Sialadenoma papilliferum: a case report and immunohistochemical study review

June-Ho Byun; Dong-Chul Kim; Gyung-Hyuck Go; Bong-Wook Park

doi:10.5125/jkaoms.2010.36.6.533

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Byun, Kim, Go, and Park: Sialadenoma papilliferum: a case report and immunohistochemical study review

Original Article

J Korean Assoc Oral Maxillofac Surg 2010;36(6):533-537.

Published online: 7 January 2010

DOI: https://doi.org/10.5125/jkaoms.2010.36.6.533

Sialadenoma papilliferum: a case report and immunohistochemical study review

June-Ho Byun^1,^∗, Dong-Chul Kim^2,^∗, Gyung-Hyuck Go², Bong-Wook Park¹

¹Department of Oral and Maxillofacial Surgery, School of Medicine, Institute of Health Sciences, Jinju, Korea

²Department of Pathology, School of Medicine, Gyeongsang National University, Jinju, Korea

^∗ These two authors contributed equally to this work.

Bong-Wook Park Department of Oral and Maxillofacial Surgery, School of Medicine, Institute of Health Science, Gyeongsang National University Chilam-dong 90, Jinju, Gyeongnam, 660-702, Korea. TEL: +82-55-750-8264 FAX: +82-55-761-7024 E-mail: parkbw@gnu.ac.kr

Received 27 August 201030 November 2010 Accepted 17 December 2010

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Sialadenoma papilliferum (SP) is a rare benign neoplasm that normally arises from the minor salivary glands, particularly in the palate. SP is normally encountered in older men with an exophytic papillary surface growth. In the present study, an SP of the hard palate of a 69-year-old woman was examined immunohistochemically. Myoepithelial cell markers, such as S-100, smooth muscle actin and vimentin, were observed in the basal or luminal layer of tumor cells, indicating that myoepithelial cells participate in the pathogenesis of SP. In addition, cytokeratin 7 was also strongly detected in the tumor cells, suggesting that excretory ductal epithelial cells have a role in its histogenesis. A review of the literature of immunohistochemical studies on SP showed that the expression and co-expression of cytokeratins and myoepithelial cell markers have been reported in tumor cells. These results suggested that excretory duct cells and myoepithelial cells participate in the pathogenesis of SP.

Keywords: Sialadenoma papilliferum, Hard palate, Immunohistochemistry.

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Fig. 1.

Perioperative radiologic and clinical features. A. In preoperative MRI view, well demarcated firm mass was observed under the palatal mucosa (white arrow). B. After removal of tumor, intact hard palatal bone was detected. C. Main mass (∗), approximately 1.5x1 cm size, was resected with peripheral normal tissues. D. After 6 months of operation, completely healed palatal mucosa was observed. (MRI: magnetic resonance imaging)

Fig. 2.

Photomicrograph of the lesion.(H&E staining) A. The central portion of the tumor was contained hyperplastic squamous epithelium connected to surface mucosa, whereas the remaining tumor showed glandular (arrow) and projecting papillary portion (arrowhead).(original magnification x12) B. Ductal proliferation and dilation with papillary folds were observed in the glandular portion under moderate magnification. (original magnification x40) C. High magnification image of a region showing papillary projection. Papillary fronds were lined by a double row of cells composed of luminal columnar cells and short cuboidal or basaloid cells.(original magnification x200) D. High magnification glandular portion. Some mucin-secreting cells (arrow) were occasionally observed among lining columnar cells of the dilated duct (original magnification x200)

Fig. 3.

Immunohistochemical findings of the lesion. A, B. CK 7 was strongly expressed, but CK20 was absent in tumor cells.(original magnification x100) C. S-100 protein was moderately expressed in tumor cell luminal and basal layers.(original magnification x100) D. SMA was strongly expressed in the basal layer of tumor cells (arrow), but was not observed in the luminal columnar cells.(original magnification x200) E. Vimentin was weakly expressed in the basal layer of tumor cells.(original magnification x200) F. CEA expression was not observed in any tumor cell layer. (original magnification x100) (CK7: cytokeratin 7, CK20: cytokeratin 20, S-100: S-100 protein, SMA: smooth muscle actin, CEA: carcinoembryonic antigen)

Table 1.

Primary antibodies, staining methods, and results of immunohistochemical staining

Antibody	Dilution	Source	Immunostaining intensity
Antibody	Dilution	Source	Basal layer	Luminal layer
CK 7	1:1,500	Zymed (CA, USA)	+++	+++
CK 20	1:1,500	Neomarker (CA, USA)	−	−
S-100	1:10	DAKO (Denmark)	++	++
SMA	1:1,500	Neomarker (CA, USA)	+++	−
Vimentin	1:20	DAKO (Denmark)	+	−
CEA	1:20	DAKO (Denmark)	−	−

(CK: cytokeratin, SMA: smooth muscle actin, CEA: carcinoembryonic antigen)

Table 2.

Summary of immunohistochmical studies conducted on sialadenoma papilliferum

Authors	Number of case/Location	Result of immunohistochemistry
Nakahata et al¹⁰, 1990	1 / No description	CK (+++)¹, Vimentin (++)², Desmin (+)³: co-expression
		CK 19, S-100: Pos in luminal cells
Maiorano et al⁹, 1996	4/ Hard palates and 1/ Cheek	1st subset of basal cells CK 14, SMA, S-100, Vimentin, GFAP: Pos
		2nd subset of basal cells SMA, Vimentin, S-100, GFAP: Neg
		CK 7, CEA, EMA, Vimentin, S-100: Pos
Ubaidat et al⁶, 2001	2/ Hard palates	GFAP, Desmin, CK 20, MSA: Neg
		CK 19, 14, 13, 7, 8: Pos
Gomes et al¹³, 2004	2/ Hard palates	Vimentin, SMA: Neg
		CK 7, 17, 19: Pos: CK 20: Neg
Bobos et al¹², 2003	1/ Bronchus	S-100, EMA: Pos in epithelium
		SMA, S-100: Pos in myoepithelium
		CKs, EMA, CEA: Pos in luminal cells
Shimoda et al², 2004	1 Malignant case/	SMA, S-100, Vimentin: Pos in basal cells
	Hard palate	P53, CKs, EMA, S-100, CEA: Pos in cancer cells
		CKs: Pos
Mahajan et al¹, 2007	1/ Cheek	Vimentin, SMA: Neg
		Basal layer: CK 7 (+++), S-100 (++), SMA (+++),
Current case	1/ Hard palate	Vimentin (+)
		Luminal layer: CK 7 (+++), S-100 (++), SMA (−)⁴
		CK 20, CEA: Neg in all tumor cells

(+++¹: strong, ++²: moderate, +³: weak, −⁴: negative, Pos: positive expression, Neg: negative expression, CK: cytokeratin, SMA: smooth muscle actin, GFAP: glial fibrillary acidic protein, EMA: epithelial membrane antigen, MSA: muscle-specific actin, CEA: carcinoembryonic antigen)

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