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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 4  |  Issue : 2  |  Page : 51-52

Delayed Presentation of H-Type Rectourethral Fistula in Male


1 Additional Professor, Department of Paediatric Surgery, IGIMS, Patna, India
2 Professor, Department of Paediatric Surgery, IGIMS, Patna, India
3 Associate Professor, Department of Paediatric Surgery, IGIMS, Patna, India
4 Senior Resident, Department of Paediatric Surgery, IGIMS, Patna, India

Date of Web Publication10-Dec-2020

Correspondence Address:
Vinit Kumar Thakur
Additional Professor, Dept. of Paediatric Surgery, IGIMS, Patna, Bihar
India
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Source of Support: None, Conflict of Interest: None


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  Abstract 


H-type anorectal malformation characterized by a fistulous communication between urethra and rectum with patency of both epithelial tracts is a rare abnormality in male children. We recently managed a 15-year old male child with on and off passage of urine per rectum. We present the clinical details and the difficulties encounterd in managing this extremely rare malformation in a grown-up male patient.

Keywords: Anorectal malformation; H-type fistula; male


How to cite this article:
Thakur VK, Kumar V, Hasan Z, Yadav R, Rahul Sk, Agarwaf A. Delayed Presentation of H-Type Rectourethral Fistula in Male. J Indira Gandhi Inst Med Sci 2018;4:51-2

How to cite this URL:
Thakur VK, Kumar V, Hasan Z, Yadav R, Rahul Sk, Agarwaf A. Delayed Presentation of H-Type Rectourethral Fistula in Male. J Indira Gandhi Inst Med Sci [serial online] 2018 [cited 2021 Dec 4];4:51-2. Available from: http://www.jigims.co.in/text.asp?2018/4/2/51/302957




  Introduction: Top


H-type rectourethral fistula in males is a rare congenital anorectal malformation. It is included in the rare variants of Krickenbeck classification commonly seen in Asian continent. It is predominant in females. This configuration in males is rare and has only been reported in isolated case reports or a few small series. Because of presence of normal anal opening, diagnosis is usually delayed. If managed properly prognosis is good as far as continence is concerned because of well developed sphincter complex.


  Case Report: Top


A 15-year-old male child presented with intermittent passage of urine per rectally. Three years back, he had noticed that at the end of defecation his stool contained urine; he was not sure about its occurrence before. While being evaluated for urinary tract infection, he was being diagnosed to have rectourethral fistula on cystoscopy. There was no history of trauma or instrumentation. Patient never complained of intractable constipation and had not noticed stool in urine either. No anomalies were detected in general examination except his small sized ears.

On examination, his perineum was well developed and the external genitalia were normal. The anal opening was at normal site and admitted the examining finger. Urine examination was suggestive of proteinuria (3+). Abdominal ultrasonography revealed small atrophic right kidney, left kidney was mildly hydronephrotic. Micturiting cystourethrogram showed bilateral grade two vesicoureteral reflux. Two- dimensional echocardiography was normal. Examination under anaesthesia was done; there was some indentation on anterior rectal wall; colonoscopy and diagnostic cystoscopy revealed a fistulous communication between rectum and urethera distal to verumontanum.

Posterior sagittal anorectoplasty (PSARP) was done without protecting colostomy. Intraoperatively, the fistula opening was seen 3cm proximal to the dentate line on anterior rectal wall [Figure 1], [Figure 2]. The fistula was ligated, the proximal rectum was mobilized adequately to reconstruct the neo-anus. The patient is continent for both urine and stool. He is under regular follow-up.
Figure 1: Fistulous opening in anterior wall of rectum with forceps within

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Figure 2: Divided fistula with stay sutures

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  Discussion: Top


H-type anorectal malformation is an extremely rare variant in the spectrum of congenital anorectal malformation[1]. This type of malformation is found more commonly in Asia and has female preponderance.[1],[2] The H-type configuration in males is distinct from other anorectal malformation, as a normal or ectopic anus is seen in addition to fistula to the urinary tract[3]. Exact embryology of this anomaly is not clear. Most authors believed that ‘ano-/recto-’urethral with normal anus is a variant of ano-rectal malformation and fistula is the result of persistence of cloacal duct[4]. Stephens et al have proposed that a misalignment of the cranial and caudal septal components between the genito-urinary and gastro-intestinal tracts causes these H-type variants[3],[4]. Since the defect results from abnormality in septation, associated anatomic anomalies in VACTERL spectrum are frequently seen[2],[5]. Diagnosis is usually delayed because of normally sited anal opening. The patients usually present with variety of symptoms such as stool in urine, recurrent urinary tract infection, straining while defecation, passage of urine through rectum and recurrent perineal infection[6],[7]. Radiological contrast studies (barium enema and micturiting cystourethrography), magnetic resonance imaging, endoscopy and examination under anaesthesia aid in confirmation of fistula site[1],[2],[8]. Proposed operative approaches have ranged from a simple perineal repair ,anterior perineal repair ,anterior perineal ano-rectoplasty, vestibular pull-through to posterior sagittal ano- rectoplasty[8],[9]. In this patient PSARP was performed in anticipation of adequate exposure of fistula. One can expect a satisfactory outcome in terms of continence as the anal sphincter complex is well developed in this malformation[1],[2],[9]. The complication reported after repair in some series are mainly recurrence and wound dehiscence.


  Conclusion: Top


H-type fistula in males is an extremely rare type of anorectal malformation. Despite its pronounced correlation with VACTERL anomalies, diagnosis is often delayed because of presence of normal looking anus. Radiological and endoscopic examination of recto-urinary tract is a must to ascertain the fistula. There are multiple operative procedures to correct this anomaly and all appear to confer satisfactory functional outcome.



 
  References Top

1.
Tiwary C, Shah H, Bothra J, Kumbhar V. Anal stenosis with H-type rectourethral fistula in a male: A rare anorectal malformation. Saudi Surgical journal 2017;5:40-42.  Back to cited text no. 1
    
2.
Slater BJ, Fallon SC, Brandt ML, Lopez ME.H-type anorectal malformation: case report and review of literature. J Paediatr Surg Case Reports 2014;2:89-92.  Back to cited text no. 2
    
3.
Stephens FD, Donnllan WL. H-type urethroanal fistula. J pediatr. Surg1977;12:95-102.  Back to cited text no. 3
    
4.
Kumar B, Sharma SB, Agarwal LD. Congenital urethral hypoplasia with urethral fistula without imperforate anus:Report of two cases. Afr J. Paediatr Surg 2008;5:57-9.  Back to cited text no. 4
    
5.
Rintala RJ, Mildh L, Lindahl H. H -type anorectal malformation :incidence and clinical characteristics. J Paedtr. Surg 1996;31:559-62.  Back to cited text no. 5
    
6.
Kelleher DC, Henderson PW, Coran A, Spigland NA. The surgical management of H-type rectovestibular fistula:a case report and brief review of the literature. Pediatr Surg Int 2012;28:653-6.  Back to cited text no. 6
    
7.
Van der Putte SC. Normal and abnormal development of the anorectum. J Pediatr. Surg 1986;21:434-40.  Back to cited text no. 7
    
8.
Lawal TA, Chatoogoon K, Bischoff A, Pena A, Levitt MA. Management of H-type rectovestibular and rectovaginal fistulas. J Pediatr. Surg 2011;46:1226-30.  Back to cited text no. 8
    
9.
Kaselas C, Philippopoulos A, Petropoulos A. Evalution of long- term functional outcomes after surgical treatment of anorectal malformations. Int J Colorectal Dis 2011;26:351-56.  Back to cited text no. 9
    


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