Biological Clock Dysfunction in Optic Nerve Hypoplasia
キーワード
概要
説明
Objectives and Hypotheses. Our objectives are to define the scope and problems related to biological clock disorders in children with ONH and to develop effective treatments for this condition. Based on our observations, we hypothesize: (1) Daily rest-activity patterns and sleep will be abnormal in up to 50% of children with ONH. (2) It is possible to identify risk factors for abnormal circadian system function and sleep problems in ONH. (3) Nocturnal melatonin administration will improve abnormal sleep and activity patterns in children with ONH.
Design: These studies will involve collaborative efforts between Yale University and Dr. Mark Borchert of Childrens Hospital Los Angeles, who follows the largest population of children with ONH in the world. We will study children ages 2-10 years with documented ONH using standard criteria. Based on these criteria, we have more than 100 eligible patients.
To test our hypotheses, we will: (1) examine expressed rhythmicity in children with ONH. These studies will use actigraphy, sleep questionnaires, and assessment of melatonin secretory profiles. (2) We will correlate hypothalamic anatomical abnormalities and the degree endocrine dysfunction with sleep and expressed rhythmicity. (3) We will test if short-term administration of melatonin improves sleep-wake patterns in children with abnormally-expressed rhythmicity.
Potential Impact: Our preliminary data raise the possibility that children with ONH will have circadian system dysfunction resulting in abnormal rhythmicity and sleep. However, to date there have been no formal attempts to identify children with ONH who are at risk for such problems, nor have there been efforts aimed at developing potential treatments. The proposed prospective clinical study will represent an important attempt to identify a group of children with circadian system dysfunction.
At the completion of this study, we anticipate having determined risk factors for circadian system dysfunction in children with ONH. Insights gained from these studies should lead to the development of new approaches for treating circadian clock lesions in ONH, with the hope of improving the well-being of circadian system function in the boys and girls with this condition.
日付
| 最終確認済み: | 08/31/2020 |
| 最初に提出された: | 10/12/2008 |
| 提出された推定登録数: | 01/18/2009 |
| 最初の投稿: | 01/20/2009 |
| 最終更新が送信されました: | 09/22/2020 |
| 最終更新日: | 09/24/2020 |
| 実際の研究開始日: | 09/30/2008 |
| 一次完了予定日: | 08/31/2011 |
| 研究完了予定日: | 08/31/2011 |
状態または病気
介入/治療
Dietary Supplement: 1
Dietary Supplement: 2
段階
アームグループ
| 腕 | 介入/治療 |
|---|---|
| Active Comparator: 1 Individuals with abnormal rhythmicity will be treated with melatonin to assess if sleep patterns are improved. | Dietary Supplement: 1 Oral administration before bed. We will test two doses (0.5 or 3.0 mg/m2. 6 week duration.) |
| Placebo Comparator: 2 | Dietary Supplement: 2 Oral administration before bed. 6 week duration. |
適格基準
| 研究の対象となる年齢 | 2 Years に 2 Years |
| 研究に適格な性別 | All |
| 健康なボランティアを受け入れる | はい |
| 基準 | Inclusion Criteria: - Confirmed diagnosis of ONH (Diagnosis will be confirmed by ocular fundus photography.Disc-macula distance 0.35 or below. In eyes without ONH, the DD/DM ratio is greater than 0.3581.) - Ages 2-10 years - Ability to ambulate independently - Under care of endocrinologist if on pituitary hormone replacement therapy. Exclusion Criteria: - Non-ambulatory - Active malignancy - Cardio-respiratory disorder |
結果
主な結果の測定
1. Assessment of rest-activity patterns [two years]
