Short Communication
Polymorphous adenocarcinoma transforming to Adenoid cystic carcinoma -
A case report
A.K. Anjali
a
, Sandhya Tamgadge
a
, Treville Pereira
a, *
, Rutuja G. Vidhale
a
, S. Sudhamani
b
a
Department of Oral Pathology, D.Y.Patil Dental College, D.Y.Patil University, India
b
Department of General Pathology, D.Y.Patil MedicalCollege, D.Y.Patil University, India
ARTICLE INFO
Keywords:
Adenoid cystic carcinoma
Immunohistochemical staining
Polymorphous adenocarcinoma
ABSTRACT
Polymorphous adenocarcinoma is an indolent salivary gland carcinoma characterized by cytologic uniformity and
architectural diversity, predominantly affecting minor salivary glands, in particular the hard and soft palate.
Adenoid cystic carcinoma (ACC) is an uncommon type of adenocarcinoma, a kind of malignant growth that starts
in glandular tissues. It most regularly emerges in the major and minor salivary organs of the head and neck. This
paper reports an instance of a 59-year-old male patient who was determined to have Polymorphous adenocar-
cinoma which ultimately ended up being Adenoid cystic carcinoma.
1. Introduction
Salivary gland tumors are a group of diverse lesions with rare and
variant morphogenetic features. Polymorphous adenocarcinoma is one
such variant characterised with cytologic uniformity and architectural
diversity predominantly affecting the minor salivary glands. This article
discusses one such case wherein the histopathological examination and
Immunohistochemical (IHC) investigation resulted in a contrasting
diagnosis.
2. Case report
A 59-year-old male patient complained of painless swelling on the left
side of the face for 6 months (Fig. 1) The swelling was small in size which
eventually grew in size measuring about 10x8x5cm. The patient had a
history of cigarette smoking 5 to 7 times per day for 35–40 years. The
histopathological diagnosis was given as Polymorphous low-grade
Adenocarcinoma (PLGA) (Figs. 2, 3 Fig. 4). IHC was performed for
further validation of the diagnosis using different markers namely Ki67,
p40, CD117, EMA (Figs. 5–8).
3. Discussion
The new term Polymorphous Low-Grade Adenocarcinoma (PLGA)
was coined in 1984 by Evans and Batsakis et al. [1]. However, it is very
rare for PAC to show high grade transformation, in this case there is a
marked transformation seen [2]. The tumor stroma consists of both
mucus and hyaline areas where the tumor cells are arranged in various
morphological patterns such as solid, cribriform, duct- like, papillary and
tubular patterns including morpho diversity and are separated by fibro-
vascular stroma. Nonetheless, IHC staining for PAC showed negative for
myoepithelial markers such as alpha-SMA and p40 showed diffuse pos-
itivity for PAC whereas it does not stain ACC [3,4]. C-kit protein marker
or CD117 is a transmembrane tyrosine kinase growth factor receptor
having its role in diagnosing ACC along with clinical findings as evalu-
ated by Lee et al., which showed no predictive value for prognosis and
recurrence. In this study, we have explored the expression of CD117 in
ACC and PLGA. Despite affecting the differentiation and function of mast
cells, hematopoietic progenitor cells, germ cells, and melanocytes
through its interaction with the c-kit receptor, CD117 have not shown
any direct relation with the pathogenesis of ACC [5]. In the current study,
we have encountered all the three patterns of ACC which showed strong
diffuse cytoplasmic staining when compared to PLGA which showed
weak cytoplasmic staining. Adding to this, p40 immunohistostaining
showed negative for ACC also correlating to the fact that PLGA has a
consistent p40 immunophenotype thus making it a diagnostic tool in
differentiating the two [6].
4. Conclusion
In the above study we examined the tumor which was diagnosed as
PAC using histopathological examination. However, with further inves-
tigation using IHC it revealed a transition from the previous diagnosis,
* Corresponding author.
E-mail addresses: anju.innova@gmail.com (A.K. Anjali), sandhya.tamgadge@
gmail.com (S. Tamgadge), trevillepereira@gmail.com (T. Pereira), rutuja.
vidhale@gmail.com (R.G. Vidhale), dr.sudhamani@gmail.com (S. Sudhamani).
Contents lists available at ScienceDirect
Advances in Oral and Maxillofacial Surgery
journal homepage: www.editorialmanager.com/adoms/default.aspx
https://doi.org/10.1016/j.adoms.2021.100079
Received 25 March 2021; Accepted 15 April 2021
Available online 19 April 2021
2667-1476/© 2021 The Authors. Published by Elsevier Ltd on behalf of British Association of Oral and Maxillofacial Surgeons. This is an open access article under the
CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Advances in Oral and Maxillofacial Surgery 3 (2021) 100079