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Year : 2014  |  Volume : 57  |  Issue : 2  |  Page : 341-342
Adenocarcinoma arising in tailgut cyst: A rare case


Department of Pathology, Grant Medical College, Mumbai, Maharashtra, India

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Date of Web Publication19-Jun-2014
 

How to cite this article:
Patil P, Jibhkate SN, Pawar V, Valand A. Adenocarcinoma arising in tailgut cyst: A rare case. Indian J Pathol Microbiol 2014;57:341-2

How to cite this URL:
Patil P, Jibhkate SN, Pawar V, Valand A. Adenocarcinoma arising in tailgut cyst: A rare case. Indian J Pathol Microbiol [serial online] 2014 [cited 2022 Jan 18];57:341-2. Available from: https://www.ijpmonline.org/text.asp?2014/57/2/341/134744


Sir,

This article refers to a case of a 33-year-old male presenting with lower back swelling since childhood which became painful since 1 year. Pain was dull aching, more on sitting. Local examination revealed sacrcococcygeal fluctuant nontender oval swelling measuring about 7 × 5 × 1 cm having regular margin with extension between gluteal cleft. Overlaying skin was normal. Magnetic resonance imaging (MRI) with contrast confirmed a cystic mass in the retrorectal presacrococcygeal space extending in the left medial gluteal region. Three differential diagnoses of sacrococcygeal teratoma, tail gut cyst, and remote possibility of soft tissue sarcoma were offered by radiologist. The patient underwent a surgery (low anterior resection). The retrorectal cyst was excised completely and sent for histopathological examination. Macroscopically it was an unencapsulated irregular variegated solid cystic mass with areas of necrosis and hemorrhage. Cystic areas were containing yellow gelatinous material, while solid areas were grayish white firm, however, there were no hair follicles. Microscopy revealed a multiloculated cyst, which was lined by variety of epithelia ranging from stratified squamous, transitional, tall columnar, gastric, and mucinous epithelia, which at places was showing transition from one epithelium to other.[Figure 1] The cyst wall showed disorganized fascicles of smooth muscle and numerous blood vessels. It also showed malignant epithelial cells arranged in glandular and tubular pattern,[Figure 2] which were positive for p53, along with large areas of necrosis. Also seen are areas of hyalinization and calcification. However, elements derived from other germ layers such as skin adnexa, neural tissue, heterologus mesenchymal tissue such as cartilage and bone were not seen. So final histopathological diagnosis was given as adenocarcinoma arising in tailgut cyst.
Figure 1: Cyst lining showing transition into different epithelia like, squamous, transitional, tall columnar

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Figure 2: Adenocarcinoma in a retrorectal cystic hamartoma

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   Discussion Top


The retrorectal cystic hamartoma or tailgut cysts are rare congenital lesions derived from the vestigial elements of the embryonic hindgut. Embryo possesses a true tail caudal to the site of subsequent formation of the anus. The primitive hindgut extends into this tail, which usually completely regresses. However, sometimes tailgut remnants in the retrorectal space persists giving rise to the tailgut cyst. [1] Owing to the rarity of the condition and its nonspecific presentation, diagnosis is frequently late. Usually it presents as a asymptomatic mass but can also present with rectal bleeding, pain with defecation, lower back pain, and urinary problems. [2] It may get secondarily infected, and often misdiagnosed as pilonidal cyst, anorectal fistula, or a recurrent retrorectal abscess. [3] Malignant change is extremely rare. Major differential diagnoses like epidermoid cyst, dermoid cyst, teratoma, rectal duplication cysts are excluded by considering the structure and epithelial lining of the cysts. [4] Presence of local invasion indicates malignancy. On review of literature, malignant change within a retrorectal cystic hamartoma had been documented in only 32 cases; in which most were adenocarcinomas or neuro-endocrine tumors and rarely carcinoid tumors are described. Malignant cells are positive for p53, Ki-67, and p21. [5] Some believe that dysplasia-carcinoma sequence, which is established in colon, also exist in tailgut cyst. Hence these lesions should be excised early to reduce the risk of malignant change and other complications. [3]

 
   References Top

1.Au E, Anderson O, Morgan B, Alarcon L, George ML. Tailgut cyst: Report of two cases. Int J Colorectal Dis 2009;24:345-50.  Back to cited text no. 1
    
2.Färber A, Nickel J, Brinckmann W, Fietkau R, Pommerencke R, Andresen R. Differential diagnosis of tailgut cyst in the case of a rectal carcinoma with presacral mass. Rontgenpraxis 2003;55:78-83.  Back to cited text no. 2
    
3.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. 3
    
4.Dahan H, Arrivé L, Wendum D, Docou le Pointe H, Djouhri H, Tubiana JM. Retrorectal developmental cysts in adults: Clinical and radiologic-histopathologic review, differential diagnosis, and treatment. Radiographics 2001;21:575-84.  Back to cited text no. 4
    
5.Moreira AL, Scholes JV, Boppana S, Melamed J. p53 mutation in adenocarcinoma arising in retrorectal cyst hamartoma (tailgut cyst): Report of 2 cases - an immunohistochemistry/ immunoperoxidase study. Arch Pathol Lab Med 2001;125:1361-4.  Back to cited text no. 5
    

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Correspondence Address:
Shubhangi Narayanrao Jibhkate
Quarte No. 512, New RMO Quarter, Sion Hospital, Sion, Mumbai, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.134744

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    Figures

  [Figure 1], [Figure 2]

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