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Pituitary Dysfunction in Pediatric Patients with Optic Nerve Hypoplasia: A Retrospective Cohort Study (1975-2014)
Naseem Alyahyawi1,2, Keira Dheensaw1, Nazrul Islam3,4
1Department of Pediatrics, University of British Columbia, Vancouver, British Columbia, Canada.
Insights
Pituitary hormone dysfunction (PHD) affects nearly half of children with optic nerve hypoplasia (ONH). Key predictors include blindness, pituitary abnormalities, and maternal substance abuse, guiding hormone testing needs.
Area of Science:
- Pediatric Endocrinology
- Neuro-ophthalmology
- Developmental Pediatrics
Background:
- Optic nerve hypoplasia (ONH) is a congenital condition that can lead to various developmental issues.
- Pituitary hormone dysfunction (PHD) is a common complication in children with ONH, but its risk factors are not fully understood.
- Early identification of PHD is crucial for timely intervention and management in affected children.
Purpose of the Study:
- To determine the incidence and timing of pituitary hormone dysfunction (PHD) in children diagnosed with optic nerve hypoplasia (ONH).
- To identify significant predictors and risk factors associated with the development of PHD in this pediatric population.
- To provide data that can inform clinical decision-making regarding hormone testing in ONH patients.
Main Methods:
- Retrospective review of 144 pediatric patient charts with ONH.
- Calculation of PHD incidence rates using a Poisson distribution.
- Multivariable Cox proportional hazards modeling to identify predictors of PHD.
Main Results:
- Nearly half (49.3%) of ONH patients developed PHD over a median follow-up of 2.88 months.
- The incidence of PHD was 11.55 per 100 person-years.
- Significant predictors included blindness, abnormal posterior pituitary, hypoplastic/absent anterior pituitary, prematurity, and maternal substance abuse.
Conclusions:
- Blindness, pituitary gland abnormalities, and maternal substance abuse are key clinical predictors of PHD in children with ONH.
- These identified predictors can aid clinicians in determining the necessity and frequency of hormone testing.
- This research supports personalized monitoring strategies for pediatric ONH patients at high risk for endocrine complications.
Background/Aims:
The risk factors for pituitary hormone dysfunction (PHD) in children with optic nerve hypoplasia (ONH) are not well understood. This study identified the type, timing, and predictors of PHD in children with ONH.
Methods:
ONH patient charts were reviewed retrospectively. The incidence rate of PHD was calculated assuming a Poisson distribution. Predictors of PHD were identified through a multivariable Cox proportional hazards model.
Results:
Among 144 subjects with ONH, 49.3% (n = 71) developed PHD over 614.7 person-years of follow-up. The incidence was 11.55 (95% confidence interval [CI]: 9.02-14.57/100 person-years). The median time to first PHD was 2.88 (interquartile range: 0.02-18.72) months. Eighty-two percent developed their first PHD by their 5th and 90% by their 10th birthday, and 89% within 5 years of ONH diagnosis. Prematurity (adjusted hazard ratio [aHR]: 0.33; 95% CI: 0.1-1.07), blindness (aHR: 1.72; 95% CI: 1.03-2.86), maternal substance abuse (aHR: 1.51; 95% CI: 0.91-2.48), abnormal posterior pituitary (aHR: 3.8; 95% CI: 2.01-7.18), and hypoplastic/absent anterior pituitary (aHR: 2.52; 95% CI: 1.29-4.91) were significant predictors of PHD.
Conclusions:
The clinical predictors of PHD included blindness, pituitary gland abnormalities, and maternal substance abuse. These predictors help clinical decision-making related to the need for and frequency of hormone testing in pediatric patients with ONH.
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