Format

Send to:

Choose Destination
See comment in PubMed Commons below
J Pediatr Surg. 2013 Jul;48(7):1573-7. doi: 10.1016/j.jpedsurg.2012.08.038.

Wnt signalling in testicular descent: a candidate mechanism for cryptorchidism in Robinow syndrome.

Author information

  • 1Douglas Stephens Surgical Research Laboratory, Murdoch Children's Research Institute, Melbourne, Australia.

Abstract

BACKGROUND/AIMS:

Robinow syndrome is caused by mutations in Wnt-5a or its receptor Ror2 and can lead to cryptorchidism, though the mechanisms are unclear. Wnt-5a knock-out mice fail to undergo gubernacular swelling, similar to insulin-like hormone 3 (INSl-3) knock-out mice. We aimed to characterise Wnt-5a and Ror2 expression in rat gubernacula to better understand how Wnt-5a signalling affects testicular descent.

METHODS:

Sprague-Dawley rats (n = 27) were collected with ethics approval (A644) at embryonic days (E) 15, 17, 19 and postnatal day (D) 2. Control and antiandrogen-treated groups were processed for immunohistochemistry for Wnt-5a, Ror2 and β-catenin. Sagittal sections were examined using confocal microscopy.

RESULTS:

Wnt-5a and Ror2 were strongly expressed in the gubernacular bulb at E17 controls, their levels declining at E19 and almost absent by D2. Wnt-5a significantly co-localised with the important transcription factor β-catenin at E17. There was no obvious difference in staining with androgen blockade.

CONCLUSION:

Wnt-5a, through Ror2 and β-catenin may play a vital role in regulating the gubernacular swelling reaction downstream of INSL-3. Human mutations in Wnt-5a or Ror2 could prevent early gubernacular growth, as suggested by undescended testes in 70% of patients with Robinow Syndrome.

Copyright © 2013 Elsevier Inc. All rights reserved.

KEYWORDS:

Cryptorchidism; Gubernaculum; Robinow syndrome; Testicular descent; Wnt-5a

PMID:
23895974
[PubMed - indexed for MEDLINE]
PubMed Commons home

PubMed Commons

0 comments
How to join PubMed Commons

    Supplemental Content

    Full text links

    Icon for Elsevier Science
    Loading ...
    Write to the Help Desk