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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 29  |  Issue : 5  |  Page : 72-74

Low-grade intraductal carcinoma of the parotid gland: Is it low-grade or locally aggressive?


Department of Plastic, Reconstructive and Aesthetic Surgery, İstanbul Medeniyet University, İstanbul, Turkey

Date of Submission24-Aug-2020
Date of Acceptance08-Oct-2020
Date of Web Publication17-Mar-2021

Correspondence Address:
Dr. Mehmet Gurler
Department of Plastic, Reconstructive and Aesthetic Surgery, İstanbul Medeniyet University, Eğitim Mah. Kadıköy/İstanbul 34722
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/tjps.tjps_93_20

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  Abstract 


Low-grade salivary ductal carcinoma is a salivary gland tumor with clinically indolent behavior, characterized by intraductal growth. From 1996 to 2019, 54 case reports of Low-Grade Intraductal Carcinoma (LGIDC) were published in the literature. In the present article, we introduce a patient in our clinic who underwent surgery due to a diagnosis of LGIDC, about whom we make new clinical comments. A 75-year-old male patient presented to our clinic with a complaint of a mass in the right parotid area. After multiple biopsies, magnetic resonance imaging (MRI), and operations, the patient health is fine and patient's follow-up continues, now in the postoperative 1st year, and there is no relapse sign on physical examination and MRI. After all, according to our clinical findings and observations, we thought that this tumor could possibly be considered a locally aggressive tumor that does not metastasize, similar to a basal cell carcinoma.

Keywords: Carcinoma, ductal, local aggressive, low grade, parotid, salivary, tumor


How to cite this article:
Gurler M, Ozturk MB, Atik B, Keskin ES. Low-grade intraductal carcinoma of the parotid gland: Is it low-grade or locally aggressive?. Turk J Plast Surg 2021;29, Suppl S1:72-4

How to cite this URL:
Gurler M, Ozturk MB, Atik B, Keskin ES. Low-grade intraductal carcinoma of the parotid gland: Is it low-grade or locally aggressive?. Turk J Plast Surg [serial online] 2021 [cited 2021 Dec 8];29, Suppl S1:72-4. Available from: http://www.turkjplastsurg.org/text.asp?2021/29/5/72/311443




  Introduction Top


Low-grade salivary ductal carcinoma was first described by Delgado et al. in 1996 as a salivary gland tumor with clinically indolent behavior, characterized by intraductal growth.[1] In 2005, the World Health Organization (WHO) described this tumor as “a rare, cystic, proliferative carcinoma that resembles the spectrum from atypical ductal hyperplasia of the breast to micropapillary and cribriform low-grade ductal carcinoma in situ.” According to the WHO's classification, it was defined as a subtype of cystadenocarcinoma and was termed low-grade cribriform cystadenocarcinoma (LGCCC). However, according to the WHO's 2017 classification, the entities, LGCCC, also known as “low-grade salivary duct carcinoma,” and (conventional) salivary duct carcinoma in situ are now collectively categorized as intraductal carcinomas, low-grade and high-grade, respectively.[2] From 1996 to 2019, 54 case reports of LGIDC were published in the literature, the largest study being a case series of 16 patients.[3] Although LGIDC is described as an indolent tumor with a good prognosis, there is inadequate information about its clinical course in the literature.[4] In the present article, we introduce a patient in our clinic who underwent surgery due to a diagnosis of LGIDC, about whom we make new clinical comments.


  Case Report Top


A 75-year-old male patient presented to our clinic with a complaint of a mass in the right parotid area for 1 year. On physical examination, a multilobular mass was detected in the right buccal region. The mass had a hard consistency and extended from the temporomandibular region to the lower boundary of the mandible [Figure 1]. The patient's facial nerve examination was normal. On magnetic resonance imaging (MRI) analysis, a shapely contoured mass lesion with well defined, measuring 26 mm × 21 mm × 27 mm, was observed just anterior to the superficial lobe of the right parotid gland [Figure 2]. The boundaries could not be clearly distinguished from that of the parotid gland. Liquid-leveling and debris were identified within the mass structure, and irregular solid components were observed within the mass wall. The cytological results of a fine-needle aspiration biopsy were interpreted as benign, consistent with the contents of a cyst. The patient's prediagnosis was considered to be pleomorphic adenoma, Warthin's tumor, or dermoid tumor, and an incisional biopsy was planned. Intraoperatively, the mass was observed to be macrocystic, multiloculated, and nonencapsulated, which differed from the prediagnosis [Figure 3]. Biopsies of the lesion were performed, and the operation was ended. The pathological diagnosis of the patient appeared to be LGIDC, which was confirmed by the reference pathology laboratory [Figure 4], [Figure 5]. A total parotidectomy was planned for the patient. However, the patient did not come for his follow-up clinical examinations, and applied to our clinic 1 year later, again with a complaint of an expansive mass in the right parotid area. On a repeat contrast-enhanced MRI examination, four hemorrhagic space-occupying lesions were identified, the largest measuring 32 mm × 30 mm. Total parotidectomy was carried out. The patient's follow-up continues, now in the postoperative 1st year and there is no relapse sign on physical examination and MRI. And also, there is no sign of facial nerve palsy [Figure 6].
Figure 1: Preoperative photograph of the patient before second operation

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Figure 2: Magnetic resonance imaging screening of patient before second operation

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Figure 3: Intraoperative photograph of the patient in second operation

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Figure 4: A massive tumor progressing with permeation, with a cribriform structure and distinctive boundaries, and accompanied by fibrous stroma (H and E, ×100)

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Figure 5: Mild cytologic atypia, in which cystic and oncocytic changes are seen (H and E, ×200)

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Figure 6: (a) Postoperative 1st year. (b) Postoperative 1st year (lateral view)

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The patient was informed verbally and consent formed was signed by the patient.


  Discussion Top


LGIDC is a rare tumor of the parotid gland. Due to its rarity, there is no definitive algorithm to follow for its diagnosis and treatment.

The long-term presence of a nonfixed soft lesion with painless growth, and the absence of pathologic findings on the facial nerve examination, were interpreted to confirm the presence of a benign lesion in our patient. However, the mass was observed to grow more quickly after the biopsy. According to Giovacchini et al., patients usually presented with an asymptomatic and slow-growing mass but on our patient, a rapid growing occurred after the incisional biopsy.[3] Moreover, we decided that this type of tumor maybe shows local aggressiveness such as basal cell carcinoma. Furthermore, the reason of that could not be an inoculation of tumor while we were taking biopsy. Because tumor expansion did not arised from biopsy taken parts.

MRI may be helpful in distinguishing between benign and malignant parotid masses. If a lesion is encapsulated and has regular boundaries, MRI may confirm its benign character.[5] However, since LGIDC lesions are pseudoencapsulated and have no regular boundaries, as in our case, it is more difficult to distinguish between benign and malignant lesions during the interpretation of MRI results. In our case, a cystic lesion was detected on ultrasonography, and the overall assessment, including the other findings, initially suggested the presence of a pleomorphic adenoma, and a type of benign tumor.

Instead of having biopsies in the first operation, obtaining frozen sections for pathology could have been used to make the distinction between benignancy and malignancy; however, it is not possible to distinguish between low grade and high grade on the frozen section analyses. In such cases, it can be difficult to decide whether to expand the scope of the surgery or keep it more regional.

In the literature, it is reported that conservative excision of the gland, without neck dissection or postoperative radiotherapy, is adequate for the treatment of LGIDC.[6] In our case, a total parotidectomy was performed because the recurrent mass was larger than 4 cm and appeared with new local-regional centers, suggesting that the tumor might have spread, progressing in a locally aggressive way.

Brandwein-Gensler et al. claimed that “the tumor cells form cribriform, sieve-like spaces, with a streaming effect as is seen in benign or atypical breast ductal proliferations” on microscopic examination.[6] And also, they saw dominant cysts and multiple small cysts. In our case, we reached similar signs and microscopic findings. Moreover, this finding shows us that diagnosis is correct and compatible with the literature.

The diagnosis of LGIDC should also be considered for lesions with cystic and benign findings, as in our case, and the surgical procedure should be planned accordingly. The literature defines LGIDC as an indolent tumor with a good clinical prognosis. However, in our case, the patient did not present for his follow-up examinations after the first surgery and applied to our clinic again 1 year later with the complaint of an expansive mass. The fact that new masses were detected in areas outside the biopsy site, but there were no findings in other regions of the face and neck that might indicate a mass or metastasis, suggested that this could be a tumor progressing in a locally aggressive way, rather than a low-grade lesion. This tumor could possibly be considered a locally aggressive tumor that does not metastasize, similar to a basal cell carcinoma. Early detection and operation and close follow-up of patient may be suitable for this type of tumor for avoiding local spread.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Delgado R, Klimstra D, Albores-Saavedra J. Low grade salivary duct carcinoma: A distinctive variant with a low grade histology and a predominant intraductal growth pattern. Cancer 1996;78:958-67.  Back to cited text no. 1
    
2.
Seethala RR. Update from the 4th ed ition of the World Health Organization classification of head and neck tumours: Preface. Head Neck Pathol 2017;11:1-2.  Back to cited text no. 2
    
3.
Giovacchini F, Bensi C, Belli S, Laurenti ME, Mandarano M, Paradiso D, et al. Low-grade intraductal carcinoma of salivary glands: A systematic review of this rare entity. J Oral Biol Craniofac Res 2019;9:96-110.  Back to cited text no. 3
    
4.
Kokabu S, Nojima J, Kayano H, Yoda T. Low-grade cribriform cystadenocarcinoma of the palatal gland: A case report. Oncol Lett 2015;10:2453-7.  Back to cited text no. 4
    
5.
Rice DH, Becker T. Magnetic resonance imaging of the salivary glands. A comparison with computed tomographic scanning. Arch Otolaryngol Head Neck Surg 1987;113:78-80.  Back to cited text no. 5
    
6.
Brandwein-Gensler M, Hille J, Wang BY, Urken M, Gordon R, Wang LJ, et al. Low-grade salivary duct carcinoma. Am J Surg Pathol 2004;28:1040-4.  Back to cited text no. 6
    


    Figures

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



 

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