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Thomas M Braxton School of Biosciences, Cardiff University, Cardiff, UK

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Dionne E A Sarpong School of Biosciences, Cardiff University, Cardiff, UK

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Janine L Dovey School of Biosciences, Cardiff University, Cardiff, UK

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Anne Guillou IGF, CNRS, INSERM, University of Montpellier, Montpellier, France

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Bronwen A J Evans School of Medicine, Cardiff University, Cardiff, UK

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Juan M Castellano Physiology Section, Faculty of Medicine, University of Cordoba, and Instituto Maimonides de Investigacion Biomedica de Cordoba (IMBIC), Cordoba, Spain

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Bethany E Keenan School of Engineering, Cardiff University, Cardiff, UK

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Saja Baraghithy Obesity and Metabolism Laboratory, Institute for Drug Research, School of Pharmacy, Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, Israel

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Sam L Evans School of Engineering, Cardiff University, Cardiff, UK

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Manuel Tena-Sempere Physiology Section, Faculty of Medicine, University of Cordoba, and Instituto Maimonides de Investigacion Biomedica de Cordoba (IMBIC), Cordoba, Spain
CIBER Fisiopatologia de la Obesidad y Nutrición (CIBEROBN), Instituto de Salud Carlos III, Cordoba, Spain

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Patrice Mollard IGF, CNRS, INSERM, University of Montpellier, Montpellier, France

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Joseph Tam Obesity and Metabolism Laboratory, Institute for Drug Research, School of Pharmacy, Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, Israel

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Timothy Wells School of Biosciences, Cardiff University, Cardiff, UK

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Human Prader–Willi syndrome (PWS) is characterised by impairments of multiple systems including the growth hormone (GH) axis and skeletal growth. To address our lack of knowledge of the influence of PWS on skeletal integrity in mice, we have characterised the endocrine and skeletal phenotype of the PWS-IC del mouse model for ‘full’ PWS and determined the impact of thermoneutrality. Tibial length, epiphyseal plate width and marrow adiposity were reduced by 6, 18 and 79% in male PWS-IC del mice, with osteoclast density being unaffected. Similar reductions in femoral length accompanied a 32% reduction in mid-diaphyseal cortical diameter. Distal femoral Tb.N was reduced by 62%, with individual trabeculae being less plate-like and the lattice being more fragmented (Tb.Pf increased by 63%). Cortical strength (ultimate moment) was reduced by 26% as a result of reductions in calcified tissue strength and the geometric contribution. GH and prolactin contents in PWS-IC del pituitaries were reduced in proportion to their smaller pituitary size, with circulating IGF-1 concentration reduced by 37–47%. Conversely, while pituitary luteinising hormone content was halved, circulating gonadotropin concentrations were unaffected. Although longitudinal growth, marrow adiposity and femoral geometry were unaffected by thermoneutrality, strengthened calcified tissue reversed the weakened cortex of PWS-IC del femora. While underactivity of the GH axis may be due to loss of Snord116 expression and impaired limb bone geometry and strength due to loss of Magel2 expression, comprehensive analysis of skeletal integrity in the single gene deletion models is required. Our data imply that thermoneutrality may ameliorate the elevated fracture risk associated with PWS.

Article Type:
Research Article
DOI:
https://doi.org/10.1530/JOE-19-0279
Volume/Issue:
Volume 243: Issue 3
Restricted access
R. V. BROOKS
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B. S. HETZEL
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R. R. McSWINEY
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F. T. G. PRUNTY
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SUMMARY

Changes in response of the adrenal cortex to corticotrophin caused by the simultaneous intravenous administration of tri-iodothyronine have been studied in two patients with anterior pituitary insufficiency.

Tri-iodothyronine caused increases in urinary excretion of ketogenic steroids and in plasma hydrocortisone. Blood eosinophils fell sharply. In addition, changes in sodium and potassium balance, and fasting blood sugar levels were observed which were consistent with increased activity of the adrenal cortex. Rapid changes in b.m.r., serum creatine and blood cholesterol were those known to be produced by tri-iodothyronine alone; so, to a large extent, was the increased negative nitrogen balance.

Article Type:
Research Article
DOI:
https://doi.org/10.1677/joe.0.0150320
Volume/Issue:
Volume 15: Issue 3
Restricted access
P. CUSHMAN Jr.
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J. G. HILTON
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SUMMARY

Intravenous 8 hr. metyrapone tests were performed in 12 patients with probable pituitary tumours. Plasma 17-hydroxycorticosteroids and substance S concentrations were measured, and the concentrations of 11-deoxycortisol present after 8 hr. of continuous metyrapone administration were employed as the quantitative criterion of responsiveness. Plasma 11-deoxycortisol, undetectable at the start, rose at 8 hr. to 10·1 ± 1·5 s.e. (range 6–14) μg./100 ml. in the 15 normal control subjects. Three patterns of pituitary adrenal function were seen in the patients. Group I (three cases) had secondary adrenal insufficiency. A second group (five cases) was normal by all tests. The third group of patients (four cases) had normal baseline plasma and urinary corticosteroids but was distinguished from the normal subjects by subnormal 11-deoxycortisol levels after metyrapone. Two subjects had also subnormal responses to corticotrophin (ACTH), indicating partial secondary hypoadrenalism. In the two others whose response to ACTH stimulation was normal, impairment of ACTH release would not have been suspected without the metyrapone test. It is in the detection of this type of patient with occult pituitary insufficiency that tests with metyrapone may be of great value.

Article Type:
Research Article
DOI:
https://doi.org/10.1677/joe.0.0430377
Volume/Issue:
Volume 43: Issue 3
Restricted access
John A H Wass Department of Endocrinology, Oxford Centre for Diabetes, Endocrinology and Metabolism, Churchill Hospital, University of Oxford, Old Road, Headington, Oxford OX3 7LJ, UK

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Raghava Reddy Department of Endocrinology, Oxford Centre for Diabetes, Endocrinology and Metabolism, Churchill Hospital, University of Oxford, Old Road, Headington, Oxford OX3 7LJ, UK

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any specific grant from any funding agency in the public, commercial or not-for-profit sector. References Burman P Deijen JB 1998 Quality of life and cognitive function in patients with pituitary insufficiency . Psychotherapy and

Article Type:
Research Article
DOI:
https://doi.org/10.1677/JOE-10-0126
Volume/Issue:
Volume 207: Issue 2
Free access
Eun Jig Lee Endocrinology, Metabolism, and Molecular Medicine, Northwestern University, Feinberg School of Medicine, Chicago, Illinois 60611, USA

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J Larry Jameson Endocrinology, Metabolism, and Molecular Medicine, Northwestern University, Feinberg School of Medicine, Chicago, Illinois 60611, USA

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means to reduce the immune responsiveness to adenoviruses. Gene therapy strategies for treating pituitary insufficiency Vector considerations for long-term hormone replacement Long-term expression of a

Article Type:
Other
DOI:
https://doi.org/10.1677/joe.1.06023
Volume/Issue:
Volume 185: Issue 3
Free access
E Nieves-Martinez Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy

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W E Sonntag Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy

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A Wilson Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy

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A Donahue Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy

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D P Molina Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy

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J Brunso-Bechtold Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy
Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy

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M M Nicolle Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy
Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy
Neuroscience Program, Physiology and Pharmacology, Department of Internal Medicine, Reynolds Oklahoma Center on Aging, Neurobiology and Anatomy

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JJ Laron Z 1968 Psychological aspects of pituitary insufficiency in children and adolescents with special reference to growth hormone . Israel Journal of Medical Sciences 4 953 – 961 . Galatzer A Aran O Nagelberg N Rubitzek J Laron

Article Type:
Research Article
DOI:
https://doi.org/10.1677/JOE-09-0323
Volume/Issue:
Volume 204: Issue 1
Free access
Marion Walser Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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Maria Teresa Samà Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden
Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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Ruth Wickelgren Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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Maria Åberg Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden
Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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Mohammad Bohlooly-Y Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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Bob Olsson Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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Jan Törnell Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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Jörgen Isgaard Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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N David Åberg Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden
Laboratory of Experimental Endocrinology, Endocrinology, Department of Clinical Chemistry and Transfusion Medicine, Institute of Neuroscience and Physiology, Department of Primary Health Care, AstraZeneca Research and Development, Clinical Neurochemistry Laboratory, InnoExt AB, Department of Internal Medicine, Sahlgrenska University Hospital, University of Gothenburg, Gröna Stråket 8, SE-41345 Göteborg, Sweden

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Characterization of a neuronal subtype of insulin-like growth factor I receptor . Journal of Biological Chemistry 262 1618 – 1622 . Burman P Deijen JB 1998 Quality of life and cognitive function in patients with pituitary insufficiency . Psychotherapy

Article Type:
Research Article
DOI:
https://doi.org/10.1530/JOE-12-0077
Volume/Issue:
Volume 215: Issue 2
Free access
Han Yan
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Matthew Mitschelen
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Georgina V Bixler Reynolds Oklahoma Center on Aging, Genome Sciences Facility, Donald W. Reynolds Department of Geriatric Medicine, University of Oklahoma Health Sciences Center, 975 NE 10th ST BRC 1305, Oklahoma City, Oklahoma 73104, USA

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Robert M Brucklacher Reynolds Oklahoma Center on Aging, Genome Sciences Facility, Donald W. Reynolds Department of Geriatric Medicine, University of Oklahoma Health Sciences Center, 975 NE 10th ST BRC 1305, Oklahoma City, Oklahoma 73104, USA

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Julie A Farley
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Song Han
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Willard M Freeman Reynolds Oklahoma Center on Aging, Genome Sciences Facility, Donald W. Reynolds Department of Geriatric Medicine, University of Oklahoma Health Sciences Center, 975 NE 10th ST BRC 1305, Oklahoma City, Oklahoma 73104, USA

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William E Sonntag
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Genomics 1 26 doi:10.1186/1755-8794-1-26 . Burman P Deijen JB 1998 Quality of life and cognitive function in patients with pituitary insufficiency . Psychotherapy and Psychosomatics 67 154 – 167 . doi:10.1159/000012276 . Burman P Broman

Article Type:
Research Article
DOI:
https://doi.org/10.1530/JOE-11-0200
Volume/Issue:
Volume 211: Issue 1
Free access
Eva Baquedano Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain
Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain

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Julie A Chowen Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain
Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain

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Jesús Argente Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain
Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain
Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain

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Laura M Frago Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain
Department of Pediatrics, Department of Endocrinology, Centro de Investigación Biomédica en Red de fisiopatología de la obesidad y nutrición (CIBERobn), Universidad Autónoma de Madrid, Madrid, Spain

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. International Journal of Biochemistry & Cell Biology 40 1984 – 1989 . ( doi:10.1016/j.biocel.2007.07.008 ) Burman P Deijen J 1998 Quality of life and cognitive function in patients with pituitary insufficiency . Psychotherapy

Article Type:
Research Article
DOI:
https://doi.org/10.1530/JOE-13-0053
Volume/Issue:
Volume 218: Issue 3
Free access
Alfhild Grönbladh
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Jenny Johansson
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Anatole Nöstl Division of Biological Research on Drug Dependence, Department of Building, Department of Pharmaceutical Biosciences, Uppsala University, PO Box 591, S-751 24 Uppsala, Sweden

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Fred Nyberg
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Mathias Hallberg
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.1055/s-2007-965808 ) Burman P Deijen JB 1998 Quality of life and cognitive function in patients with pituitary insufficiency . Psychotherapy and Psychosomatics 67 154 – 167 . ( doi:10.1159/000012276 ) Burman P Broman JE Hetta J Wiklund

Article Type:
Research Article
DOI:
https://doi.org/10.1530/JOE-12-0315
Volume/Issue:
Volume 216: Issue 1
Free access