NREM Sleep Architecture and Relation to GH/IGF-1 Axis in Laron Syndrome

被引:5
作者
Verrillo, Elisabetta [1 ]
Bizzarri, Carla [2 ]
Cappa, Marco [2 ]
Bruni, Oliviero [3 ]
Pavone, Martino [1 ]
Cutrera, Renato [1 ]
机构
[1] Bambino Gesu Pediat Hosp & Res Inst, Resp Unit, IT-00165 Rome, Italy
[2] Bambino Gesu Pediat Hosp & Res Inst, Endocrinol Unit, IT-00165 Rome, Italy
[3] Univ Roma La Sapienza, Ctr Pediat Sleep Disorders, Dept Dev Neurol & Psychiat, Rome, Italy
来源
HORMONE RESEARCH IN PAEDIATRICS | 2010年 / 73卷 / 05期
关键词
Laron syndrome; Cyclic alternating pattern; Growth hormone; GROWTH-FACTOR-I; PRADER-WILLI-SYNDROME; HORMONE INSENSITIVITY; ALTERNATING PATTERN; LONG-TERM; IGF-I; CHILDREN; SECRETION;
D O I
10.1159/000308177
中图分类号
R5 [内科学];
学科分类号
1002 ; 100201 ;
摘要
Background: Laron syndrome (LS), known as growth hormone (GH) receptor deficiency, is a rare form of inherited GH resistance. Sleep disorders were described as a common feature of adult LS patients, while no data are available in children. Bi-directional interactions between human sleep and the somatotropic system were previously described, mainly between slow wave sleep and the nocturnal GH surge. Aims: To analyze the sleep macro- and microstructure in LS and to evaluate the influence of substitutive insulin-like growth factor 1 (IGF-1) therapy on it. Methods: Two young LS females underwent polysomnography; the first study was performed during IGF-1 therapy, the second one after a 3-month wash-out period. Results: In both patients, the sleep macrostructure showed that time in bed, sleep period time, total sleep time, sleep efficiency and rapid eye movement ( REM) percentage were all increased during wash-out. The sleep microstructure ( cyclic alternating pattern: CAP) showed significantly higher EEG slow oscillations (A1%) in NREM sleep, both during IGF-1 therapy and wash-out. Conclusions: Sleep macrostructure in LS children is slightly affected by substitutive IGF-1 therapy. Sleep microstructure shows an increase of A1%, probably related to abnormally high hypothalamic GHRH secretion, due to GH insensitivity. Copyright (C) 2010 S. Karger AG, Basel
引用
收藏
页码:414 / 419
页数:6
相关论文
共 20 条
[1]   Sleep cyclic alternating pattern in normal school-age children [J].
Bruni, O ;
Ferri, R ;
Miano, S ;
Verrillo, E ;
Vittori, E ;
Della Marca, G ;
Farina, B ;
Mennuni, G .
CLINICAL NEUROPHYSIOLOGY, 2002, 113 (11) :1806-1814
[2]   Long-term treatment with recombinant insulin-like growth factor (IGF)-I in children with severe IGF-I deficiency due to growth hormone insensitivity [J].
Chernausek, Steven D. ;
Backeljauw, Philippe F. ;
Frane, James ;
Kuntze, Joyce ;
Underwood, Louis E. .
JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, 2007, 92 (03) :902-910
[3]   The role of recombinant human insulin-like growth factor-I in treating children with short stature [J].
Collett-Solberg, Paulo F. ;
Misra, Madhusmita .
JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, 2008, 93 (01) :10-18
[4]   THE EFFECT OF RECOMBINANT HUMAN INSULIN-LIKE GROWTH FACTOR-I TREATMENT ON GROWTH-HORMONE SECRETION IN 2 SUBJECTS WITH GROWTH-HORMONE INSENSITIVITY (LARON SYNDROME) [J].
COTTERILL, AM ;
CAMACHOHUBNER, C ;
HOLLY, JMP ;
SAVAGE, MO .
CLINICAL ENDOCRINOLOGY, 1993, 39 (01) :119-122
[5]   Severe obstructive sleep apnoea syndrome in an adult patient with Laron syndrome [J].
Dagan, Y ;
Abadi, J ;
Lifschitz, A ;
Laron, Z .
GROWTH HORMONE & IGF RESEARCH, 2001, 11 (04) :247-249
[6]   Growth hormone receptor gene mutations in two Italian patients with Laron Syndrome [J].
Fassone, L. ;
Corneli, G. ;
Bellone, S. ;
Camacho-Hubner, C. ;
Aimaretti, G. ;
Cappa, M. ;
Ubertini, G. ;
Bona, G. .
JOURNAL OF ENDOCRINOLOGICAL INVESTIGATION, 2007, 30 (05) :417-420
[7]  
Kornreich L, 2002, AM J NEURORADIOL, V23, P625
[8]   Commentary - The essential role of IGF-I: Lessons from the long-term study and treatment of children and adults with Laron syndrome [J].
Laron, Z .
JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, 1999, 84 (12) :4397-4404
[9]   Extensive personal experience - Laron syndrome (primary growth hormone resistance or insensitivity): The personal experience 1958-2003 [J].
Laron, Z .
JOURNAL OF CLINICAL ENDOCRINOLOGY & METABOLISM, 2004, 89 (03) :1031-1044
[10]   GHRH and sleep [J].
Obal, F ;
Krueger, JM .
SLEEP MEDICINE REVIEWS, 2004, 8 (05) :367-377