Is aglossia adactylia syndrome and prenatal exposure to misoprostol a causal or casual relationship? A case report
DOI:
https://doi.org/10.18597/rcog.411Keywords:
congenital defect, misoprostol, pregnancyAbstract
Introduction and objective: thea glossiaa dactylia syndrome is an infrequently occurring congenital polymalformative syndrome, having nuclear aetiology and inheritance pattern. A bibliographical search was carried out, orientated towards the association between the prenatal exposure to misoprostol and congenital malformations. A case of this syndrome is presented which was associated with prenatal exposure to misoprostol during the 10th week of gestation; such association has not been established in the literature reviewed.
Presentation of the case: a newborn having phenotypical aglossia adactylia syndrome characteristics, the son of a 17-year-old mother having antecedents of having used a 400 microgram oral dose and a 200 microgram vaginal dose of misoprostol at 10 weeks' gestation with the aim of interrupting her pregnancy.
Discussion: misoprostol is a synthetic prostaglandin E1 analogue; it has been associated with increased risk of Moebius sequence occurring, arthrogryposis, aglossia adactylia syndrome and terminal and cross-sectional extremity defects in mothers who have used this drug during their first gestation trimester.
Author Biographies
Julián Ramírez-Cheyne
Harry Pachajoa
Carolina Isaza
Wilmar Saldarriaga
References
Online Mendelian Inheritance in Man, OMIM (TM). Johns Hopkins University, Baltimore, MD. MIM Number: 103300: Date last edited: 07/07/2003. Disponible en:http://www.ncbi.nlm.nih.gov/omim/
Vargas FR, Schuler-Faccini L, Brunoni D, Kim C, Meloni VF, Sugayama SM, et al. Prenatal exposure to misoprostol and vascular disruption defects: a case– control study. Am J Med Genet 2000;95:302-6.
da Silva Dal Pizzol T, Knop FP, Mengue SS. Prenatal exposure to misoprostol and congenital anomalies: systematic review and meta-analysis. Reproductive Toxicology 2006;22:666-71.
Isaza C, Saldarriaga W, García, R, López MC, Suárez F, Zarante I. Riesgos del misoprostol cuando falla como abortivo en el primer trimestre. Salud UIS 2006;38:66-70.
Firth HV, Boyd PA, Chamberlain PF, MacKenzie IZ, Morriss-Kay GM, Huson SM. Analysis of limb reduction defects in babies exposed to chorionic villus sampling. Lancet 1994;343:1069-71.
Tuncbilek E, Yalcin C, Atasu M. Aglossia-adactylia syndrome (special emphasis on the inheritance pattern). Clin Genet 1977;11:421-3.
Jones K. Smith's recognizable patterns of human malformations. 6th ed. Philadelphia: W.B. Elselvier Saunders Co; 2007. p. 724-43
Norman JE, Thong KG, Baird DT. Uterine contractility and induction of abortion in early pregnancy by misoprostol and mifepristone. Lancet 1991;338:1233-6.
Costa SH. Commercial availability of misoprostol and induced abortion in Brazil. Int J Gynaecol Obstet 1998;63:S131-9.
Schüler L, Ashton PW, Sanseverino MT. Teratogenicity of misoprostol. Lancet 1992;339:437-8.
Schüler L, Pastuszak A, Sanseverino TV, Orioli IM, Brunoni D, Ashton-Prolla P, et al. Pregnancy outcome after exposure to misoprostol in Brazil: a prospective, controlled study. Reprod Toxicol 1999;13:147-51.
Vargas FR, Schuler-Faccini L, Brunoni D, Kim C, Meloni VF, Sugayama SM, et al. Prenatal exposure to misoprostol and vascular disruption defects: a case– control study. Am J Med Genet 2000;95:302-6.
da Silva Dal Pizzol T, Knop FP, Mengue SS. Prenatal exposure to misoprostol and congenital anomalies: systematic review and meta-analysis. Reprod Toxicol 2006;22:666-71.
Fonseca W, Alencar AJ, Mota FS, Coelho HL. Misoprostol and congenital malformations. Lancet 1991;338:56.
González CH, Vargas FR, Pérez AB, Kim CA, Brunoni D, Marques-Dias MJ, et al. Limb deficiency with or without Möbius sequence in seven Brazilian children associated with misoprostol use in first trimester of pregnancy. Am J Med Genet 1993;47:59-64.
González CH, Marques-Dias MJ, Kim CA, Sugayama SM, Da Paz JA, Huson SM, et al. Congenital abnormalities in Brazilian children associated with misoprostol misuse in first trimestre of pregnancy. Lancet 1998;351:1624-7.
Robinow M, Marsh JL, Edgerton MT, Sabio H, Johnson GF. Discordance in monozygotic twins for aglossia-adactylia, and possible clues to the pathogenesis of the syndrome. Birth Defects Orig Art Ser 1978;14:223-30.
Shepard TH. Möbius syndrome after misoprostol: a possible teratogenic mechanism. Lancet 1995;346:780.
Fawcett LB, Buck SJ, Brent RL. Limb reduction defects in the A/J mouses train associated with maternal blood loss. Teratology 1998;58:183-9.
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