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CORRESPONDENCE
Year : 2017  |  Volume : 13  |  Issue : 1  |  Page : 148-151

Sialadenoma papilliferum: A rare case report and review of literature


1 Department of Oral and Maxillofacial Pathology, Pushpagiri College of Dental Sciences, Tiruvalla, Kerala, India
2 Department of Pathology, Pushpagiri Medical College, Tiruvalla, Kerala, India
3 Department of Oral and Maxillofacial Surgery, Pushpagiri College of Dental Sciences, Tiruvalla, Kerala, India

Date of Web Publication16-May-2017

Correspondence Address:
S Sunil
Department of Oral and Maxillofacial Pathology, Pushpagiri College of Dental Sciences, Tiruvalla, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-1482.187300

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 > Abstract 

Sialadenoma papilliferum (SP) classified under the ductal papillomas by the WHO is a rare benign tumor of minor salivary glands. It is a rare lesion of salivary glands predominantly affecting the minor glands. It has characteristic exophytic and endophytic clinical growth pattern. Histopathologically, it is characterized by papillary projections supported by fibrovascular connective tissue core and infiltrated with mixed inflammatory cells. The ductal lining epithelium of double-layered cells of luminal layer of tall columnar cells and a basilar layer of small cuboidal cells shows additional papillary projections into the lumen. We report a case of SP of mid palate.

Keywords: Adenoma, ductal, papillary, tumor


How to cite this article:
Sunil S, Babu SS, Panicker S, Pratap N. Sialadenoma papilliferum: A rare case report and review of literature. J Can Res Ther 2017;13:148-51

How to cite this URL:
Sunil S, Babu SS, Panicker S, Pratap N. Sialadenoma papilliferum: A rare case report and review of literature. J Can Res Ther [serial online] 2017 [cited 2019 Nov 16];13:148-51. Available from: http://www.cancerjournal.net/text.asp?2017/13/1/148/187300


 > Introduction Top


The sialadenoma papilliferum (SP) first described by Abrams and Finck in 1969 as a benign exophytic tumor of salivary gland origin constitutes for <1% of all minor salivary gland tumors.[1] This name was proposed based on its gross and microscopic appearances, which closely resembled those of the syringocystadenoma papilliferum of the sweat gland.[2]

SP is a rare benign tumor of salivary glands. It mostly arises from minor salivary glands, but it is reported in parotid gland as well. It occurs in adults and males are commonly affected than females in the ratio 1.5:1.[3],[4],[5]

The salivary gland tumors that show papillomatous growth pattern histologically include Warthins tumor, SP, intraductal papilloma, and inverted ductal papilloma.[3],[4]

Histopathologically, it is characterized by multiple ductal lumina lined by double-layered cells of luminal layer of tall columnar cells and a basilar layer of small cuboidal cells. A mixed inflammatory cell infiltrate is often noticed.[1],[2],[3],[4] The diagnosis is made by clinicohistopathological correlation.


 > Case Report Top


A 58-year-old female patient reported with the complaint of a painless swelling in the middle region of palate of 1-month duration. On examination, an exophytic erythematous growth of 1 cm × 1 cm, with papillary projections was noticed [Figure 1]. A provisional diagnosis of papilloma/fibroma was made. After routine blood investigation, excisional biopsy was performed. Histopathological examination of the tissue showed fibrous connective tissue covered with keratinized stratified squamous epithelium with surface papillary projections in some areas. The submucosa showed multiple ductal lumens lined by a double rowed layer of cells consisting of luminal columnar cells and basilar cuboidal cells. A few ductal cells have an oncocytic appearance. The lining epithelium of ducts shows papillary projections into the lumen. The connective tissue showed a diffuse collection of mixed inflammatory cells chiefly plasma cells, lymphocytes, and neutrophils [Figure 2],[Figure 3],[Figure 4],[Figure 5],[Figure 6]. A diagnosis of SP was made.
Figure 1: Clinical photograph showing exophytic growth on the right side of palate

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Figure 2: Photomicrograph showing papillary growth of surface epithelium (H and E, ×4)

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Figure 3: Photomicrograph showing multiple ductal proliferations (H and E, ×4)

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Figure 4: Photomicrograph showing multiple ductal lumen lined by double layer of cells (H and E, ×10)

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Figure 5: Photomicrograph showing ductal lumen lined by luminal columnar cells and basilar cuboidal cells (H and E, ×10)

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Figure 6: Photomicrograph showing multiple ductal lumen lined by double layer of cells reaching juxta-epithelial connective tissue (H and E, ×10)

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 > Discussion Top


SP occurs mainly in the minor salivary glands of the oral and pharyngeal mucosa.[5] The oral sites involved include posterior hard palate or soft palate, the junction of hard and soft palate, buccal mucosa, mandibular retromolar trigone, faucial pillar, and labial mucosa in the decreasing order.[3],[4],[5] Rarely major salivary glands are also affected.[5]

The size of the lesions varies from 1 to 2.5 cm when affecting minor glands, a lesion as large as 7.5 cm, involving parotid gland was reported. The affected patient's age ranged from 2 to 87 years with a male to female ratio of 1.5:1.[5],[6]

The lesion is most slowly growing, usually painless, and the majority well-circumscribed, the surface is papillary and in clinical examination this can be confused with others papillary lesions such as squamous papilloma.[3],[4],[5],[6],[7],[8] The exophytic growth of SP contrasts markedly with most other intraoral salivary gland tumors, which presented as submucosal nodular swelling.[5],[6],[7]

Macroscopically, it appears as round to oval lesion and well circumscribed. Usually, it is an erythematous pedunculated or sessile mass with the rough, pebbly, verrucous, or papillary surface.[3],[4],[5]

Histologically, it is characterized by both exophytic and endophytic proliferation of ductal epithelium composed of a double layer of cells.[8],[9],[10] The mucosal surface has many papillary projections supported by fibrovascular connective tissue core, infiltrated with mixed inflammatory cells. The surface stratified squamous epithelium merges with the ductal epithelium in some area. The ductal structures, often are dilated, and the lining epithelium shows additional papillary projections into the lumen.[5],[6],[7],[8],[9] The double-layered ductal epithelium lining the ducts, cysts, and papillary folds include tall columnar luminal cells and smaller cuboidal basal cells. They are eosinophilic, resembling striated and interlobular duct cells. Some of them appear oncocytic.[5]

SP should be differentiated from, squamous papilloma, warty dyskeratoma, and incipient verrucous carcinoma and cystadenomas. The ductal and glandular proliferations of SP readily delineates it from the other exophytic and papillary lesions.[5]

Warthin's tumor has characteristic lymphoid tissue with germinal centers in addition to the double layered ductal pattern.[4]

If there is marked pseudoepitheliomatous hyperplasia of the surface epithelium, necrotizing sialometaplasia should also be differentiated. Furthermore, necrotizing sialometaplasia and mucoepidermoid carcinoma lack the ductal and glandular proliferations of SP.[5]

The intraductal papilloma and inverted ductal papilloma though resembles SP, neither exhibits the exophytic growth pattern.[3],[4],[5]

The histogenesis of SP remains unclear. Some authors have suggested that it has an excretory duct or pluripotential myoepithelial cellular origin, while others have proposed that it originates from intercalated salivary ducts, or excretory ducts, because of the presence of squamous differentiation.[11],[12],[13],[14]

Whether SP is a tumor or a reactive lesion is debatable. The small size and prominent inflammatory infiltrate, suggest a reactive hyperplastic nature.[5],[15] Whittaker and Turner called SP as “focal oncocytic adenomatous hyperplasia.”[15]

On the other hand, reported larger lesions, recurrence, and the proliferation of ductal elements suggest a neoplastic behavior.[5],[15]

The treatment is surgical excision. The prognosis of the lesion is very good. Only one case has been reported to have recurred.[3],[4],[5]


 > Conclusion Top


SP, one of the ductal papillomas is a rare benign tumor of salivary glands with pathognomonic clinicohistopathologic correlation.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 > References Top

1.
Abrams AM, Finck FM. Sialadenoma papilliferum. A previously unreported salivary gland tumor. Cancer 1969;24:1057-63.  Back to cited text no. 1
[PUBMED]    
2.
Helving EB, Hackeny VC. Siringoadenoma papilliferum. Lesions with or without naevus sebaceous and basal cell carcinoma. Arch Dermatol 1955;71:361-72.  Back to cited text no. 2
    
3.
Rajendran R, Sivapathasundharam B. Shafer's Text Book of Oral Pathology. 6th ed. India: Elsevier; 2009. p. 230.  Back to cited text no. 3
    
4.
Neville WB, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 3rd ed. India: Elsevier; 2009. p. 485-6.  Back to cited text no. 4
    
5.
Gary EL, Auclair LP, Gnepp RD. Surgical Pathology of the Salivary Glands. Vol. 25. Philadelphia: W.B. Saunder Co.; 1991. p. 238-44.  Back to cited text no. 5
    
6.
Pimentel MT, López Amado M, García Sarandeses A. Recurrent sialadenoma papilliferum of the buccal mucosa. J Laryngol Otol 1995;109:787-90.  Back to cited text no. 6
    
7.
Solomon MP, Rosen Y, Alfonso A. Intraoral papillary squamous cell tumor of the soft palate with features of sialadenoma papilliferum? Malignant sialadenoma papilliferum. Cancer 1978;42:1859-69.  Back to cited text no. 7
[PUBMED]    
8.
Shimoda M, Kameyama K, Morinaga S, Tanaka Y, Hashiguchi K, Shimada M, et al. Malignant transformation of sialadenoma papilliferum of the palate: A case report. Virchows Arch 2004;445:641-6.  Back to cited text no. 8
    
9.
Markopoulos A, Kayavis I, Papanayotou P. Sialadenoma papilliferum of the oral cavity: Report of a case and literature review. J Oral Maxillofac Surg 1997;55:1181-4.  Back to cited text no. 9
    
10.
Ubaidat MA, Robinson RA, Belding PJ, Merryman DJ. Sialadenoma papilliferum of the hard palate: Report of 2 cases and immunohistochemical evaluation. Arch Pathol Lab Med 2001;125:1595-7.  Back to cited text no. 10
    
11.
Freedman PD, Lumerman H. Sialadenoma papilliferum. Oral Surg Oral Med Oral Pathol 1978;45:88-94.  Back to cited text no. 11
    
12.
Maiorano E, Favia G, Ricco R. Sialadenoma papilliferum: An immunohistochemical study of five cases. J Oral Pathol Med 1996;25:336-42.  Back to cited text no. 12
    
13.
Nakahata A, Deguchi H, Yanagawa T, Yoshida H, Sato M, Hayashi Y. Coexpression of intermediate-sized filaments in sialadenoma papilliferum and other salivary gland neoplasms. J Oral Pathol Med 1990;19:313-8.  Back to cited text no. 13
    
14.
Brannon RB, Sciubba JJ, Giulani M. Ductal papillomas of salivary gland origin: A report of 19 cases and a review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:68-77.  Back to cited text no. 14
    
15.
Whittaker JS, Turner EP. Papillary tumours of the minor salivary glands. J Clin Pathol 1976;29:795-805.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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