David J Cullingford, Jacqueline A Curran, Mary B Abraham, Aris Siafarikas, A Marie Blackmore, Jenny Downs, Catherine S Y Choong
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This was compared with predictive REE (pREE) based on the Schofield equation to determine mREE/pREE quotient. A BMI z-score >1 was considered elevated. Parents/carers completed a questionnaire about pituitary function, the Hyperphagia Questionnaire and the Sleep Disturbances Scale for Children (SDSC).</p><p><strong>Results: </strong>Twenty-six participants underwent testing (9 female, mean age 12.1 years) with 11 having elevated BMI and 15 with pituitary hormone deficiencies. Mean mREE was 1309 kcal/day (838-1732), mREE/pREE quotient was 88.8% ± 10.1. mREE/pREE quotient was similar in those with elevated BMI compared with normal BMI (83.3% ± 12.5 vs 92.1% ± 7.2, <i>P</i> = .068). Those with midline defects had a higher mREE/pREE quotient (91.8% ± 8.1 vs 80.4% ± 11.3, <i>P</i> = .026). Hyperphagia and SDSC scores were similar between BMI groups. Hyperphagia domain scores were higher in children with multiple hypopituitarism, pituitary structural defects, and normal septum pellucidum (<i>P</i> = .044, .042, and .033, respectively).</p><p><strong>Conclusion: </strong>Children with SOD had lower mREE than predicted and hyperphagia scores were higher in those with biochemical or structural pituitary changes, suggesting that hypothalamic dysfunction could drive BMI elevation in SOD. Indirect calorimetry may be used to guide the management of overweight and obesity in SOD.</p>","PeriodicalId":17334,"journal":{"name":"Journal of the Endocrine Society","volume":"9 4","pages":"bvaf031"},"PeriodicalIF":3.0000,"publicationDate":"2025-02-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11914970/pdf/","citationCount":"0","resultStr":"{\"title\":\"Resting Energy Expenditure and Metabolic Features in Children With Septo-Optic Dysplasia.\",\"authors\":\"David J Cullingford, Jacqueline A Curran, Mary B Abraham, Aris Siafarikas, A Marie Blackmore, Jenny Downs, Catherine S Y Choong\",\"doi\":\"10.1210/jendso/bvaf031\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Context: </strong>Septo-optic dysplasia (SOD) is a major cause of congenital hypopituitarism and is known to be associated with overweight and obesity in up to 44% of children. Given the role of the hypothalamus in hormonal regulation, we sought to assess the association of resting energy expenditure (REE), appetite and physical activity with SOD.</p><p><strong>Objective: </strong>To characterize REE and other metabolic features in patients with SOD and evaluate relationships with elevated body mass index (BMI).</p><p><strong>Methods: </strong>Children with SOD above 5 years of age attending Perth Children's Hospital participated. A CosMED Q-NRG indirect calorimeter was used to calculate mean measure REE (mREE). This was compared with predictive REE (pREE) based on the Schofield equation to determine mREE/pREE quotient. A BMI z-score >1 was considered elevated. Parents/carers completed a questionnaire about pituitary function, the Hyperphagia Questionnaire and the Sleep Disturbances Scale for Children (SDSC).</p><p><strong>Results: </strong>Twenty-six participants underwent testing (9 female, mean age 12.1 years) with 11 having elevated BMI and 15 with pituitary hormone deficiencies. Mean mREE was 1309 kcal/day (838-1732), mREE/pREE quotient was 88.8% ± 10.1. mREE/pREE quotient was similar in those with elevated BMI compared with normal BMI (83.3% ± 12.5 vs 92.1% ± 7.2, <i>P</i> = .068). Those with midline defects had a higher mREE/pREE quotient (91.8% ± 8.1 vs 80.4% ± 11.3, <i>P</i> = .026). Hyperphagia and SDSC scores were similar between BMI groups. Hyperphagia domain scores were higher in children with multiple hypopituitarism, pituitary structural defects, and normal septum pellucidum (<i>P</i> = .044, .042, and .033, respectively).</p><p><strong>Conclusion: </strong>Children with SOD had lower mREE than predicted and hyperphagia scores were higher in those with biochemical or structural pituitary changes, suggesting that hypothalamic dysfunction could drive BMI elevation in SOD. 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引用次数: 0
摘要
背景：视隔发育不良（SOD）是先天性垂体功能低下的主要原因，已知与高达44%的儿童超重和肥胖有关。考虑到下丘脑在激素调节中的作用，我们试图评估静息能量消耗（REE）、食欲和身体活动与SOD的关系。目的：探讨超氧化物歧化酶（SOD）患者的REE及其他代谢特征，并评价其与体重指数（BMI）升高的关系。方法：参与在珀斯儿童医院就诊的5岁以上SOD患儿。采用CosMED Q-NRG间接量热仪计算平均测量REE （mREE）。将其与基于Schofield方程的预测REE （pREE）进行比较，以确定mREE/pREE商。BMI z-评分bb0.1被认为是升高。家长/照顾者填写垂体功能问卷、嗜食问卷和儿童睡眠障碍量表（SDSC）。结果：26名参与者接受了测试（9名女性，平均年龄12.1岁），其中11名BMI升高，15名垂体激素缺乏。平均mREE为1309 kcal/day (838 ~ 1732)， mREE/pREE商为88.8%±10.1。BMI升高组的mREE/pREE商数与BMI正常组相似（83.3%±12.5 vs 92.1%±7.2,P = 0.068）。中线缺陷组mREE/pREE比值较高（91.8%±8.1 vs 80.4%±11.3,P = 0.026）。暴饮暴食和SDSC评分在BMI组之间相似。多发性垂体功能低下、垂体结构缺损、透明隔正常患儿贪食区评分较高（P值分别为0.044、0.042、0.033）。结论：SOD患儿的mREE低于预期，而垂体生化或结构性改变患儿的贪食评分较高，提示下丘脑功能障碍可能导致SOD的BMI升高。间接量热法可用于指导超氧化物歧化酶超重和肥胖的管理。
Resting Energy Expenditure and Metabolic Features in Children With Septo-Optic Dysplasia.
Context: Septo-optic dysplasia (SOD) is a major cause of congenital hypopituitarism and is known to be associated with overweight and obesity in up to 44% of children. Given the role of the hypothalamus in hormonal regulation, we sought to assess the association of resting energy expenditure (REE), appetite and physical activity with SOD.
Objective: To characterize REE and other metabolic features in patients with SOD and evaluate relationships with elevated body mass index (BMI).
Methods: Children with SOD above 5 years of age attending Perth Children's Hospital participated. A CosMED Q-NRG indirect calorimeter was used to calculate mean measure REE (mREE). This was compared with predictive REE (pREE) based on the Schofield equation to determine mREE/pREE quotient. A BMI z-score >1 was considered elevated. Parents/carers completed a questionnaire about pituitary function, the Hyperphagia Questionnaire and the Sleep Disturbances Scale for Children (SDSC).
Results: Twenty-six participants underwent testing (9 female, mean age 12.1 years) with 11 having elevated BMI and 15 with pituitary hormone deficiencies. Mean mREE was 1309 kcal/day (838-1732), mREE/pREE quotient was 88.8% ± 10.1. mREE/pREE quotient was similar in those with elevated BMI compared with normal BMI (83.3% ± 12.5 vs 92.1% ± 7.2, P = .068). Those with midline defects had a higher mREE/pREE quotient (91.8% ± 8.1 vs 80.4% ± 11.3, P = .026). Hyperphagia and SDSC scores were similar between BMI groups. Hyperphagia domain scores were higher in children with multiple hypopituitarism, pituitary structural defects, and normal septum pellucidum (P = .044, .042, and .033, respectively).
Conclusion: Children with SOD had lower mREE than predicted and hyperphagia scores were higher in those with biochemical or structural pituitary changes, suggesting that hypothalamic dysfunction could drive BMI elevation in SOD. Indirect calorimetry may be used to guide the management of overweight and obesity in SOD.
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