Abstract
Experience with the use of cytotoxic drugs in the first trimester of pregnancy is limited. We report on the clinical phenotype and infantile development of a girl born to a 36-year-old mother. Before recognition of pregnancy, the latter had been treated for acute myelocytic leukaemia receiving cytarabine, daunorubicin and doxorubicin at conception and cytarabine and thioguanine at about 35–37 days post conception. At delivery, there were severe brachycephaly, hypoplasia of the anterior cranial base and the midface as well as synostoses of both coronal and metopic sutures. Further findings included bilateral four-finger hands with hypoplastic thumbs and absent radii. This phenotype is reminiscent of the Baller-Gerold syndrome. The child, at present 15 months old, has had to undergo two operations for fronto-orbital advancement because of insufficient growth of the mid-face, nasal airway hypoplasia and increased intracranial pressure. Motor milestones are slightly retarded—neurodevelopment is otherwise normal. These findings are discussed in the context of the few previous reports and are particularly important for future genetic counselling.
Similar content being viewed by others
References
Avilés A, Diaz-Maqueo JC, Talavera A, Guzman R, Garcia EL (1991) Growth and development of children of mothers treated with chemotherapy during pregnancy: current status of 433 children. Am J Hematol 36:243–248
Baller F (1950) Radiusaplasie und Inzucht. Z Menschl Vererb Konstit Lehre 29:782–790
Beeley L (1986) Adverse effects of drugs in the first trimester of pregnancy. Clin Obstet Gynecol 13:177–195
Caligiuri MA, Mayer RJ (1989) Pregnancy and leukemia. Semin Oncol 16:388–396
D'Emilio A, Dragone P, DeNegri G, Montaldi A, Stella M, Battista R (1989) Acute myelogenous leukemia in pregnancy. Haematologica 74:601–604
Doll DC, Ringenberg QS, Yarbro JW (1988) Management of cancer during pregnancy. Arch Intern Med 148:2058–2064
DuBois A, Runge M, Schmid J, Hillemans HG (1990) Disseminated, highly malignant non-Hodgkins's lymphoma and pregnancy: polychemotherapy in the 2nd and 3rd trimester. Geburtshilfe Frauenheilkd 50:405–409
Gerold M (1959) Frakturheilung bei einem seltenen Fall kongenitaler Anomalie der oberen Gliedmaßen. Zentralbl Chir 21:831–834
Huson SM, Rodgers CS, Hall CM, Winter RM (1990) The Baller-Gerold syndrome: phenotypic and cytogenetic overlap with Roberts syndrome. J Med Genet 27:371–375
Jones KL (1988) Smith's recognizable patterns of human malformation, 4th edn. W. B. Saunders, Philadelphia
Lambert J, Wijermans PW, Dekker GA, Ossenkoppele GJ (1991) Chemotherapy in non-Hodgkin's lymphoma during pregnancy. Neth J Med 38:80–85
Lemire RJ (1986) Embryology of the skull. In: Cohen MM jr (ed) Craniosynostosis. Diagnosis, evaluation and management. Raven Press, New York, pp 105–129
Lilleyman JS, Hill AS, Anderton KJ (1977) Consequences of acute myelogenous leukemia in early pregnancy. Cancer 40:1300–1303
Melzer HJ (1956) Congenital anomalies due to attempted abortion with 4-amino pteroyl-glutamic acid. JAMA 161:1253
Milunsky A, Graef JW, Gaynor MF (1968) Methotrexate-induced congenital malformations. J Pediatr 72:790–795
Pelias MZ, Superneau DW, Thurmon TF (1981) Brief clinical report: a sixth report (eigth case) of craniosynostosisradial aplasia (Baller-Gerold) syndrome. Am J Med Genet 10:133–139
Roy V, Gutteridge CN, Nysenbaum A, Newland AC (1989) Combination chemotherapy with conservative obstetric management in the treatment of pregnant patients with acute myeoloblastic leukaemia. Clin Lab Haematol 11:171–178
Schafer AI (1981) Teratogenic effects of antileukemic chemotherapy. Arch Int Med 141:514–515
Selevan SG, Lindbohm ML, Hornung RW, Hemminki K (1985) A study of occupational exposure to antineoplastic drugs and fetal loss in nurses. N Engl J Med 313:1173–1178
Shaw EB, Steinbach HL (1968) Aminopterin-induced fetal malformation. Survival of infant after attempted abortion. Am J Dis Child 115:477–482
Shepard TH (1986) Catalog of teratogenic agents, 5th edn. Johns Hopkins University Press, Baltimore
Wagner VM, Hill JS, Weaver D, Baehner RL, (1980) Congenital abnormalities in baby born to cytarabine treated mother. Lancet II:98–100
Warkany J, Beaudry PH, Hornstein S (1959) Attempted abortion with aminopterin (4-amino-pteroylglutamic acid). Am J Dis Child 97:274–281
Zuazu J, Julia A, Sierra J, Valentin MG, Coma A, Sanz MA, Battle J, Flores A (1991) Pregnancy outcome in haematologic malignancies. Cancer 67: 703–709
Author information
Authors and Affiliations
Rights and permissions
About this article
Cite this article
Artlich, A., Möller, J., Kruse, K. et al. Teratogenic effects in a case of maternal treatment for acute myelocytic leukaemia—neonatal and infantile course. Eur J Pediatr 153, 488–491 (1994). https://doi.org/10.1007/BF01957002
Received:
Accepted:
Issue Date:
DOI: https://doi.org/10.1007/BF01957002