Abstract
A wide body of evidence shows that patients with clefts exhibit decreased growth in their early years. Less evidence regarding their growth trajectory, specifically their body mass indices (BMIs), in later years is available. This study analyzes BMIs of patients with isolated clefts and/or palate over time using age-adjusted BMI z -scores based on the CDC 2000 references for height and weight for age. At least two height and weight measurements were obtained during 2–10 years and at least two height and weight measurements were obtained during 10–20 years. Mean BMI z -score for all patients was 0.117. Males had a mean z -score of 0.087 and females a mean of 0.160 ( p = 0.407). Patients under 10 years of age had a mean z -score of 0.208, while patients older than 10 years had a mean z -score of −0.028 ( p = 0.223). While significance was not achieved in the statistical analysis, the results suggest that overall, the cleft population maintains a mean BMI that is similar to that of the general population during childhood and adolescence. A larger analysis is warranted to investigate this phenomenon further and to investigate specifically the rate of obesity within this group.
The medical implications of cleft lip and/or palate do not end with successful surgical repair, as evidenced by many reports in the literature. One such manifestation is the poor weight gain often experienced by infants with clefts. Many articles describe the growth trends among patients with clefts in their early years; the common conclusion is that such patients experience decreased growth and an elevated prevalence of failure to thrive. Despite these early setbacks, by the toddler years, overall patients with clefts are able to draw near the level of their unaffected peers with regard to anthropomorphic measurements including height and weight for age. Most of the literature examining growth patterns in patients with clefts is limited to the early childhood years. Recent increases in the prevalence of obesity among children and adolescents in the USA warrant investigation into modern growth profiles in the cleft population.
While data on the growth of cleft patients into their later childhood and adolescence is relatively less available, there has been much data to suggest that in their later years, patients born with clefts experience negative health outcomes. Multiple studies have found a correlation between being born with a cleft and certain types of cancer during later stages of life. One long-term follow-up study found that patients with congenital clefts experience an increased risk of death from all major causes, and that this risk is present throughout all phases of the lifespan despite undergoing surgical repair for their abnormality. Many theories attempt to explain these observations, including that the idiopathic genetic cause of the cleft deformity may have wide-ranging manifestations in other aspects of the patient’s health. Some have concluded that cleft patients should be subject to preventive-based healthcare throughout their lives.
The present study represents an investigation of the growth of cleft children ranging from infancy to early adulthood. Specifically, the body mass index (BMI) was used as an anthropomorphic measurement to compare patients with clefts to normative data in order to determine whether individuals born with isolated cleft deformities go on to have BMIs that differ from their unaffected peers. Any such differences may be clinically significant within the context of understanding the long-term clinical trajectories of patients within this population, as well as gaining insight into the particular healthcare needs, be they preventive or otherwise, of this group.
Materials and methods
Following institutional review board approval, patients were identified through the roster of invitees to the craniofacial team’s annual summer camp. Comprising this roster are the patients aged 0–21 years who have seen physicians or other providers on the craniofacial team for either surgical or medical care. To be eligible for inclusion in the study, patients must have presented with either isolated cleft lip and/or palate. Patients with both complete and incomplete clefts and unilateral and bilateral clefts were included in the study population. Patients were excluded if their cleft palate was part of a syndrome or sequence, such as the Pierre-Robin Sequence or DiGeorge’s Syndrome. Patients with significant co-existing birth deformities that were not a part of recognized sequences/syndromes, or patients with karyotype abnormalities were excluded from the analysis.
A database for prospective analysis was constructed with data consisting of each patient’s date of birth, gender, specific deformity (i.e. unilateral incomplete cleft lip and palate), and height and weight measurements at all available time points during which the patient was between 2 and 20 years of age. Data were obtained by retrospective analysis using the Clinical Information System (CIS) electronic medical records of the University’s primary teaching and patient-care facility. All available surgical, office, discharge and correspondence notes from healthcare providers present within each patient’s CIS record were reviewed for information regarding their deformity and for height and weight measurements.
The primary anthropomorphic measurement that was calculated from the data was the BMI. BMI is defined as the weight (in kg) divided by height (in m) squared. The clinically oriented perspective from which this study was formulated is that of gaining insight into underlying growth patterns associated with having an orofacial cleft. Thus, the BMI, a measure of relative weight, was the appropriate metric due to its use as the primary screening tool for risk assessment of children and adolescents in the primary care setting. For determination of BMIs, the Center for Disease Control and Prevention’s Epi Info™ Software was employed. For each measurement available, the patient’s gender, height, weight and age in months were input into the software for calculation of the BMI.
In order to compare BMIs across the varying ages at which all of the measurements were taken, a z -score for each measurement was calculated using Epi Info™ based upon the CDC’s clinical growth charts published in the year 2000. The z -score is the comparison (in standard deviations) of the study subjects (cleft patients) from the mean of the population (CDC data) (in contrast to Student’s t test, which is a comparison of the study subjects to a population sample mean). Data were analyzed by determination of central tendencies (mean BMI z -score) and via Student’s t test for the entire patient population and for stratifications on the basis of age and gender. For patients with measurements present in both age categories, the most recent measurement in each was employed in age-stratified analysis.
Results
The medical records of 318 craniofacial patients were reviewed. Patients were included if they had height and weight assessments recorded for at least two time points under 10 years of age and two points older than 10 years of age. Following exclusion of patients with clefting associated with syndromes/sequences, karyotype abnormalities or significant other birth abnormalities, and the exclusion of patients without necessary height and weight measurements for BMI calculation between the ages of 2 and 20 years, 59 patients were identified for inclusion in the analysis.
39 males and 20 females were included. Male cleft distribution included 7 bCL/CP, 16 uCL/CP, 7 CL, 9 CP. Female cleft distribution included 4 bCL/CP, 8 uCL/CP, 5 CL, 3 CP ( Fig. 1 ). Owing to the small sample size, it was impossible to calculate a reliable z -score based on type of cleft. The mean BMI z -score for all patients was 0.117. Patients were stratified by gender, with males having a mean z -score of 0.087 and females a mean z -score of 0.160 ( p = 0.407). Patients were stratified into two groups according to age, with age younger than 10 years having a mean z -score of 0.208, and age greater than 10 years having a mean z -score of −0.028. While statistical significance was approached, it was not reached ( p = 0.223).
