Anus,Rectum and Colon

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1 JOURNAL OF THE Anus,Rectum nd Colon CASE REPORT Anl Cnl Dupliction Associted with Prescrl Cyst in n Adult Tkyuki Toyong 1), Hiromitsu Mtsud 1), Ryuichi Miu 1), Yohei Toming 1), Keiji Hirt 1), Msfumi Tkeyoshi 2) nd Mszumi Tsuneyoshi 3) 1) Deprtment of Surgery, Fukuok Snno Hospitl, Fukuok, Jpn 2) Deprtment of Rdiology, Fukuok Snno Hospitl, Fukuok, Jpn 3) Deprtment of Pthology, Fukuok Snno Hospitl, Fukuok, Jpn Astrct: Anl cnl dupliction (ACD) is rre congenitl mlformtion, usully detected erly in life. We report cse of 67-yer-old femle with symptomtic ACD ssocited with prescrl cyst. Physicl exmintion reveled n ccessory opening locted in the midline, posterior to the true nus. Imging exmintions, including fistulogrphy, endonl ultrsonogrphy, nd mgnetic resonnce imging, reveled lindending fistulous trct without connecting with the rectum nd prescrl cyst posterior to the rectum. Complete surgicl excision of the trct with cyst ws performed through posterior sgittl pproch. Histologic exmintion reveled squmous epithelium lining nd smooth muscle undles, therey confirming ACD. The postopertive course ws uneventful, nd the ptient ws doing well; no recurrence ws oserved 4 yers fter surgery. ACD cn present for the first time in infnts, children, nd dults. Imging exmintions re useful for the dignosis nd preopertive ssessment of ACD. Therefore, ACD should e considered in the differentil dignosis, even in dults, when posterior perinel orifice is encountered, prticulrly in femle ptients. Once ACD is suspected, intense imging inspection is recommended to visulize the ACD nd ssocited nomlies, nd surgicl removl is wrrnted to prevent inflmmtory complictions or mlignnt chnges. Keywords: nl cnl dupliction, prescrl cyst, endonl ultrsonogrphy, fistulogrphy, mgnetic resonnce imging J Anus Rectum Colon 2018; 2(1): Introduction Anl cnl dupliction (ACD) is rre congenitl mlformtion, with only 66 cses reported in the English-lnguge literture since ). ACD is defined s second nl orifice, locted posterior to the true nus. Most ACDs end lindly, without connecting with the rectum, nd contin histologic fetures of true nl cnl, including squmous epithelium t the distl end nd smooth muscle cells nd nl glnds in the cnl wll 7). Other diseses, such s fistul-in-no or norectl scess, need to e differentited from ACD using ntomicl or pthologicl exmintion. ACDs re usully detected erly in life 2,3), nd only few cses remin undignosed until dulthood 5,8). Here, we report rre cse of n dult femle with ACD, review the clinicl nd dignostic fetures, nd discuss the current stndrd tretment for this condition. Cse Report A 67-yer-old femle ws referred to our hospitl for the evlution of nl pin. She ws norml t irth nd under- Corresponding uthor: Tkyuki Toyong, Received: July 3, 2017, Accepted: August 25, 2017 Copyright 2018 The Jpn Society of Coloproctology 31

2 J Anus Rectum Colon 2018; 2(1): c d Figure 1. Physicl nd rdiologic imges. () Physicl exmintion shows smll, ccessory opening (rrow hed), locted t midline, pproximtely 1 cm posterior to the true nus (rrow). () Fistulogrphy shows 2-cm cnl (rrow hed) without connecting with the rectum (rrow). (c) Endonl ultrsonogrphy shows rightly hyperechoic lind-ending fistulous trct (rrow hed) nd hypoechoic cystic mss (rrow) in the superior retrorectl re. (d) Mgnetic resonnce imging shows multiloculted prescrl cyst (rrow hed) posterior to the rectum. went surgery for perinl scess t the ge of 20 yers. She egn to experience intermittent nl pin 3 yers prior to the presenttion; however, she did not notice two nl holes. Physicl exmintion reveled smll ccessory opening, ppering s secondry nus. The orifice ws locted t midline, pproximtely 1 cm posterior to the true nus (Figure 1). Fistulogrphy reveled 2-cm-long nrrow cnl without connection with the rectum (Figure 1). Endonl ultrsonogrphy (EAUS) with hydrogen peroxide injection reveled rightly hyperechoic, lind-ending fistulous trct nd hypoechoic cystic mss in the superior retrorectl re (Figure 1c). Mgnetic resonnce imging (MRI) reveled multiloculted prescrl cyst posterior to the rectum (Figure 1d). The clinicl nd rdiologic fetures were consistent with n ACD ssocited with prescrl cyst. The ptient underwent surgicl excision under spinl nesthesi in the prone position through posterior sgittl p32 proch. The lesion circumscriing the second nl orifice ws removed with incision, nd the fistulous trct ws seprted crefully from the externl nl sphincter nd prt of the strited muscle complex (Figure 2). The fistulous trct ws tuulr, 2-cm long, nd connected to the superior prescrl cyst (Figure 2). The nl opening nd cnl were removed in continuity with the cyst, which ws djcent to ut seprte from the rectum. The resected specimen showed distl, 2.0-cm-long duct superiorly connected vi firm restiform tissue with cm cyst filled with seceous mteril (Figure 2c). Histologic exmintion reveled tht the duct ws predominntly lined y squmous epithelium nd tht the cyst ws lined y squmous, columnr, nd trnsitionl epitheli (Figure 3). Smooth muscle undles were present throughout the duct wll (Figure 3). Pthology confirmed finl dignosis of ACD. The postopertive course ws without compliction, nd the ptient ws doing well; no sign of recurrence ws

3 Anl Cnl Dupliction with Adult Prescrl Cyst c Figure 2. Intropertive imges. () The lesion circumscriing the second nl orifice (rrow hed) ws removed with incision, nd the fistulous trct ws crefully seprted from the externl nl sphincter. () The fistulous trct ws found to e tuulr, 2-cm long (rrow hed), nd connected with the superior prescrl cyst (rrow). (c) The resected specimen shows the distl portion is 2.0-cm-long duct (rrow hed) nd is superiorly connected, vi the firm restiform tissue, with cm cyst (rrow), filled with seceous mteril. Figure 3. Histologicl findings. () The duct is predominntly lined y squmous epithelium (rrow hed) nd the cyst is lined y squmous, columnr, nd trnsitionl epitheli (rrow). (hemtoxylin-eosin stin, 40 ). () Smooth muscle undles re present in the wll of the duct (rrow hed). (hemtoxylin-eosin stin, 40 ). 33

4 J Anus Rectum Colon 2018; 2(1): oserved 4 yers fter the surgery. 34 Discussion Treting this cse required two importnt clinicl determintions. First, lthough ACD is very rre congenitl mlformtion tht is usully detected erly in infnts or children, it cn present for the first time in dults. Second, imging exmintions, including fistulogrphy, EAUS, nd MRI, re useful for the dignosis nd preopertive ssessment of ACD in dults. In the literture pertining to ACD, pproximtely hlf of ptients (47%) re reported to e symptomtic. They re discovered incidentlly or during the creful inspection of the perineum during the neontl period 4). In two-thirds of ptients, dignosis is mde during the first yer of life. In the English-lnguge medicl literture, there re no more thn nine documented cses of ptients older thn 15 yers of ge, who were dignosed with ACD 2,5,8,9). Mild symptoms, such s dirrhe, constiption, nl pin, or mucous dischrge, occur in 33% of ptients, nd more severe symptoms, such s recurrent fistuls, skin scess, or sepsis, occur in 20% of ptients. The severity of symptoms ppers to increse with increse in ge t presenttion. Most ACDs re tuulr (87%) lthough they occsionlly contin cystic component (13%), s oserved in our ptient. Approximtely 36% cses re ssocited with other nomlies, such s tertoms, meningocele, cleft plte, nd urogenitl mlformtions. Femles represent 89% of ll cses; however, the reson for this predominnce is uncler. The dignosis of ACD requires strong clinicl suspicion followed y rdiologicl nd histologicl confirmtion. ACD should e differentited from fistul-in-no or norectl scess. Imging modlities, such s fistulogrphy, ultrsound, nd MRI, re used to confirm the dignosis 2). Fistulogrphy llows the evlution of the ACD configurtion (cystic or tuulr), length, nd connection with the nl cnl 3). Pelvic nd dominl ultrsound exmintions re useful for evluting the presence of ssocited nomlies in neontes, nd MRI should e routinely used in older children nd young dults ecuse ultrsounds do not dequtely visulize the intrspinl nd prescrl spce in ptients elonging to these ge groups. EAUS is relile technique for the ssessment of fistul-in-no nd is useful for distinguishing this condition from other inflmmtory or tumorous conditions tht ffect the region round the nl cnl 10,11). In this study, EAUS with hydrogen peroxide injection reveled rightly hyperechoic lind-ending fistulous trct without connection with the nl cnl. Furthermore, EAUS reveled hypoechoic cystic mss in the superior retrorectl re. Therefore, EAUS ws useful for distinguishing ACD from fistulin-no nd for visulizing ssocited prescrl nomlies. The min complictions documented in the literture include infection risk rising from the ccessry nl glnds nd the possiility for mlignnt progression 6,12). Therefore, complete surgicl excision through perinel or posterior sgittl pproch is considered the gold stndrd tretment for ACD 3). Cre must e tken to not excise the norml gut during complete ACD excision nd to not injure the nl sphincter, which my potentilly disrupt norml owel function. After complete excision, the prognosis is excellent nd the moridity is miniml. In conclusion, lthough ACD is very rre condition, it cn present for the first time not only in infnts nd children ut lso in dults. Imging exmintions, including fistulogrphy, EAUS, nd MRI, re useful for the dignosis nd preopertive ssessment of ACD. When we encounter posterior perinel orifice, prticulrly in femle ptients, ACD should e considered in the differentil dignosis, even in dults. Once ACD is suspected, intense imging inspection should e performed to visulize the ACD nd ssocited nomlies, nd surgicl removl is wrrnted to prevent inflmmtory complictions or mlignnt chnges. Conflicts of Interest There re no conflicts of interest. Source of Funding No funding ws provided for this cse series. References 1. Aronson I. Anterior scrl meningocele, nl cnl dupliction cyst nd covered nus occurring in one fmily. J Peditr Surg Oct; 5(5): Krtz JR, Deshpnde V, Ryn DP, et l. Anl cnl dupliction ssocited with prescrl cyst. J Peditr Surg Sep; 43(9): Kog H, Okzki T, Kto Y, et l. Anl cnl dupliction: experience t single institution nd literture review. Peditr Surg Int Oct; 26(10): Vn Biervliet S, Mris E, Vnde Velde S, et l. Anl cnl dupliction in n 11-yer-old-child. Cse Rep Gstrointest Med Sep; 2013: Mirzei R, Mhjui B, Alvndipoor M, et l. Lte presenttion of nl cnl dupliction in dults: series of four rre cses. Ann Coloproctol Fe; 31(1): Hond S, Minto M, Miygi H, et l. Anl cnl dupliction presenting with scess formtion. Peditr Int Apr; 59(4): Ochii K, Umed T, Murhshi O, et l. Anl-cnl dupliction in 6-yer-old child. Peditr Surg Int Mr; 18(2-3): Sinny S, Curtis K, Wlsh M, et l. Lte presenttion of nl cnl dupliction in n dolescent femle-- rre dignosis. Int J Colorectl Dis Aug; 28(8): Tgrt RE. Congenitl nl dupliction: cuse of pr-nl sinus. Br J Surg Jul; 64(7): Engin G. Endosonogrphic imging of norectl diseses. J Ultrsound Med Jn; 25(1): Toyong T, Tnk Y, Song JF, et l. Comprison of ccurcy of

5 physicl exmintion nd endonl ultrsonogrphy for preopertive ssessment in ptients with cute nd chronic nl fistul. Tech Coloproctol Sep; 12(3): Dukes CE, Glvin C. Colloid Crcinom rising within Fistule in the Ano-Rectl Region. Ann R Coll Surg Engl Apr; 18(4): Anl Cnl Dupliction with Adult Prescrl Cyst Journl of the Anus, Rectum nd Colon is n Open Access rticle distriuted under the Cretive Commons Attriution-NonCommercil-NoDerivtives 4.0 Interntionl License. To view the detils of this license, plese visit ( 35

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