Literature DB >> 11933692

Extreme caudal agenesis. Possible drug-related etiology?

Nathan Rojansky1, Sozos J Fasouliotis, Ilana Ariel, Michael Nadjari.   

Abstract

BACKGROUND: Caudal regression syndrome (CRS) is a rare anomaly of the lower body pole that represents a continuum of congenital malformations ranging from isolated sacral agenesis to absence of the lumbosacral spine and major visceral anomalies. While the exact etiology of this syndrome is unclear, maternal diabetes, genetic factors, teratogens and vascular anomalies altering blood flow have been hypothesized to play a role in its pathogenesis. CASE: A fetus had extreme hypotrophy of the caudal body pole, aplasia of the lower spine and complete renal agenesis diagnosed in the second trimester by ultrasound. Maternal history revealed the use of minoxidil solution for preventing hair loss for four years prior to and during gestation. Also, the mother had taken trimethoprim-sulfamethoxazole during the first trimester for treatment of upper respiratory disease. No maternal diabetes or history of familial genetic diseases was evident.
CONCLUSION: In an extreme form of CRS consisting of complete aplasia of the lower body pole and viscera and additional malformations, a possible drug-related etiology was suggested but should be confirmed by more studies.

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Year:  2002        PMID: 11933692

Source DB:  PubMed          Journal:  J Reprod Med        ISSN: 0024-7758            Impact factor:   0.142


  5 in total

1.  Lumbopelvic stabilization with external fixator in a patient with lumbosacral agenesis.

Authors:  Jacques Griffet; Julien Leroux; Toni El Hayek
Journal:  Eur Spine J       Date:  2010-05-26       Impact factor: 3.134

Review 2.  Caudal regression syndrome--case report and review of literature.

Authors:  Santosh Kumar Singh; Rupa Dalmia Singh; Akhilesh Sharma
Journal:  Pediatr Surg Int       Date:  2005-06-24       Impact factor: 1.827

3.  Medulloblastoma in association with sacral agenesis; a case report.

Authors:  Zahra Ebrahim Soltani; Zohreh Habibi; Farideh Nejat
Journal:  Childs Nerv Syst       Date:  2018-02-05       Impact factor: 1.475

4.  Sacral agenesis: a pilot whole exome sequencing and copy number study.

Authors:  Robert M Porsch; Elisa Merello; Patrizia De Marco; Guo Cheng; Laura Rodriguez; Manting So; Pak C Sham; Paul K Tam; Valeria Capra; Stacey S Cherny; Maria-Mercè Garcia-Barcelo; Desmond D Campbell
Journal:  BMC Med Genet       Date:  2016-12-22       Impact factor: 2.103

5.  The hydroxypropyl-β-cyclodextrin-minoxidil inclusion complex improves the cardiovascular and proliferative adverse effects of minoxidil in male rats: Implications in the treatment of alopecia.

Authors:  Fatima Maqoud; Nicola Zizzo; Antonietta Mele; Nunzio Denora; Giuseppe Passantino; Rosa Scala; Annalisa Cutrignelli; Antonella Tinelli; Valentino Laquintana; Flavia la Forgia; Sergio Fontana; Massimo Franco; Angela Assunta Lopedota; Domenico Tricarico
Journal:  Pharmacol Res Perspect       Date:  2020-06
  5 in total

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