Papillary cystadenocarcinoma of the parotid gland: a rare sub-variant of salivary gland adenocarcinoma

Dutta, Satya; Dey, Biswajit; Mishra, Jaya; Raphael, Vandana; Das, Jonali; Lynser, Donboklang

doi:10.4322/acr.2021.357

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Clinical Case Report and Review • Autops. Case Rep. 12 • 2022 • https://doi.org/10.4322/acr.2021.357 copy

Papillary cystadenocarcinoma of the parotid gland: a rare sub-variant of salivary gland adenocarcinoma

Authorship SCIMAGO INSTITUTIONS RANKINGS

Abstract

Papillary cystadenocarcinoma of the salivary gland is a very rare malignant neoplasm accounting for only 2% of all salivary gland lesions. In 1991 it was first included as a separate entity in the World Health Organization (WHO) classification of salivary gland tumors and in 2017 WHO Classification, the tumor was clubbed as a sub-variant of adenocarcinoma, not otherwise specified. It most commonly occurs in the major salivary glands. Herein we report a case of salivary papillary cystadenocarcinoma in a 54-year-old female, who presented with rapid enlargement of the right parotid swelling. Based on radiology and fine-needle aspiration cytology, a working diagnosis of the malignant tumor involving the superficial lobe of the right parotid gland was made. In view of the malignant nature of the swelling, superficial parotidectomy was done. The histopathology and immunohistochemistry of the mass confirmed the diagnosis of papillary cystadenocarcinoma of the right parotid. With the revised 2017 WHO classification of salivary gland tumors, it is important to report all rare subtypes in order to understand their biology and behavior.

Keywords:
Papillary cystadenocarcinoma; Salivary gland neoplasm; Parotid

Ref	# cases	Age	Sex	Size (cm)	Duration	Side	Grade	IHC
Ref	# cases	(Y)	Sex	Size (cm)	Duration	Side	Grade	IHC
			M-81.8%	NM	M 10.8 y	NA	NM	Positive CK (KL1 & K8.12), Vimentin & MAM-6;
5	9	a.m 45.7	F-18.2%	NM	M 10.8 y	NA	NM	Varying intensity for S-100 & NSE
						19 right, 13 left, 3 cases NM	NM	NM
6	35	a.m 58.8	M=F	0.4 to 0.6	1 m - 5 y	19 right, 13 left, 3 cases NM	NM	NM
		62	M	3x 2.5	6 m - 14 y	NA	LG	NM
7	2	60	F	4x 2	6 m - 14 y	NA	LG	NM
8	1	33	F	4x 3	5 m	Left	LG	NM
								Positive CK AE1/AE3 & CEA
9	1	62	M	1.5 X 1.5	NM	NM	LG	Weak positivity for S-100
								Negative Vimentin & SMA
10	1	34	M	6x 6	10 y	Right	LG	NM
11	1	55	M	3	3 m	Right	LG	Positive CK & EMA
11	1	55	M	3	3 m	Right	LG	Negative TTF-1
12	1	57	M	6x 5	2 y	Right	LG	Positive CK7, EMA & S-100
12	1	57	M	6x 5	2 y	Right	LG	Negative CEA, SMA, GFAP, ER, PR & Her2neu
13	1	40	F	1.5X0.5	10 y	Right	LG	NM
14	1	33	F	4x 3	5 m	Left	LG	NM
15	1	58	M	5x4x2	4 m	Right	NM	NM
16	1	55	M	6 × 5	3 y	Right	NM	Positive CK7
16	1	55	M	6 × 5	3 y	Right	NM	Negative CK20, p63 & Cerb2
17	1	47	M	9x6. 5x3	4 y	Right	IG	NM
18	1	51	M	3.3 × 3.2 × 2.8	4 y	Right	HG	NM
		a.m 49.55	M-66.7%	<3-29.6%	≤ 12 m- 59.3%	Right-55.6%	NM	NM
			F-33.3%	≥3- 70.4%	13-36 m-25.9%	Left-44.4%
19	19				>36 m- 14.8%
20	1	67	F	6x 5 cm	6 months	Right	LG	NM

Entity	Most common site	Tumor architecture	Tumor cells	Cytokeratin	Calponin/ SMA	Other IHC markers
PLGA	Minor salivary glands	Variable patterns of different proportions including trabecular, tubular, papillary, solid and cribriform patterns	tumor cells having pale nuclei with marked chromatin clearing	Positive for CK7, CK AE1/AE3	Variable	Positive for Vimentin and S-100
Papillary cystic variant of acinic cell carcinoma	Parotid glands	Transition of usual dense cellularity into papillary folds interspersed with cystic spaces.	Diverse tumor cell types including acinic cells, vacuolated cells, intercalated cells, non-specific glandular cells forming ‘hobnail cells’ and clear cells	Positive for CK7	Negative	Positive for DOG-1
Salivary duct carcinoma	Parotid glands	Invasive tumor forming cords, nests and cribriform glands in a desmoplastic stroma	Tumor cells have ample eosinophilic cytoplasm showing marked nuclear pleomorphism	Positive for CK7, CK AE1/AE3	Negative	Positive for Androgen receptor, Her2neu, and GCDFP-15
Muco epidermoid carcinoma	Parotid glands	Incompletely encapsulated or unencapsulated tumor showing papillary or glandular growth pattern	Varying proportions of Squamoid, intermediate cells, and mucophages	Positive for CK7, CK19	Negative	Positive for p63
Papillary cystadenocarcinoma	Parotid glands	Cysts with papillary endocystic projections	Cuboidal to tall columnar cells with vesicular nuclei and slightly granular cytoplasm	Positive for CK7, CK AE1/AE3	Negative	Negative for S-100, DOG-1, AR, Her2neu, GCDFP-15, p63

Hospital Universitário da Universidade de São Paulo Hospital Universitário da Universidade de São Paulo, Av. Prof. Lineu Prestes, 2565 - Cidade Universitária, 05508-000 - São Paulo - SP - Brasil, (16) 3307-2068, (16) 3307-2068 - São Paulo - SP - Brazil
E-mail: autopsy.hu@gmail.com

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[1] Authors’ contributions: Satya Dutta, Biswajit Dey, and Jonali Das wrote the manuscript. Jaya Mishra and Vandana Raphael diagnosed the histopathology of the case. Donboklang Lynser reported the radiology. All authors collectively proof read the manuscript and approved the final version for publication.