Reliability of Magnetic Resonance Imaging for the Detection of Hypopituitarism in Children with Optic Nerve Hypoplasia

Ramakrishnaiah, Raghu H.; Shelton, Julie; Glasier, Charles M.; Phillips, Paul H.

doi:10.1016/j.ophtha.2013.07.001

Cited by 31 publications

(26 citation statements)

References 13 publications

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“…Previously reported prevalence rates of PHD in patients with ONH vary between 26 and 82% and have been increasing since 1980 [14], with GH deficiency and TSH being the most prevalent PHDs [6, 9, 13, 15, 16]. We report a PHD prevalence of almost 50% among patients with ONH.…”

Section: Discussionmentioning

confidence: 56%

“…A previous study reported lack of significant association between PHD and structural abnormalities of the pituitary gland on MRI; however, this finding was based on unadjusted analysis, and other risk factors for developing ONH (i.e., age at ONH diagnosis, maternal substance abuse) were not included in the analysis [18]. Ramakrishnaiah et al [9] report that an abnormal posterior pituitary gland has 96% sensitivity and 92% specificity in detecting PHD in patients with ONH.…”

Section: Discussionmentioning

confidence: 99%

“…The presence of other midline structural abnormalities of the brain (i.e., absent septum pellucidum or corpus callosum) was not a significant predictor for the development of PHD [9, 12]. One published study reported a higher frequency of PHD in patients with abnormal/absent septum pellucidum; however, the study was cross-sectional, included a small sample of patients with ONH, and did not account for other risk factors for PHD in patients with ONH [8].…”

Section: Discussionmentioning

confidence: 99%

“…Current studies, although cross-sectional in design, point to an increased risk of developing pituitary hormone deficiencies when structural abnormalities of the pituitary gland are present [9, 10], as well as ophthalmologic factors such as bilateral ONH and blindness [2, 11]. Neurodevelopmental issues, including seizures, autism, and behavioral difficulties, may also occur more frequently in children with ONH [2, 12, 13].…”

Section: Introductionmentioning

confidence: 99%

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Pituitary Dysfunction in Pediatric Patients with Optic Nerve Hypoplasia: A Retrospective Cohort Study (1975–2014)

et al. 2017

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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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Section: Discussionmentioning

confidence: 56%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Pituitary Dysfunction in Pediatric Patients with Optic Nerve Hypoplasia: A Retrospective Cohort Study (1975–2014)

et al. 2017

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“…The coexistence of SOD and ONH in a subset of patients suggests that SOD may be one possible causal factor leading to ONH. 33,34 Severe structural changes in the macula and optic nerve of patients with ONH, often associated with midbrain dysgenesis, also may involve the vasculature of the eye. 35 Therefore, an investigation of the relationship between abnormalities in vascular morphology and structural abnormalities in the retina and optic nerve in ONH would be interesting.…”

Section: Foveal Changesmentioning

confidence: 99%

High resolution imaging of the optic nerve and retina in optic nerve hypoplasia

Gottlob

Pilat

Sibley

et al. 2015

Journal of the Neurological Sciences

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Purpose: To investigate the optic nerve and macular morphology in patients with optic nerve hypoplasia (ONH) using spectral-domain optical coherence tomography (SD OCT).Design: Prospective, cross-sectional, observational study. Subjects: A total of 16 participants with ONH (10 female and 6 male; mean age, 17.2 years; 6 bilateral involvement) and 32 gender-, age-, ethnicity-, and refraction-matched healthy controls.Methods: High-resolution SD OCT (Copernicus [Optopol Technology S.A., Zawiercie, Poland], 3 mm resolution) and handheld SD OCT (Bioptigen Inc [Research Triangle Park, NC], 2.6 mm resolution) devices were used to acquire horizontal scans through the center of the optic disc and macula.Main Outcome Measures: Horizontal optic disc/cup and rim diameters, cup depth, peripapillary retinal nerve fiber layer (RNFL), and thickness of individual retinal layers in participants with ONH and in controls.Results: Patients with ONH had significantly smaller discs (P < 0.03 and P < 0.001 compared with unaffected eye and healthy controls, respectively), horizontal cup diameter (P < 0.02 for both), and cup depth (P < 0.02 and P < 0.01, respectively). In the macula, significantly thinner RNFL (nasally), ganglion cell layer (GCL) (nasally and temporally), inner plexiform layer (IPL) (nasally), outer nuclear layer (ONL) (nasally), and inner segment (centrally and temporally) were found in patients with ONH compared with the control group (P < 0.05 for all comparisons). Continuation of significantly thicker GCL, IPL, and outer plexiform layer in the central retinal area (i.e., foveal hypoplasia) was found in more than 80% of patients with ONH. Clinically unaffected fellow eyes of patients with ONH showed mild features of underdevelopment. Visual acuity and presence of septo-optic dysplasia were associated with changes in GCL and IPL. Sensitivity and specificity for the detection of ONH based on disc and retinal optical coherence tomography (OCT) parameters were >80%.Conclusions: Our study provides evidence of retinal changes in ONH. In addition to thinning of retina layers mainly involving the RNFL and GCL, signs reminiscent of foveal hypoplasia were observed in patients with ONH. Optic nerve and foveal parameters measured using OCT showed high sensitivity and specificity for detecting ONH, demonstrating their useful for clinical diagnosis. Ophthalmology 2015;-:1e10 ª

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Neuro-Ophthalmologic Manifestations of Systemic and Intracranial Disease

Brodsky¹

2016

Pediatric Neuro-Ophthalmology

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Reliability of Magnetic Resonance Imaging for the Detection of Hypopituitarism in Children with Optic Nerve Hypoplasia

Cited by 31 publications

References 13 publications

Pituitary Dysfunction in Pediatric Patients with Optic Nerve Hypoplasia: A Retrospective Cohort Study (1975–2014)

Pituitary Dysfunction in Pediatric Patients with Optic Nerve Hypoplasia: A Retrospective Cohort Study (1975–2014)

High resolution imaging of the optic nerve and retina in optic nerve hypoplasia

Neuro-Ophthalmologic Manifestations of Systemic and Intracranial Disease

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