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CORRESPONDENCE
Year : 2019  |  Volume : 15  |  Issue : 1  |  Page : 261-263

Adenocarcinoma in a tailgut cyst: A rare case report


1 Department of Radiology, Chirayu Medical College and Hospital, Bhopal, Madhya Pradesh, India
2 Department of Pathology, Chirayu Medical College and Hospital, Bhopal, Madhya Pradesh, India
3 Department of Surgery, Chirayu Medical College and Hospital, Bhopal, Madhya Pradesh, India

Date of Web Publication13-Mar-2019

Correspondence Address:
Dr. Sandeep S Ojha
Department of Pathology, Chirayu Medical College and Hospital, Bhopal Indore Highway, Bairagarh, Bhopal - 462 030, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcrt.JCRT_212_17

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 > Abstract 


Tailgut cysts (TGCs) are rare congenital lesions derived from the remnants of primitive hindgut and are usually lined by squamous, transitional, or glandular epithelium. Malignant transformation in TGC may occur which is still rarer. Most common malignancies that arise from these cysts are adenocarcinomas. Preoperative diagnosis is difficult as high degree of suspicion is required for the diagnosis. We report here a case of adenocarcinoma arising in a tale gut cyst diagnosed preoperatively and till date very few cases have been reported in literature.

Keywords: Adenocarcinoma, malignancy, tailgut cysts


How to cite this article:
Valecha J, Ojha SS, Sharma A, Nilkanthe R. Adenocarcinoma in a tailgut cyst: A rare case report. J Can Res Ther 2019;15:261-3

How to cite this URL:
Valecha J, Ojha SS, Sharma A, Nilkanthe R. Adenocarcinoma in a tailgut cyst: A rare case report. J Can Res Ther [serial online] 2019 [cited 2019 Aug 20];15:261-3. Available from: http://www.cancerjournal.net/text.asp?2019/15/1/261/244444




 > Introduction Top


Tailgut cysts (TGCs) are rare benign congenital lesions derived from vestigial remnants of embryonic hindgut and located in retro-rectal or presacral area.[1] These remnants usually completely regress, however, sometimes persists giving rise to the TGC. Sometimes, malignancies may arise in TGC but are rare and reported as case reports. Malignancies that have been reported in TGC include adenocarcinoma, carcinoid tumors, neuroendocrine carcinomas, endometrioid carcinoma, adenosquamous carcinoma, transitional cell carcinoma, and sarcoma.[2],[3],[4] Tumors arise from cystic lesions of the embryological postanal gut which contain a variety of lining epithelia including glandular, squamous, cuboidal, and transitional.[5] The diagnosis of this tumor is difficult and usually late, because of the rarity of the condition and nonspecific presentation. We report this case because of rarity of the lesion and describing the role of radiology as well as pathology in diagnosis of the condition.


 > Case Report Top


A 38-year-old male came with a history of increased frequency and urgency of micturition and defecation, abdominal fullness, and pain for 6 months duration. There was no history of any fever, palpable mass, or bleeding from any sites. Ultrasound of abdomen was asked for and showed an ill-defined mass in the presacral area. Magnetic resonance imaging (MRI) showed a cystic mass measuring 7.7 cm × 4 cm with craniocaudal extent of 3 cm containing amorphous calcification and soft tissue component along posterior aspect in presacral region causing anterior displacement of rectum. Mass appears to be adherent to serosal aspect of rectum. The solid component shows enhancement on contrast with extension into lateral pelvic wall, sacral bone and also shows marrow edema. Cystic component shows heterogeneous hyperintense signal on T2 and T1 sequence and shows no enhancement on contrast image [Figure 1]. Based on radiology, a diagnosis of presacral tumor mass was considered, and differentials of immature teratoma or a soft-tissue sarcoma were considered.
Figure 1: T1 sagittal fat sat postcontrast (a) showing bladder (orange arrow), rectum (green arrow) and tumor mass (pink arrow) and T2 sagittal sequence (b) showing bladder (orange arrow), rectum (green arrow) and tumor mass (pink arrow)

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CT-guided fine-needle aspiration from the mass was asked for and showed many scattered as well as clusters of malignant epithelial cells with features consistent with adenocarcinoma. Endoscopy of anorectal region was performed to rule out invasion from a rectal primary; however, it does not show any growth or ulceration or mucosal thickening. Mass was palpable on per rectum examination. Radiology was again reviewed to look for any other possibilities and the possibility of an adenocarcinoma arising in a TGC was considered based on clinical, pathological, and radiological findings. Surgical excision was done and specimen was received in histopathology department of our laboratory. On gross examination, the tumor was received in bits and pieces, few of them were flat and few nodular. One surface of the flat tissue showed irregular lining with areas of necrosis. Representative sections were submitted from the tissue pieces and it showed features of a poorly differentiated adenocarcinoma with solid, glandular, and papillary growth pattern. Focal areas show extracellular mucin with few signet ring cells. At one focus pseudo-stratified columnar lining was seen with severe dysplasia suggesting that the cyst was at one time lined by pseudostratified columnar epithelial cells and the tumor may have derived from this lining [Figure 2]. Thus, a final diagnosis of adenocarcinoma arising in a tailgut cyst was proposed. The patient was discharged and came back after 6 months with a metastatic focus in lung; and after this diagnosis, he was lost to follow-up.
Figure 2: Section showing cyst wall lined by a pseudostratified columnar epithelium with dysplasia and a foci of invasion (arrow) (inset showing adenocarcinoma with mucin) (×400)

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 > Discussion Top


TGCs are rare congenital lesion occurring in the presacral space and are supposed to be derived from hindgut remnants.[5] TGC should be distinguished from other lesions which may be seen in retro-rectal space such as epidermal cysts, rectal duplication cysts, teratoma, and anterior sacral meningocele.[6] TGCs are commonly found in middle-aged females, however, can be seen in all age groups.[6] Most of the cases remain asymptomatic, and the cyst is discovered as an incidental finding while evaluating for other causes. Symptoms are usually nonspecific with most of the symptoms related to compression of the adjacent structures like rectum and ureter like abdominal pain tenesmus and urinary frequency.[6] Clinical presentation of the lesion ranges from infection, abscess, and fistulas. Most of the TGC are benign; however, malignant transformations of the epithelial components have been reported as case reports in English literature.[2] Most common type of the malignancies reported in literature was of type adenocarcinoma and carcinoid tumor. Although the most common type, there are only <30 reported cases so far to the best of our knowledge.[7] Preoperative diagnosis is usually difficult as there are very few cases reported and a high index of suspicion is required for the diagnosis. On MRI, TGC classically shows low-signal intensity on T1-weighted images and high signal intensity on T2-weighted images, however, it may vary based on the contents.[8] Fine-needle aspiration cytology (FNAC) can be an easy, minimally invasive and less traumatic procedure and can be helpful in preoperative diagnosis of the case as in our case. All TGC should be assessed for malignancy in view of literature mentioning a risk of development of carcinoma within them. There is no definite treatment guidelines for TGC-associated adenocarcinomas because of very few cases reported so far. Surgery is the diagnostic and therapeutic tool and adjuvant radiation therapy, and chemotherapy may be applied to kill any residual tumor to prevent recurrence.[9] In our case, the patient presented after 1 year postsurgery with a metastatic deposit in lungs and was lost to follow-up after that.


 > Conclusion Top


Tailgut cyst should be considered in any case with presacral mass not arising from rectum and with a cystic component. Malignancy should be suspected in cases with large mass with solid cystic components and calcification. FNAC can be used as a helpful tool for preoperative diagnosis and subsequent management.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 > References Top

1.
Cho BC, Kim NK, Lim BJ, Kang SO, Sohn JH, Roh JK, et al. Acarcinoembryonic antigen-secreting adenocarcinoma arising in tailgut cyst: Clinical implications of carcinoembryonic antigen. Yonsei Med J 2005;46:555-61.  Back to cited text no. 1
    
2.
Chhabra S, Wise S, Maloney-Patel N, Rezac C, Poplin E. Adenocarcinoma associated with tail gut cyst. J Gastrointest Oncol 2013;4:97-100.  Back to cited text no. 2
    
3.
Vinciguerra GL, Mercantini P, La Torre M, Pilozzi E, Ziparo V, Vecchione A, et al. Transitional cell carcinoma of the retrorectal space arisen in tailgut cyst: A case report and review of the literature. Int J Surg Pathol 2014;22:280-5.  Back to cited text no. 3
    
4.
Moulopoulos LA, Karvouni E, Kehagias D, Dimopoulos MA, Gouliamos A, Vlahos L, et al. MR imaging of complex tail-gut cysts. Clin Radiol 1999;54:118-22.  Back to cited text no. 4
    
5.
Jang SH, Jang KS, Song YS, Min KW, Han HX, Lee KG, et al. Unusual prerectal location of a tailgut cyst: A case report. World J Gastroenterol 2006;12:5081-3.  Back to cited text no. 5
    
6.
Prasad AR, Amin MB, Randolph TL, Lee CS, Ma CK. Retrorectal cystic hamartoma: Report of 5 cases with malignancy arising in 2. Arch Pathol Lab Med 2000;124:725-9.  Back to cited text no. 6
    
7.
Haydar M, Griepentrog K. Tailgut cyst: A case report and literature review. Int J Surg Case Rep 2015;10:166-8.  Back to cited text no. 7
    
8.
Aflalo-Hazan V, Rousset P, Mourra N, Lewin M, Azizi L, Hoeffel C, et al. Tailgut cysts: MRI findings. Eur Radiol 2008;18:2586-93.  Back to cited text no. 8
    
9.
Maruyama A, Murabayashi K, Hayashi M, Nakano H, Isaji S, Uehara S, et al. Adenocarcinoma arising in a tailgut cyst: Report of a case. Surg Today 1998;28:1319-22.  Back to cited text no. 9
    


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  [Figure 1], [Figure 2]



 

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