medwireNews: Factors including age at diagnosis and the specific disease influence the likelihood of children with chronic endocrine conditions being lost to follow-up, show French study findings.
“[T]hese original findings have important clinical implications for patient management”, say the researchers, who stress the need to monitor children for these risk factors, “potentially improving their long-term outcomes”.
The data, published in Hormone Research in Paediatrics, come from 1067 patients seen at a single academic centre between 2007 and 2014. During a median of 5.9 years, 14% of these children were lost to follow-up.
Laura Atger-Lallier (Hôpital Universitaire Robert Debré, Paris, France) and co-researchers regarded patients as lost to follow-up if they had not been seen in clinic for at least 2 years, except for those with congenital hypogonadotropic hypogonadism or disorders of sexual development (DSDs), who were counted as lost to follow-up if they did not attend clinic after the start of puberty, when regular medical assessment is particularly important for children with these conditions.
Encouragingly, the likelihood of children being lost to follow-up was significantly lower for those diagnosed after 2006, the year in which the centre became a reference centre for rare endocrine growth and developmental disorders, with the inclusion of a therapeutic education nurse. Children diagnosed before this time were 4.8-fold more likely to be lost to follow-up than those diagnosed later, after accounting for potential confounders.
Retention also varied with the specific endocrine condition; children with isolated growth hormone deficiency (GHD) comprised 26% of the whole cohort, but 40% of those lost to follow-up. At the other end of the scale, patients with congenital adrenal hyperplasia (CAH), for which treatment disruption is life-threatening, accounted for 6% of the whole cohort but just 1% of those lost to follow-up.
When compared with children with CAH, those who had isolated GHD were a significant 5.24 times more likely to be lost to follow-up. In addition, children with idiopathic 46 XY DSD accounted for 8% of the cohort versus 12% of those lost to follow-up, and this only just failed to retain statistical significance in the multivariate analysis.
For both GHD and 46 XY DSD, the patients were aged around 14 years at their last clinic visit, and the researchers note that stopping treatment at this age would have no immediate clinical effects.
“However, treatment at this crucial period of the patient’s life may have a determinant effect on future health, in terms of sexual maturation, the optimization of adult height and body composition, including the acquisition of bone mass, the prevention of metabolic syndrome, fertility, and quality of life”, they observe.
An increasing number of medical evaluations in the past 3 years was associated with a reduced likelihood of loss to follow-up, and lower socioeconomic health status was associated with an increased risk, despite universal access to medical care in France.
Older age at diagnosis was also associated with an increased risk, which the study authors suggest “may reflect the parents’ perceptions of their child’s health or illness and their fears of the consequences of an unwanted diagnosis”.
By Eleanor McDermid
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Horm Res Paediatr 2020; doi:10.1159/000505517