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Congenital Anterior Urethrocutaneous Fistula: A Case Report and Review of Literature

Received: 8 May 2022     Accepted: 28 October 2022     Published: 8 December 2022
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Abstract

Introduction: Congenital anterior urethro-cutaneous fistula (CAUF) is an extremely rare abnormality that can register as a malformative entity with hypospadias or cord, or present in an isolated form. Purpose: The purpose of our observation is to present our surgical management of a case of CAUF. Observation: We describe the clinical case of a 10-year-old patient, who presented with CAUF in the ventral mid-penile position, diagnosed at birth. He underwent urethroplasty using Mathieu's technique with simple consequences. The uretral catheter removed at 21 days post-surgery. No complications at 30 days post-operatively. Discussion: CAUF is an extremely rare condition. The etiology is unknown, and several pathogenetic theories have been used to explain its causes.The clinical characteristics are not well defined. An overall assessment of the disease can be carried out in search of other associated abnormalities. CAUF also associated with other congenital anomalies, such as deficient distal urethra, distal hypospadias, ventral penile curvature, anorectal malformations, stenosed bulbar urethra, epispadic urethral duplication, and megalourethra. Abdominal ultrasound and cystoscopy and/or cystography may be performed Therapeutically, surgery remains the only treatment, it includes many methods of surgical repair. Mathieu's technique is the most practiced and gives excellent results. Conclusion: CAUF is a rare anomaly, which must be diagnosed in order to be treated. Surgical treatment offers satisfactory results.

Published in International Journal of Clinical Urology (Volume 6, Issue 2)
DOI 10.11648/j.ijcu.20220602.18
Page(s) 119-121
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2022. Published by Science Publishing Group

Keywords

Fistula, Urethrocutaneous, Congenital, Urethroplasty

References
[1] Siregar S, Albert A. Congenital anterior urethrocutaneous fistulas with chordee: A case report. Int J Surg Case Rep. 2022 Aug 27; 99: 107561.
[2] H. H. Alhazmi. Congenital anterior urethrocutaneous fistula: two case reports and review of literature. Urol. Ann., 6 (3) (2014), pp. 239-241, 10.4103/0974-7796.134285
[3] M. L. Ritchey, A. Sinha, L. Argueso. Congenital fistula of the penile urethra. J. Urol., 151 (4) (1994), pp. 1061-1062, 10.1016/s0022-5347(17)35182-0
[4] Karnak, F. C. Tanyel, A. Hiçsönmez. Congenital urethrocutaneous fistula: a case report and literature review, with a nomenclature proposal. J. Pediatr. Surg., 30 (10) (1995), pp. 1504-1505, 10.1016/0022-3468(95)90422-0
[5] Lin Y, Deng C, Peng Q. Congenital anterior urethrocutaneous fistula: A systematic review. Afr J Paediatr Surg. 2018 Apr-Jun; 15 (2): 63-68.
[6] Caldamone AA, Chen SC, Elder JS, Ritchey ML, Diamond DA, Koyle MA. Congenital anterior urethrocutaneous fistula. J Urol. 1999 Oct; 162 (4): 1430-2.
[7] Hassan A, Kotb M, Shehata S. Congenital anterior urethrocutaneous fistula: A case report and review of literature. Urol Case Rep. 2019 Jul 31; 27.
[8] Bhattacharya. S. Congenital anterior urethrocutaneous fistula. Indian J Plast Surg. 2012 Sep; 45 (3): 566-7.
[9] Mosa H, Garriboli M. Congenital Anterior Urethrocutaneous Fistula with a Persistent Urethral Groove. European J Pediatr Surg Rep. 2021 Jan; 9 (1): e9-e12.
[10] Jindal T, Kamal MR, Mandal SN, Karmakar D. Isolated congenital urethrocutaneous fistula of the anterior urethra. Korean J Urol. 2011 May; 52 (5): 368-70.
[11] User IR, Karakus SC, Akçaer V, Özokutan BH, Ceylan H. Congenital anterior urethrocutaneous fistula: 3 new cases and review of literature. Arch Esp Urol. 2016 Jun; 69 (5): 238-43.
[12] Matsumoto F, Matsuyama S, Matsui F, Yazawa K. Congenital perineal urethrocutaneous fistula without rupture in a neonate. Urol Case Rep. 2021 Mar 17; 37: 10164.
[13] Gupta AK, Kumar M, Singh K, Sokhal AK. Rare association of congenital penile urethrocutaneous fistula with Y-type urethral duplication. BMJ Case Rep. 2017 Feb 24; 2017: bcr2016217331
[14] Batie SF, Coco CT, Jacobs MA. Proximal isolated congenital anterior urethrocutaneous fistula resulting in a urethral "skip" lesion. Urol Case Rep. 2021 Dec 7; 41: 101975.
[15] Bhatnagar A, Upadhyaya VD, Kumar B. Congenital urethrocutaneous fistula: Case report with review of literature. Indian J Plast Surg. 2012 Sep; 45 (3): 563-5.
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    Smith Giscard Olagui, Steevy Ndang Ngou Milama, Christelle Mezene, Dimitri Mbethe, Adrien Mougougou, et al. (2022). Congenital Anterior Urethrocutaneous Fistula: A Case Report and Review of Literature. International Journal of Clinical Urology, 6(2), 119-121. https://doi.org/10.11648/j.ijcu.20220602.18

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    ACS Style

    Smith Giscard Olagui; Steevy Ndang Ngou Milama; Christelle Mezene; Dimitri Mbethe; Adrien Mougougou, et al. Congenital Anterior Urethrocutaneous Fistula: A Case Report and Review of Literature. Int. J. Clin. Urol. 2022, 6(2), 119-121. doi: 10.11648/j.ijcu.20220602.18

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    AMA Style

    Smith Giscard Olagui, Steevy Ndang Ngou Milama, Christelle Mezene, Dimitri Mbethe, Adrien Mougougou, et al. Congenital Anterior Urethrocutaneous Fistula: A Case Report and Review of Literature. Int J Clin Urol. 2022;6(2):119-121. doi: 10.11648/j.ijcu.20220602.18

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  • @article{10.11648/j.ijcu.20220602.18,
      author = {Smith Giscard Olagui and Steevy Ndang Ngou Milama and Christelle Mezene and Dimitri Mbethe and Adrien Mougougou and Gloire Allogho Mbouye and Mariette Nsa Bidzo and Fatou Moutsinga and Jean Placide Owono Mbouengou and Eric Jacob Benizri},
      title = {Congenital Anterior Urethrocutaneous Fistula: A Case Report and Review of Literature},
      journal = {International Journal of Clinical Urology},
      volume = {6},
      number = {2},
      pages = {119-121},
      doi = {10.11648/j.ijcu.20220602.18},
      url = {https://doi.org/10.11648/j.ijcu.20220602.18},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ijcu.20220602.18},
      abstract = {Introduction: Congenital anterior urethro-cutaneous fistula (CAUF) is an extremely rare abnormality that can register as a malformative entity with hypospadias or cord, or present in an isolated form. Purpose: The purpose of our observation is to present our surgical management of a case of CAUF. Observation: We describe the clinical case of a 10-year-old patient, who presented with CAUF in the ventral mid-penile position, diagnosed at birth. He underwent urethroplasty using Mathieu's technique with simple consequences. The uretral catheter removed at 21 days post-surgery. No complications at 30 days post-operatively. Discussion: CAUF is an extremely rare condition. The etiology is unknown, and several pathogenetic theories have been used to explain its causes.The clinical characteristics are not well defined. An overall assessment of the disease can be carried out in search of other associated abnormalities. CAUF also associated with other congenital anomalies, such as deficient distal urethra, distal hypospadias, ventral penile curvature, anorectal malformations, stenosed bulbar urethra, epispadic urethral duplication, and megalourethra. Abdominal ultrasound and cystoscopy and/or cystography may be performed Therapeutically, surgery remains the only treatment, it includes many methods of surgical repair. Mathieu's technique is the most practiced and gives excellent results. Conclusion: CAUF is a rare anomaly, which must be diagnosed in order to be treated. Surgical treatment offers satisfactory results.},
     year = {2022}
    }
    

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  • TY  - JOUR
    T1  - Congenital Anterior Urethrocutaneous Fistula: A Case Report and Review of Literature
    AU  - Smith Giscard Olagui
    AU  - Steevy Ndang Ngou Milama
    AU  - Christelle Mezene
    AU  - Dimitri Mbethe
    AU  - Adrien Mougougou
    AU  - Gloire Allogho Mbouye
    AU  - Mariette Nsa Bidzo
    AU  - Fatou Moutsinga
    AU  - Jean Placide Owono Mbouengou
    AU  - Eric Jacob Benizri
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    DO  - 10.11648/j.ijcu.20220602.18
    T2  - International Journal of Clinical Urology
    JF  - International Journal of Clinical Urology
    JO  - International Journal of Clinical Urology
    SP  - 119
    EP  - 121
    PB  - Science Publishing Group
    SN  - 2640-1355
    UR  - https://doi.org/10.11648/j.ijcu.20220602.18
    AB  - Introduction: Congenital anterior urethro-cutaneous fistula (CAUF) is an extremely rare abnormality that can register as a malformative entity with hypospadias or cord, or present in an isolated form. Purpose: The purpose of our observation is to present our surgical management of a case of CAUF. Observation: We describe the clinical case of a 10-year-old patient, who presented with CAUF in the ventral mid-penile position, diagnosed at birth. He underwent urethroplasty using Mathieu's technique with simple consequences. The uretral catheter removed at 21 days post-surgery. No complications at 30 days post-operatively. Discussion: CAUF is an extremely rare condition. The etiology is unknown, and several pathogenetic theories have been used to explain its causes.The clinical characteristics are not well defined. An overall assessment of the disease can be carried out in search of other associated abnormalities. CAUF also associated with other congenital anomalies, such as deficient distal urethra, distal hypospadias, ventral penile curvature, anorectal malformations, stenosed bulbar urethra, epispadic urethral duplication, and megalourethra. Abdominal ultrasound and cystoscopy and/or cystography may be performed Therapeutically, surgery remains the only treatment, it includes many methods of surgical repair. Mathieu's technique is the most practiced and gives excellent results. Conclusion: CAUF is a rare anomaly, which must be diagnosed in order to be treated. Surgical treatment offers satisfactory results.
    VL  - 6
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Author Information
  • Urology and Visceral Service, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology and Visceral Service, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology Department, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology Department, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology Department, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology Department, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology Department, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology and Visceral Service, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology and Visceral Service, Faculty of Medicine, Urology Department University of Health Sciences, Military Instruction of Omar Bongo Ondimba Hospital, Libreville, Gabon

  • Urology Department of Nice, Nice Polyclinic, Surgery Department, Faculty of Medicine, University of Health Sciences, Nice, France

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