Description of the methodology used in an ongoing pediatric care interventional study of children born with cleft lip and palate in South America [NCT00097149]

Abstract

BackgroundThe contribution of birth defects, including cleft lip and palate, to neonatal and infant mortality and morbidity is substantial. As other mortality and morbidity causes including infections, hygiene, prematurity, and nutrition are eradicated in less developed countries, the burden of birth defects will increase proportionally.Methods/DesignWe are using cleft lip and palate as a sentinel birth defect to evaluate its burden on neonatal and infant health and to assess the effectiveness of systematic pediatric care during the first month and first two years of life in decreasing this burden. The neonatal intervention, consisting of weekly pediatric evaluation and referral to appropriate care, is delivered to about 696 infants born with cleft lip and/or palate in 47 hospitals in South America. Neonatal mortality in this group will be compared to that in a retrospective control group of about 464 infants born with cleft lip and/or palate in the same hospitals. The subgroup of infants with isolated clefts of both the lip and palate (about 264) is also randomized into two groups, intervened and non-intervened, and further followed up over 2 years. Intervened cases are evaluated by pediatricians every three months and referred for appropriate care. The intervened and non-intervened cases will be compared over study outcomes to evaluate the intervention effectiveness. Non-intervened cases are matched and compared to healthy controls to assess the burden of cleft lip and palate. Outcomes include child's neurological and physical development and family social and economic conditions.DiscussionLarge-scale clinical trials to improve infant health in developing countries are commonly suggested, making it important to share the methods used in ongoing studies with other investigators implementing similar research. We describe here the content of our ongoing pediatric care study in South America. We hope that this may help researchers targeting this area to plan their studies more effectively and encourage the development of similar research efforts to target other birth defects or infant outcomes such as prematurity and low birth weight.

The Impact of Maternal Smoking during Pregnancy on Early Child Neurodevelopment

“…These children participated in a study of child neurodevelopment in South America, which was part of the Global Network for Women’s and Children’s Health Research study (Wehby et al 2006; McCarthy 2010). Given its focus on normal development, the main study that provides the data for this paper included only children with normal birth outcomes and without major health complications such as requiring oxygen after birth, hospitalization for more than 5 days before hospital discharge after birth, or admission to the intensive neonatal care unit.…”

Application of neurodevelopmental screening to a sample of South American infants: The Bayley Infant Neurodevelopmental Screener (BINS)

“…Since 1967, ECLAMC has provided surveillance of birth defects throughout South America (Castilla & Orioli, 2004) and a unique research structure for numerous child health studies (Lopez-Camelo, Campafia, Santos, & Poletta, 2006; Wehby, Castilla, Goco, et al, 2006; Wehby, Castilla, & Lopez-Camelo, 2010; Wehby, Murray, Castilla, Lopez-Camelo, & Ohsfeldt, 2009). The physicians voluntarily report to ECLAMC the occurrence of birth defects among infants born in affiliated hospitals.…”

Genetics of cleft lip and cleft palate

“…Clefting is also associated with a higher risk of various cancer types, including breast, brain, and colon cancers, in the individual with a cleft as well as their family members [Bille et al, 2005; Dietz et al, 2012; Menezes et al, 2009; Zhu et al, 2002]. The complications of clefting in early life are particularly devastating in developing countries where access to medical care may be limited [Wehby et al, 2006]. Understanding the etiologies of clefting is important not only for our knowledge of developmental biology, but ultimately for improved prevention, treatment, and prognosis for individuals affected by orofacial clefting.…”