Congenital prepubic sinus-An aborted dorsal urethral duplication or a cloacal remnant?

被引:6
作者
Nazir, Zafar [1 ]
Khan, Muhammad Arif Mateen [1 ]
Qamar, Javaria [1 ]
机构
[1] Aga Khan Univ Hosp, Dept Surg, Sect Pediat Surg, Karachi 74800, Pakistan
关键词
Congenital prepubic sinus; Cloacal membrane; Sinus; Duplication of urethra; Infants and children; Congenital; POSSIBLE VARIANT;
D O I
10.1016/j.jpedsurg.2018.07.014
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Background: Congenital Prepubic Sinus (CPS) is an uncommon urogenital anomaly characterized by a blind tract between the skin over the pubis to anterior of the urinary bladder. Urethra or umbilicus. We report four such cases to emphasize varied clinical presentation and embryological conundrum. Methods: Following Ethical Review Committee (ERC) approval, medical records of pediatric patients (<16 years) presenting with CPS (identified through operating room records and Hospital Information Management System (HIMS) between 1994 and 2018 were reviewed for demographics, clinical presentation, investigations including histopathology, management and outcome. Results: Four cases of CPS. 3 females and 1 male, age range 9 months to 13 years were managed over 25-years. Clinical presentation includes a discharging sinus and recurrent episodes of cellulitis and abscess formation in pubic area and labia majora. Urological investigations were mostly normal. Insertion of lacrimal probe or plastic sheath of intravenous cannula through the sinus opening was useful to determine the course of sinus and aid its excision. Histology of excised sinus highlights the possible embryological origin. Conclusions: CPS is a rare anomaly with varied clinical presentation. It seems CPS is an aborted urethral duplication (Stephen Type 3) or a Cloacal remnant. Complete excision of the tract in the reported cases was curative. Type of study: Case series. (C) 2018 Elsevier Inc. All rights reserved.
引用
收藏
页码:1467 / 1470
页数:4
相关论文
共 22 条
  • [1] Congenital prepubic sinus - A variant of urethral duplication
    Aihole, J. S.
    Babu, M. Narendra
    Javaregowda, D.
    Jadhav, V.
    [J]. AFRICAN JOURNAL OF UROLOGY, 2016, 22 (03) : 196 - 198
  • [2] Congenital prepubic sinus: An epispadiac variant of urethral duplication: Case report and review of literature
    Al-Wattar, KMS
    [J]. JOURNAL OF PEDIATRIC SURGERY, 2003, 38 (04)
  • [3] Balster S, 2003, AKTUEL UROL, V34, P484
  • [4] CONGENITAL PREPUBIC SINUS - POSSIBLE VARIANT OF DORSAL URETHRAL DUPLICATION (STEPHENS TYPE-2)
    CAMPBELL, J
    BEASLEY, S
    MCMULLIN, N
    HUTSON, JM
    [J]. JOURNAL OF UROLOGY, 1987, 137 (03) : 505 - 506
  • [5] SUBPUBIC SINUS - A REMNANT OF CLOACA
    CHOU, TD
    CHU, CC
    DIAU, GY
    CHIANG, JH
    [J]. JOURNAL OF UROLOGY, 1995, 153 (05) : 1671 - 1672
  • [6] CONGENITAL PREPUBIC SINUS
    DAHER, P
    DIAB, N
    MOUSSA, C
    KORKMAZ, G
    [J]. EUROPEAN JOURNAL OF PEDIATRIC SURGERY, 1994, 4 (02) : 119 - 121
  • [7] Congenital prepubic sinus:: Possible variant of dorsal urethral duplication
    Ergün, O
    Sayan, A
    Arikan, A
    [J]. EUROPEAN JOURNAL OF PEDIATRIC SURGERY, 1998, 8 (06) : 380 - 381
  • [8] Pseudo-clitoromegaly associated with congenital prepubic sinus
    Hayase, Masa
    Kojima, Yoshiyuki
    Hayashi, Yutaro
    Maruyama, Tetsuji
    Sasaki, Shoichi
    Kohri, Kenjiro
    [J]. INTERNATIONAL JOURNAL OF UROLOGY, 2006, 13 (07) : 1031 - 1032
  • [9] Congenital prepubic sinus: A variant of dorsal urethral duplication suggested by immunohistochemical analysis
    Huang, CC
    Wu, WH
    Chai, CY
    Wu, TC
    Chuang, JH
    [J]. JOURNAL OF UROLOGY, 2001, 166 (05) : 1876 - 1879
  • [10] CONGENITAL PREPUBIC SINUS
    KOMURA, J
    YANO, H
    KANAZAWA, M
    TANAKA, Y
    TAKAGISHI, T
    SASAGURI, Y
    [J]. PEDIATRIC SURGERY INTERNATIONAL, 1994, 9 (04) : 287 - 289