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  Table of Contents  
Year : 2015  |  Volume : 52  |  Issue : 4  |  Page : 644-645

Low-grade papillary adenocarcinoma of minor salivary glands in pregnancy

1 Department of Oral Medicine and Radiology, Yenepoya Dental College, Yenepoya University, Mangalore, Karnataka, India
2 Department of Oral Medicine and Radiology, Goa Dental College, Goa, India
3 Department of Oral and Maxillofacial Pathology, Goa Dental College, Goa, India
4 Department of Oral and Maxillofacial Pathology, Yenepoya Dental College, Yenepoya University, Mangalore, Karnataka, India

Date of Web Publication10-Mar-2016

Correspondence Address:
R V Prabhu
Department of Oral Medicine and Radiology, Yenepoya Dental College, Yenepoya University, Mangalore, Karnataka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0019-509X.178438

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How to cite this article:
Prabhu R V, Dinkar A, Spadigam A, Prabhu V. Low-grade papillary adenocarcinoma of minor salivary glands in pregnancy. Indian J Cancer 2015;52:644-5

How to cite this URL:
Prabhu R V, Dinkar A, Spadigam A, Prabhu V. Low-grade papillary adenocarcinoma of minor salivary glands in pregnancy. Indian J Cancer [serial online] 2015 [cited 2020 Jul 14];52:644-5. Available from:


A 34-year-old female in her 34th week of gestation reported with pain and a sudden increase in the size of the swelling on the left palate [Figure 1], which was otherwise present since last 6-7 months. She also complained of watering from the left eye and nasal obstruction on the left side with difficulty in swallowing and speech. Computed tomography scan of Neck and Paranasal Sinuses showed Large heterogeneously enhancing soft tissue density mass in left maxillary sinus with erosion of its bony walls [Figure 2]. Fine needle aspiration cytology showed epithelial and myoepithelial cells arranged in clusters and single that was in favor of minor salivary gland (MSG) neoplasm. Histopathological features [Figure 3], were suggestive of a Low-Grade Papillary Adenocarcinoma (LGPA) of the MSG. Immunohistochemistry was positive for Cytokeratin, Vimentin, and S-100 suggestive of myoepithelial cell differentiation.
Figure 1: Intra oral photograph showing massive swelling of the left palate

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Figure 2: Computed tomography image – axial view showing soft tissue mass in the left maxillary sinus. Posterolaterally extending- into infra-temporal fossa, antero laterally into the buccal spaces and medially into the nasal cavity. Superiorly extending into the extraconal compartment of the orbit, inferiorly upto the level of the hard palate causing bony erosion and infero-.medially into the nasopharynx. In addition near complete disruption of normalarchitecture of ethmoid sinus was seen no intra cranial extension and cervical lymphadenopathy was noticed

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Figure 3: Photomicrograph under ×40 showing solid nests and clusters of fairly uniform, dark staining epithelial cells proliferating in a loose fibro vascular connective tissue stroma with red blood corpuscles extravasation. Areas of papillary projections supported by a thin core of fibrous connective tissue within cystic spaces were appreciated

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Preterm delivery was conducted in her 36th week of gestation at a leading oncology centre in Mumbai. Left total hemimaxillectomy was done with neck dissection followed by reconstruction [Figure 4]. Left level 2 lymph nodes were found to be negative. No evidence of any local recurrence has been noticed since last 7 years.
Figure 4: Post-operative intra-oral photograph showing reconstructive flap

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LGPA of the MSG is a rare, slow growing tumor. The main concern of this neoplasm is its local recurrence over several years and greater tendency to metastasize.[1] LGPA presenting in pregnancy is a very rare combination. Suppression of immunity is seen in the first 20 weeks of gestation. Estrogen activity is increased by 100 fold and progesterone levels by 10 fold which may stimulate latent breast carcinoma to an active growth during immunosuppression.[2]

Polymorphous low-grade adenocarcinoma of MSG origin has microscopic features that mimic those of infiltrating lobular carcinoma of breast. Adenoid cystic carcinoma (ACC) of salivary gland origin is indistinguishable histologically from ACC breast and vulva.[3] It is suggested that the estrogen acts as a promoting factor for some salivary gland neoplasm based on the study in which 8 of 9 salivary gland tumors had Estrogen Receptor (ER) levels that would be considered hormone dependent in breast carcinoma.[4] ER has been reported in minority of cases of ACC, mucoepidermoid carcinoma, and salivary duct carcinoma.[5]

Very few cases, concerning pregnancy coexistent with head, and neck malignancies are reported. To the best of our knowledge, there is not a single case reported in the medical literature regarding analyzing the ER or association of pregnancy with LGPA of MSG. Our case is probably the first to mention this observation which raises the suspicion about a possible etiologic relation between pregnancy and LGPA of MSG.

 » Acknowledgment Top

We would like to thank Dr. Vikas Dhupar, Professor and Head and Dr. Francis Akkara, Professor, Department of Oral and Maxillofacial Surgery, Dr. Anita Dhupar, Professor, Department of Oral and Maxillofacial Pathology, Goa Dental College and Hospital, Goa, India for their valuable contribution.

 » References Top

Allen MS Jr, Fitz-Hugh GS, Marsh WL Jr. Low-grade papillary adenocarcinoma of the palate. Cancer 1974;33:153-8.  Back to cited text no. 1
Yue W, Wang JP, Li Y, Fan P, Liu G, Zhang N, et al. Effects of estrogen on breast cancer development: Role of estrogen receptor independent mechanisms. Int J Cancer 2010;127:1748-57.  Back to cited text no. 2
Abrao FS, Marques AF, Marziona F, Abrao MS, UchoaJunqueira LC, Torloni H. Adenoid cystic carcinoma of Bartholin's gland: Review of the literature and report of two cases. J SurgOncol 1985;30:132-7.  Back to cited text no. 3
Dimery IW, Jones LA, Verjan RP, Raymond AK, Goepfert H, Hong WK. Estrogen receptors in normal salivary gland and salivary gland carcinoma. Arch Otolaryngol Head Neck Surg 1987;113:1082-5.  Back to cited text no. 4
Auclair PL. Cystadenocarcinoma. Pathology and genetics of head and neck tumours. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. World Health Organization Classification of Tumours. Lyon: IARC Press; 2005. p. 232-6.  Back to cited text no. 5


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

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