Lemus-Rodríguez Y, Morán-Martínez RG, Santana-Ramírez A

Language: Spanish
References: 7
Page: 28-32
PDF size: 534.94 Kb.

ABSTRACT

Infant 1 year and 11 months old with severe macrocephaly secondary to hydrocephalus and dysmorphic syndrome by computed tomography (CT) revealed holoprosencefaly, hydrocephalus, agenesis of corpus callosum and absence of shell. The mother had history of taking methotrexate during the first trimester of pregnancy, taking this in consideration, so aminopterin/methotrexate syndrome is diagnosed. A ventricular peritoneal shunt (VP shunt) was installed and subsequently several surgical craneoplasties were performed, achieving a reconstruction of the frontal skull and a decrease of temporal bumps was made using bone grafting in occipitoparietal region, which resulted in benefical changes in the patient's neurological development. Aminopterin/methotrexate syndrome is found in children from women who take acid folic antagonist during pregnancy. It is characterized by defects in neural tube closure. The mainly afected weeks are between six and eigth post conception. Methotrexate is classificated as an X drug by FDA and it must be not prescribed during pregnancy. Because the low incidence of this syndrome, it is important to report the case and the treatment response, as well the interdisciplinary work that it requires.

REFERENCES

1. Bawle EV, Conard JV, Weiss L. Methotrexate syndrome. Teratology 1998; 57:51–55.

2. Lloyd ME, Carp M, McElhatton P, Hall GM, Hughes RA. The effects of methotrexate on pregnancy, fertility and lactation. J Med 1999; 92:551-563.

3. 3.Thomas H, Shepard, Ronald J., Lemire. Catalog of theratogenic agents, 11° ed. John Hopkins University Press, 2004.

4. Lorenz LB, Knudtson EJ. Anomalies in a fetus exposed to methotrexate in the first trimester. Am J Pharmac Toxicol 2007; 2(4): 146-147.

5. Seidahmed MZ, Shaheed MM, Abdulbasit OB, Al Dohami H, Babiker M, Abdullah MA, Abomelha AM. A case of methotrexate embryopathy with holoprosencephaly, expanding the phenotype. Birth Defects Res A Clin Mol Teratol 2006;76(2):138-42.

6. Aftimos S. Fetal methotrexate/aminopterin syndrome in an adult: a likely case with ectodermal abnormalities. Clinical Dysmorphology 2009; 18: 53–55 .

7. Garcia-Minaur1 S, Botella MP. Further case of aminopterin syndrome sine aminopterin in a spanish child. American Journal of Medical Genetics 2000; 95:320–324.