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 Table of Contents     
UNUSUAL CASE
Year : 2019  |  Volume : 15  |  Issue : 3  |  Page : 262-264
 

Laparoscopic approach to tailgut cyst (retrorectal cystic hamartoma)


1 Department of Surgery, Hospital Quironsalud San José, Madrid, Spain
2 Department of Critical Care Medicine, Hospital San Rafael, Madrid, Spain
3 Department of Surgery, Ministry of Defence, IMIDEF, Madrid, Spain
4 Department of Medicine, School of Medicine, Complutense University, Madrid, Spain

Date of Submission03-Apr-2018
Date of Acceptance20-Sep-2018
Date of Web Publication4-Jun-2019

Correspondence Address:
Martín Gascón Hove
Department of Surgery, Hospital Quironsalud San José, Madrid 28002
Spain
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jmas.JMAS_71_18

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 ¤ Abstract 

Retrorectal cystic hamartomas, or tailgut cysts, are complex congenital cystic lesions which arise from embryologic tissues. Fewer than 200 cases have been reported worldwide, with women outnumbering men by 3:1. They are asymptomatic in 50% of the cases; the remainder present with back pain or mass effect as the most common symptoms. Malignant transformation rarely occurs. Guided biopsy is controversial, while surgery is the therapy of choice. We report the case of a 31-year-old woman complaining about perineal and vague lower abdominal pain, who was submitted to magnetic resonance imaging, which revealed a multilocular cystic, well-circumscribed retrorectal mass. Subsequently, laparoscopic excision was successfully accomplished. Operative time was 175 min. Intra- and post-operative course was uneventful. Hospital stay was 75 h. While any malignancy suspicion should lead to open surgery, given the risk of rupture, we support the benefits of laparoscopy may also be applied.


Keywords: Congenital disease, cysts, presacral space, retroperitoneal tumour, retrorectal hamartoma


How to cite this article:
Hove MG, Gil JM, Rodríguez TS, Lomas &F, Casajús JP, López-Farré A, Guijarro Jd. Laparoscopic approach to tailgut cyst (retrorectal cystic hamartoma). J Min Access Surg 2019;15:262-4

How to cite this URL:
Hove MG, Gil JM, Rodríguez TS, Lomas &F, Casajús JP, López-Farré A, Guijarro Jd. Laparoscopic approach to tailgut cyst (retrorectal cystic hamartoma). J Min Access Surg [serial online] 2019 [cited 2019 Jun 17];15:262-4. Available from: http://www.journalofmas.com/text.asp?2019/15/3/262/245155



 ¤ Introduction Top


Tailgut cysts are an uncommon[1] and complex entity in adults and children, which arise from embryological tissues. If they show malignant transformation, retrorectal cystic hamartomas (RCHs) are more frequently diagnosed as adenocarcinoma or carcinoid. Complete surgical excision is the treatment of choice, and definitive diagnosis is based on pathological examination of the specimen.[2]

The most commonly described surgical techniques are through standard Kraske or open abdominal approach. Laparoscopic surgery of RCHs has recently been reported and allows complete excision with low morbidity. Potential benefits should include less post-operative pain, shorter hospital stay, less blood loss, faster recovery time, fewer complications and better cosmesis.[3]

We communicate the excision of a tailgut cyst utilising a conventional laparoscopic procedure. The authors would like to underscore that, despite this technique has been applied to various surgical procedures, very few RCH excisions have been published.


 ¤ Case Report Top


A 31-year-old female presented abdominal discomfort and perineal and vague lower abdominal pain. Her medical history and her physical examination were irrelevant. Digital rectal examination revealed an extraluminal tumour bulging from the posterior rectal wall. The magnetic resonance imaging (MRI) examination showed a multilocular, well-defined cystic mass of 6 × 5 cm × 5 cm × 8 cm in the presacral space [Figure 1]. It displaced the rectum and the inferior aspect of the levator ani anteriorly and showed no evidence of invasion or distant metastases. Surgical access was gained with a 12-mm supraumbilical port followed by one 10-mm and three 5-mm ports in both the right upper/lower and left upper/lower quadrants. The mesorectal dissection from the sacral promontory was continued up to the level of puborectalis sling and the levator ani muscles [Figure 2]. The cyst was accidentally opened before last adhesion was excised and approximately 2 ml of mucoid fluid were aspirated. Before specimen removal, rectal integrity and haemostasis of the operative field were ensured.
Figure 1: Sagittal magnetic resonance imaging view of cystic mass in the retrorectal space

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Figure 2: Resection of the specimen from adjacent tissues

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Operative time was 175 min. A drain was placed and removed 48 h after surgery. The patient was discharged 75 h after surgery without specific analgesia requirements (only on-request conventional analgesia) and continued with oral antibiotic treatment during 4 days. Cosmetic appearance of the incisions was very satisfactory. The postoperative period was uneventful. No evidence of recurrence has been observed after a follow-up of 18 months, and the patient has hence recovered nicely and remains asymptomatic.

A multilocular, well-circumscribed cyst of 65 cm × 55 cm × 86 mm in size filled with mucoid fluid was sent to pathological examination. No evidence of malignancy was found. Pathologic diagnosis was benign tailgut cyst with negative lateral and vertical margins.


 ¤ Discussion Top


There is an important lack of information on retrorectal cystic hamartoma natural history and biological behaviour: fewer than 200 cases have been published, with women outnumbering men by 3:1. Nearly half the patients are asymptomatic, which explains the frequent delay in clinical diagnosis; the remainder present with symptoms as back pain, rectal fullness, perianal pain and rectal bleeding. Malignant transformation is rare (2%–13%), and the largest series of 53 cases was published by Hjermstad with only one case of malignancy.[4] Preoperative biopsy is controversial, due to risk of spreading dysplastic cells, bleeding and infection, especially with the improvements in the imaging technique. MRI has evolved to be the cornerstone of the evaluation of the tailgut cyst, as it provides excellent anatomic detail, visualises soft-tissue planes and evaluates relationships with adjacent structures and local invasion. Complete en bloc resection with clear surgical margins is the therapy of choice to prevent infection and malignant change. Indeed, incomplete resection can lead to recurrence. Historically, multiple techniques have been reported. Laparoscopic resection, with the reduction of surgical trauma, has recently been reported as a valid alternative to standard Kraske or other abdominal procedures.[5] Potential advantages should include fewer complications, faster recovery of bowel function, shorter hospital stays, less post-operative pain, less blood loss, and improved cosmesis results.

In conclusion, laparoscopic approach of tailgut cysts is feasible, even when tumours contact the elevator ani and coccygeus muscle. Due to the potential risk of recurrence and spreading dysplastic cells if incidentally opening the cyst, other approach should be considered if preoperative studies show malignancy signs.

Declaration of patient consent

The authors certify that they have obtained all appropiate 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 patient 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.
Palanivelu C, Patil KP, Srikanth B, Rajapandian S, Praveenraj P. Laparoscopic assisted management of a pararectal tailgut cyst. Indian J Surg 2015;77:1415-6.  Back to cited text no. 1
    
2.
Dalmonte G, Tartamella F, Mita MT, De Sario G, Roncoroni L, Marchesi F. Laparoscopic resection of tailgut cysts: the size is not the issue. Ann Ital Chir 2016;87. pii: S2239253X16025871.  Back to cited text no. 2
    
3.
Udwadia TE. Single-incision laparoscopic surgery: An overview. J Minim Access Surg 2011;7:1-2.  Back to cited text no. 3
    
4.
Hjermstad BM, Helwig EB. Tailgut cysts. Report of 53 cases. Am J Clin Pathol 1988;89:139-47.  Back to cited text no. 4
    
5.
Marinello FG, Targarona EM, Luppi CR, Boguña I, Molet J, Trias M. Laparoscopic approach to retrorectal tumors: Review of the literature and report of 4 cases. Surg Laparosc Endosc Percutan Tech 2011;21:10-3.  Back to cited text no. 5
    


    Figures

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