands, [4] including the parotid gland. [5] [6] Overall, salivary gland tumors are relatively rare, with approximately 2.5–3 cases per 100,000 people per year seen in the Western world; however, salivary gland malignancies account for 3–5% of all head and neck cancers. [7] However, salivary tumors show a great deal of morphological diversity, as well as variations in the nature of the lesion (malignant vs. benign): approximately 20% to 25% of parotid tumors, 35% to 40% of submandibular tumors, and more than 90% of sublingual gland tumors are malignant. [8] This situation underscores the diagnostic challenges in respect of Küttner's tumor; despite being benign, this condition mimics the clinical appearance of malignancy in the salivary gland.

The swollen masses of Küttner's tumor are generally painful, and patients are advised surgical resection (known as 'sialoadenectomy') of a part or whole of the glandular tissue upon suspicion of possible malignancy. It is only upon post-surgical histopathology of the excised mass that the diagnosis of Küttner's tumor is definitively made.

Histological features

The histopathological features that characterize Küttner's tumor [9] include:

Pathogenesis

The cause and pathogenesis of this chronic condition are not very well understood. Several factors have been postulated:

This chronic condition is primarily observed in adult (40–70 years) patients. However, Küttner's tumor, with prominent immunopathological features, has been described in an 11-year-old boy in Brazil in 2012. [19]

Diagnosis

Given the difficulties of a definitive pre-operative diagnosis, the clinical entity of Küttner's tumor has so far remained significantly under-reported and under-recognized. In recent times, armed with a better understanding of the occurrences and observable features of this condition, surgeons are increasingly depending upon pre-operative ultrasonography along with Fine-needle aspiration cytological (FNAC) examinations to make an accurate presumptive diagnosis, [20] and according to one estimate, about 44% of patients undergoing submandibular resection are found to have this condition. [20] In the ultrasonogram, Küttner's tumor is characterized by a diffuse, heterogeneous zone of echo-shadows. [21] The FNAC finds cells greatly reduced in number (called 'paucicellularity') along with scattered tubular ducts against a backdrop of lymphoplasmacytic infiltration and fibrous depositions. [21] There may be a reduced but moderate number of cells and ducts enveloped in fibrous sheaths, as well as fibrous proliferation of the gland's septa. [22] The cytologic findings by themselves may not be specific, and the diagnosis requires adjunct consideration of both the ultrasonogram and clinical presentation. Application of magnetic resonance imaging (MRI) has been tried to non-invasively examine the morphological variations in Küttner's tumor and differentiate them from those seen in malignant tumors; [23] while MRI findings of the affected tissue and the pattern of cellular infiltration may offer some diagnostic clues for this condition, so far the results have been inconclusive. [23]

Existing treatment

Standard, and most effective, therapy to date is glandular sialoadenectomy, which is associated with fairly low operative morbidity; however, in recent times, the administration of steroid (which can shrink the inflammatory lesion and is known to reduce serum IgG4 values) has been considered favorably, [16] [17] and may be useful in younger patients or those who refuse surgery.

See also

Related Research Articles

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The salivary glands in many vertebrates including mammals are exocrine glands that produce saliva through a system of ducts. Humans have three paired major salivary glands, as well as hundreds of minor salivary glands. Salivary glands can be classified as serous, mucous, or seromucous (mixed).

<span class="mw-page-title-main">Parotid gland</span> Major salivary gland in many animals

The parotid gland is a major salivary gland in many animals. In humans, the two parotid glands are present on either side of the mouth and in front of both ears. They are the largest of the salivary glands. Each parotid is wrapped around the mandibular ramus, and secretes serous saliva through the parotid duct into the mouth, to facilitate mastication and swallowing and to begin the digestion of starches. There are also two other types of salivary glands; they are submandibular and sublingual glands. Sometimes accessory parotid glands are found close to the main parotid glands.

<span class="mw-page-title-main">Submandibular gland</span> Human salivary gland

The paired submandibular glands are major salivary glands located beneath the floor of the mouth. In adult humans, they each weigh about 15 grams and contribute some 60–67% of unstimulated saliva secretion; on stimulation their contribution decreases in proportion as parotid gland secretion rises to 50%. The average length of the normal adult human submandibular salivary gland is approximately 27 mm, while the average width is approximately 14.3 mm.

<span class="mw-page-title-main">Parotitis</span> Medical condition

Parotitis is an inflammation of one or both parotid glands, the major salivary glands located on either side of the face, in humans. The parotid gland is the salivary gland most commonly affected by inflammation.

The oral mucosa is the mucous membrane lining the inside of the mouth. It comprises stratified squamous epithelium, termed "oral epithelium", and an underlying connective tissue termed lamina propria. The oral cavity has sometimes been described as a mirror that reflects the health of the individual. Changes indicative of disease are seen as alterations in the oral mucosa lining the mouth, which can reveal systemic conditions, such as diabetes or vitamin deficiency, or the local effects of chronic tobacco or alcohol use. The oral mucosa tends to heal faster and with less scar formation compared to the skin. The underlying mechanism remains unknown, but research suggests that extracellular vesicles might be involved.

<span class="mw-page-title-main">Warthin's tumor</span> Medical condition

Warthin's tumor, also known as papillary cystadenoma lymphomatosum, is a benign cystic tumor of the salivary glands containing abundant lymphocytes and germinal centers. It is named for pathologist Aldred Scott Warthin, who described two cases in 1929.

<span class="mw-page-title-main">Pleomorphic adenoma</span> Medical condition

Pleomorphic adenoma is a common benign salivary gland neoplasm characterised by neoplastic proliferation of epithelial (ductal) cells along with myoepithelial components, having a malignant potentiality. It is the most common type of salivary gland tumor and the most common tumor of the parotid gland. It derives its name from the architectural Pleomorphism seen by light microscopy. It is also known as "Mixed tumor, salivary gland type", which refers to its dual origin from epithelial and myoepithelial elements as opposed to its pleomorphic appearance.

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<span class="mw-page-title-main">Mucoepidermoid carcinoma</span> Medical condition

Mucoepidermoid carcinoma (MEC) is the most common type of minor salivary gland malignancy in adults. Mucoepidermoid carcinoma can also be found in other organs, such as bronchi, lacrimal sac, and thyroid gland.

<span class="mw-page-title-main">Sialadenitis</span> Medical condition

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<span class="mw-page-title-main">Sialolithiasis</span> Medical condition

Sialolithiasis is a crystallopathy where a calcified mass or sialolith forms within a salivary gland, usually in the duct of the submandibular gland. Less commonly the parotid gland or rarely the sublingual gland or a minor salivary gland may develop salivary stones.

<span class="mw-page-title-main">Acinic cell carcinoma</span> Medical condition

Acinic cell carcinoma is a malignant tumor representing 2% of all salivary tumors. 90% of the time found in the parotid gland, 10% intraorally on buccal mucosa or palate. The disease presents as a slow growing mass, associated with pain or tenderness in 50% of the cases. Often appears pseudoencapsulated.

<span class="mw-page-title-main">Stafne defect</span> Mandibular depression

The Stafne defect is a depression of the mandible, most commonly located on the lingual surface. The Stafne defect is thought to be a normal anatomical variant, as the depression is created by ectopic salivary gland tissue associated with the submandibular gland and does not represent a pathologic lesion as such. This cavity is commonly observed on panoramic radiograph.

<span class="mw-page-title-main">Salivary gland tumour</span> Medical condition

Salivary gland tumours, also known as mucous gland adenomas or neoplasms, are tumours that form in the tissues of salivary glands. The salivary glands are classified as major or minor. The major salivary glands consist of the parotid, submandibular, and sublingual glands. The minor salivary glands consist of 800 to 1000 small mucus-secreting glands located throughout the lining of the oral cavity. Patients with these types of tumours may be asymptomatic.

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<span class="mw-page-title-main">IgG4-related disease</span> Medical condition

IgG4-related disease (IgG4-RD), formerly known as IgG4-related systemic disease, is a chronic inflammatory condition characterized by tissue infiltration with lymphocytes and IgG4-secreting plasma cells, various degrees of fibrosis (scarring) and a usually prompt response to oral steroids. In approximately 51–70% of people with this disease, serum IgG4 concentrations are elevated during an acute phase.

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References

  1. ↑ John H. Stone; Arezou Khosroshahi; Vikram Deshpande; et al. (October 2012). "Recommendations for the nomenclature of IgG4-related disease and its individual organ system manifestations". Arthritis & Rheumatism. 64 (10): 3061–3067. doi:10.1002/art.34593. PMC   5963880 . PMID   22736240.
  2. ↑ Küttner, H (1896). "Ueber entzündliche Tumoren der submaxillar-speicheldrüse". Beiträge zur Klinischen Chirurgie. 15: 815–828. hdl:2027/uc1.b3731679.
  3. ↑ Kiverniti, E; Singh A; Clarke P (2008). "Küttner's tumour: an unusual cause of salivary gland enlargement". Hippokratia. 12 (1): 56–58. PMC   2532958 . PMID   18923750.
  4. ↑ Blanco, Mario; Mesko T; Cura M; Cabello-Inchausti B (2003). "Chronic sclerosing sialadenitis (Kuttner's tumor): unusual presentation with bilateral involvement of major and minor salivary glands". Annals of Diagnostic Pathology. 7 (1): 25–30. doi:10.1053/adpa.2003.50004. PMID   12616471.
  5. ↑ Beriat, GK; Akmansu SH; Kocatürk S; Ataoğlu O (2010). "Chronic Sclerosing Sialadenitis (Küttner's tumour) of the Parotid Gland". Malaysian Journal of Medical Sciences. 17 (4): 57–61. PMC   3216182 . PMID   22135562.
  6. 1 2 de Vicente, JC; López-Arranz E; García J; López-Arranz JS (2003). "Chronic sclerosing sialadenitis of the parotid gland". Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 96 (1): 77–80. doi:10.1016/s1079-2104(03)00096-9. PMID   12847448.
  7. ↑ Speight, PM; Barrett AW (2002). "Salivary gland tumours". Oral Diseases. 8 (5): 229–240. doi: 10.1034/j.1601-0825.2002.02870.x . PMID   12363107.
  8. ↑ National Cancer Institute (NCI/NIH) (20 July 2012). "General Information About Salivary Gland Cancer". cancer.gov. Archived from the original on 27 April 2015. Retrieved 15 November 2013.
  9. ↑ Chan ACL; Chan JKC; Abbondanzo SL (2005). "Chapter 5. Tumours of the Salivary Glands" (PDF). In Leon Barnes; John W. Eveson; Peter Reichart; David Sidransky (eds.). World Health Organization Classification of Tumours: Pathology and Genetics of Head and Neck Tumours. Lyon, France: WHO/IARC Press. p. 278. ISBN   92-83-22417-5. Archived from the original (PDF) on 1 April 2017. Retrieved 15 November 2013.
  10. ↑ Epivatianos, A; Harrison JD; Dimitriou T (2006). "Ultrastructural and histochemical observations on microcalculi in chronic submandibular sialadenitis". Journal of Oral Pathology and Medicine. 16 (10): 514–517. doi:10.1111/j.1600-0714.1987.tb00683.x. PMID   3127566.
  11. ↑ Chan, JKC (1998). "Kuttner tumor (chronic sclerosing sialadenitis) of the submandibular gland: an underrecognized entity". Advances in Anatomic Pathology. 5 (4): 239–51. doi:10.1097/00125480-199807000-00004. PMID   9859756. S2CID   33288884.
  12. ↑ Ikeda, M; Ikui A; Tanaka M; Omori H; Tomita H (1994). "Immunohistopathological investigation on unspecific chronic sclerosing sialadenitis of the submandibular gland (Kuttner tumor)". Auris Nasus Larynx. 21 (2): 103–110. doi:10.1016/s0385-8146(12)80028-9. PMID   7993223.
  13. ↑ Tiemann, M; Teymoortash A; Schrader C; Werner JA; Parwaresch R; Seifert G; Klöppel G (2002). "Chronic sclerosing sialadenitis of the submandibular gland is mainly due to a T lymphocyte immune reaction". Modern Pathology. 15 (8): 845–852. doi: 10.1097/01.MP.0000022280.72359.04 . PMID   12181270.
  14. ↑ Tsuneyama, K; Saito K; Ruebner BH; Konishi I; Nakanuma Y; Gershwin ME (2000). "Immunological similarities between primary sclerosing cholangitis and chronic sclerosing sialadenitisreport of the overlapping of these two autoimmune diseases". Digestive Diseases and Sciences. 45 (2): 366–372. doi:10.1023/A:1005429130150. PMID   10711453. S2CID   24581402.
  15. ↑ Sekine, S; Nagata M; Watanabe T (1999). "Chronic sclerosing sialadenitis of the submandibular gland associated with idiopathic retroperitoneal fibrosis". Pathology International. 49 (7): 663–667. doi:10.1046/j.1440-1827.1999.00926.x. PMID   10504530. S2CID   21548017.
  16. 1 2 Kamisawa, T; Nakajima H; Hishima T (2006). "Close correlation between chronic sclerosing sialadenitis and immunoglobulin G4". Internal Medicine Journal. 36 (8): 527–529. doi:10.1111/j.1445-5994.2006.01119.x. PMID   16866659. S2CID   33328260.
  17. 1 2 Nagai, K; Andoh K; Ogata A; Aoki N; Nakamura N; Hosaka H; Kurihara R (2010). "A new category for chronic sclerosing sialadenitis as an IgG4 related syndrome". BMJ Case Reports. 2010: bcr1020092412. doi:10.1136/bcr.10.2009.2412. PMC   3028012 . PMID   22448185.
  18. ↑ Cheuk, W; Yuen HK; Chu SY; Chiu EK; Lam LK; Chan JK (2008). "Lymphadenopathy of IgG4-related sclerosing disease". Am J Surg Pathol. 32 (5): 671–681. doi:10.1097/PAS.0b013e318157c068. PMID   18344866. S2CID   27175700.
  19. ↑ Melo, JC; Kitsko D; Reyes-Múgica M (2012). "Pediatric chronic sclerosing sialadenitis: Küttner tumor". Pediatric and Developmental Pathology. 15 (2): 165–169. doi:10.2350/11-04-1023-OA.1. PMID   21985393. S2CID   26785007.
  20. 1 2 Chow, TL; Chan TT; Choi CY; Lam SH (2008). "Kuttner's tumour (chronic sclerosing sialadenitis) of the submandibular gland: a clinical perspective" (PDF). Hong Kong Medical Journal. 14 (1): 46–9. PMID   18239243 . Retrieved 15 November 2013.
  21. 1 2 Chou, YH; Tiu CM; Li WY; Liu CY; Cheng YC; Chiou HJ; Chiou SY; Wang HK; Hung GS (2005). "Chronic sclerosing sialadenitis of the parotid gland: diagnosis using color Doppler sonography and sonographically guided needle biopsy". Journal of Ultrasound in Medicine. 24 (4): 551–555. doi: 10.7863/jum.2005.24.4.551 . PMID   15784774. S2CID   24563080 . Retrieved 17 November 2013.
  22. ↑ Cheuk, W; Chan, JKC (2002). "Kuttner Tumor of the Submandibular Gland: Fine-Needle Aspiration Cytologic Findings of Seven Case" (PDF). American Journal of Clinical Pathology. 117 (1): 103–108. doi: 10.1309/g9t3-22mh-q7kl-g2dl . PMID   11791589 . Retrieved 5 December 2013.
  23. 1 2 Abu, A; Motoori K; Yamamoto S; Hanazawa T; Nagai Y; Kaneoya K; Ito H (2008). "MRI of chronic sclerosing sialoadenitis". British Journal of Radiology. 81 (967): 531–536. doi:10.1259/bjr/38960620. PMID   18316344.


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