Eur J Pediatr Surg 2022; 32(06): 504-511
DOI: 10.1055/s-0042-1742300
Original Article

Urological Impact of Epididymo-orchitis in Patients with Anorectal Malformation: An ARM-Net Consortium Study

Anna Morandi
1   Department of Pediatric Surgery, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milano, Italy
Maria Fanjul
2   Department of Pediatric Surgery, Hospital Gregorio Marañón, Madrid, Spain
Barbara Daniela Iacobelli
3   Newborn Surgery Unit, Department of Medical and Surgical Neonatology, Research Institute, Bambino Gesù Children's Hospital, Rome, Italy
Inbal Samuk
4   Department of Pediatric and Adolescent Surgery, Schneider Children's Medical Center, Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel
Dalia Aminoff
5   AIMAR-Italian Patients' and Parents' Organization for Anorectal Malformation, Rome, Italy
Paola Midrio
6   Pediatric Surgery Unit, Cà Foncello Hospital, Treviso, Italy
7   Department of Pediatric Surgery, Radboudumc Amalia Children's Hospital, Nijmegen, the Netherlands
8   Clinic for Paediatric Surgery and Paediatric Urology, Klinikum Bremen-Mitte, Bremen, Germany
9   Pediatric Surgery Unit, The Children Hospital, Azienda Ospedaliera SS Antonio e Biagio e Cesare Arrigo, Alessandria, Italy
Wout Feitz
10   Division of Paediatric Urology, Department of Urology, Radboudumc Amalia Children's Hospital, Nijmegen, the Netherlands
Hendrik J. J. van der Steeg
7   Department of Pediatric Surgery, Radboudumc Amalia Children's Hospital, Nijmegen, the Netherlands
11   Pediatric Urology Unit, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milano, Italy
Cornelius E. J. Sloots
12   Department of Pediatric Surgery, Erasmus MC Sophia Children's Hospital, Rotterdam, the Netherlands
Francesco Fascetti-Leon
13   Pediatric Surgery Unit, Department of Women's and Children's Health, University of Padua, Padua, Italy
Igor Makedonsky
14   Department of Pediatric Surgery, Rudnev Dnipropetrovsk Specialized Clinical Medical Center for Mother and Child Health, Dnipro, Ukraine
Araceli Garcia
15   Pediatric Surgery Unit, University Hospital 12 de Octubre, Madrid, Spain
16   Department of Pediatric Surgery, Skane University Hospital, Lund University, Lund, Sweden
› Author Affiliations
Funding None.


Introduction To investigate the current experience of the ARM-Net Consortium in the management of epididymo-orchitis (EO) in patients with anorectal malformations (ARMs), and to identify specific risk factors and the need for urological care involvement.

Materials and Methods We retrospectively collected data of EO in patients with ARM between 2015 and 2019. Data on urological aspects, ARM type, surgical approach, associated anomalies, diagnosis, and treatment of EO were collected and analyzed.

Results Twenty-nine patients were reported by 12 centers. Twenty-six patients with EO (90%) had ARM with a rectourinary fistula. Median age at first EO was 2 years (range: 15 days–27 years). Twenty patients (69%) experienced multiple EO, and 60% of recurrences were ipsilateral. Associated urological anomalies included vesicoureteral reflux (48%), urethral anomalies (41%), neurogenic bladder (41%), and ectopic vas (10%). A positive urine culture during EO was present in 69%. EO was treated with antibiotics (90%), limiting surgical exploration to 14%. Prevention of recurrences included surgery (bulking agents 15%, vasectomy 15%, and orchiectomy 5%) and antibiotic prophylaxis (20%).

Conclusion Urologists may encounter patients with EO in ARM patients, frequently with positive urine culture. An appropriate urologic work-up for most ARM patients is necessary to identify and treat underlying risk factors. A practical scheme for the work-up is suggested for a close collaboration between pediatric surgeons and urologists.

Publication History

Received: 21 September 2021

Accepted: 13 December 2021

Article published online:
24 January 2022

© 2022. Thieme. All rights reserved.

Georg Thieme Verlag KG
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  • References

  • 1 Kiyan G, Dagli TE, Iskit SH, Tugtepe H. Epididymitis in infants with anorectal malformation. Eur Urol 2003; 43 (05) 576-579
  • 2 Raveenthiran V, Sam CJ. Epididymo-orchitis complicating anorectal malformations: collective review of 41 cases. J Urol 2011; 186 (04) 1467-1472
  • 3 Oğuzkurt P, Tanyel FC, Büyükpamukçu N. Acute scrotum due to edidymo-orchitis associated with vasal anomalies in children with anorectal malformations. J Pediatr Surg 1998; 33 (12) 1834-1836
  • 4 Rubio Cordero JL, Nuñez Nuñez R, Blesa Sanchez E. Ano-rectal malformation and recurring orchioepididymitis in infants. Eur J Pediatr Surg 1994; 4 (01) 46-48
  • 5 Graves RS, Engel WJ. Experimental production of epididymitis with sterile urine; clinical implications. J Urol 1950; 64 (04) 601-613
  • 6 Holt B, Pryor JP, Hendry WF. Male infertility after surgery for imperforate anus. J Pediatr Surg 1995; 30 (12) 1677-1679
  • 7 Zaccara A, Ragozzino S, Iacobelli BD. et al. Epidydimo-orchitis and anorectal malformations: when and in whom?. Pediatr Surg Int 2015; 31 (03) 305-309
  • 8 VanderBrink BA, Sivan B, Levitt MA, Peña A, Sheldon CA, Alam S. Epididymitis in patients with anorectal malformations: a cause for urologic concern. Int Braz J Urol 2014; 40 (05) 676-682
  • 9 Wijers CH, de Blaauw I, Marcelis CL. et al. Research perspectives in the etiology of congenital anorectal malformations using data of the International Consortium on Anorectal Malformations: evidence for risk factors across different populations. Pediatr Surg Int 2010; 26 (11) 1093-1099
  • 10 Khan K, Khan MJ, Khan MY. Anorectal malformations: functional outcome of posterior sagittal anorectoplasty. J Postgrad Med Inst 2011; 22 (04) 304-308
  • 11 Somekh E, Gorenstein A, Serour F. Acute epididymitis in boys: evidence of a post-infectious etiology. J Urol 2004; 171 (01) 391-394 , discussion 394
  • 12 Trovalusci E, Rossato M, Gamba P, Midrio P. Testicular function and sexuality in adult patients with anorectal malformation. J Pediatr Surg 2020; 55 (09) 1839-1845
  • 13 Likitnukul S, McCracken Jr GH, Nelson JD, Votteler TP. Epididymitis in children and adolescents. A 20-year retrospective study. Am J Dis Child 1987; 141 (01) 41-44
  • 14 Siegel A, Snyder H, Duckett JW. Epididymitis in infants and boys: underlying urogenital anomalies and efficacy of imaging modalities. J Urol 1987; 138 (4 Pt 2): 1100-1103
  • 15 Merlini E, Rotundi F, Seymandi PL, Canning DA. Acute epididymitis and urinary tract anomalies in children. Scand J Urol Nephrol 1998; 32 (04) 273-275
  • 16 Umeyama T, Kawamura T, Hasegawa A, Ogawa O. Ectopic ureter presenting with epididymitis in childhood: report of 5 cases. J Urol 1985; 134 (01) 131-133
  • 17 Kajbafzadeh AM, Payabvash S. Endoscopic treatment of vesicovasal and vesicoureteral reflux in infants with persisting mesonephric duct. J Urol 2006; 176 (6 Pt 1): 2657-2662
  • 18 Gupta AD, Loeb S, Stec A, Wang MH. Unusual presentation of a Mullerian remnant in an infant with recurrent epididymo-orchitis. Urology 2011; 78 (06) 1414-1416
  • 19 Hester AG, Kogan SJ. The prostatic utricle: an under-recognized condition resulting in significant morbidity in boys with both hypospadias and normal external genitalia. J Pediatr Urol 2017; 13 (05) 492.e1-492.e5
  • 20 Carachi R, Gobara D. Recurrent epididymo-orchitis in a child secondary to a stone in the seminal vesicle. Br J Urol 1997; 79 (06) 997
  • 21 Gkentzis A, Lee L. The aetiology and current management of prepubertal epididymitis. Ann R Coll Surg Engl 2014; 96 (03) 181-183
  • 22 Hong AR, Acuña MF, Peña A, Chaves L, Rodriguez G. Urologic injuries associated with repair of anorectal malformations in male patients. J Pediatr Surg 2002; 37 (03) 339-344
  • 23 Vordermark II JS. The persisting mesonephric duct syndrome: the description of a new syndrome. J Urol 1983; 130 (05) 958-961
  • 24 Thind P, Gerstenberg TC, Bilde T. Is micturition disorder a pathogenic factor in acute epididymitis? An evaluation of simultaneous bladder pressure and urine flow in men with previous acute epididymitis. J Urol 1990; 143 (02) 323-325
  • 25 Franco I. Functional bladder problems in children: pathophysiology, diagnosis, and treatment. Pediatr Clin North Am 2012; 59 (04) 783-817
  • 26 Santos JD, Lopes RI, Koyle MA. Bladder and bowel dysfunction in children: an update on the diagnosis and treatment of a common, but underdiagnosed pediatric problem. Can Urol Assoc J 2017; 11 (1-2Suppl1): S64-S72
  • 27 Faure A, Haddad M, Hery G, Merrot T, Guys JM. Endoscopic injection of bulking agent around the ejaculatory ducts at the verumontanum for recurrent paediatric epididymitis. J Pediatr Urol 2018; 14 (05) 476-482
  • 28 Kajbafzadeh AM, Shirazi M, Dianat S, Mehdizadeh M. Management of recurrent epididymitis in children: application of neurovascular sparing vas clipping in refractory cases. J Pediatr Urol 2011; 7 (05) 552-558
  • 29 Negri L, Albani E, Di Rocco M, Levi-Setti PE. Aspermia and chronic testicular pain after imperforate anus correction. Cryopreservation of sperm cells extracted from whole orchiectomized testis: case report. Hum Reprod 2002; 17 (11) 2935-2937