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Mark R Postma Department of Endocrinology, University Medical Center Groningen, University of Groningen, Groningen, The Netherlands

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Pia Burman Department of Endocrinology, Skane University Hospital Malmö, University of Lund, Lund, Sweden

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André P van Beek Department of Endocrinology, University Medical Center Groningen, University of Groningen, Groningen, The Netherlands

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Introduction:

Adult-onset growth hormone deficiency (AGHD) is usually the last deficiency to be substituted in hypopituitarism. In children with documented GH deficiency, treatment without delay is crucial for achieving optimal effects on growth and development. In adults, it is not known whether a delay in treatment initiation influences biochemical response and the favourable physiological effects resulting from GH replacement therapy (GHRT).

Methods:

A total of 1085 GH-deficient adults from KIMS (Pfizer International Metabolic Database) were included, adequately replaced with all pituitary hormones except for GH at baseline. Patients were stratified by sex and age (20–50 years and ≥50 years) and subsequently divided into two groups below and above the median duration of unsubstituted AGHD for that subgroup. The median time of unsubstituted GHD for the total cohort was 2.53 years (P5 = 0.35, P95 = 24.42).

Results:

Beneficial effects of 4 years of GHRT were observed on lipids and quality of life in all subgroups. A decrease in waist circumference was observed only in older (>50 years) patients. There was no difference in IGF-I SDS and in GH dose required to normalize IGF-I in patients with a duration of unsubstituted AGHD above or below the median. No relevant differences were found between the groups for anthropometric measures, cardiovascular risk factors and quality of life scores.

Conclusion:

In contrast to GHD in children and adolescents, no difference could be established in treatment response between early or late initiation of GHRT in AGHD in terms of required GH dose, IGF-I, metabolic health and quality of life.

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Agnieszka Pazderska Department of Endocrinology, Newcastle-upon-Tyne Hospitals, Newcastle upon Tyne, UK
Department of Endocrinology, St James’s Hospital, Dublin, Ireland

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Yaasir Mamoojee Department of Endocrinology, Newcastle-upon-Tyne Hospitals, Newcastle upon Tyne, UK

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Satish Artham Department of Endocrinology, Newcastle-upon-Tyne Hospitals, Newcastle upon Tyne, UK

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Margaret Miller Department of Endocrinology, Newcastle-upon-Tyne Hospitals, Newcastle upon Tyne, UK

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Stephen G Ball Department of Endocrinology, Central Manchester University Hospitals, Manchester, UK
Department of Endocrinology, University of Manchester, Manchester, UK

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Tim Cheetham Endocrine Research Group, Institute of Genetic Medicine, University of Newcastle-upon-Tyne, Newcastle upon Tyne, UK
Department of Paediatric Endocrinology & Diabetes, Newcastle-upon-Tyne Hospitals, Newcastle upon Tyne, UK

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Richard Quinton Department of Endocrinology, Newcastle-upon-Tyne Hospitals, Newcastle upon Tyne, UK
Endocrine Research Group, Institute of Genetic Medicine, University of Newcastle-upon-Tyne, Newcastle upon Tyne, UK

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presented with life-changing injuries resulting from major self-harm episode influenced by low self-esteem. Median age at treatment initiation was 56 years (range 38.4–69.5). Treatment over the first year was with IM Sustanon 250 mg administered monthly

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Yuka Goto Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Yoshie Otsuka Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Kenji Ashida Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Ayako Nagayama Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Nao Hasuzawa Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Shimpei Iwata Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Kento Hara Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Munehisa Tsuruta Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Nobuhiko Wada Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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Seiichi Motomura Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan
Division of Endocrinology and Metabolism, Diabetes Center, Kurume Medical Center, Kokubu-machi, Kurume-city, Fukuoka, Japan

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Yuji Tajiri Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan
Division of Endocrinology and Metabolism, Diabetes Center, Kurume Medical Center, Kokubu-machi, Kurume-city, Fukuoka, Japan

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Masatoshi Nomura Division of Endocrinology and Metabolism, Department of Internal Medicine, Kurume University School of Medicine, Asahi-machi, Kurume-city, Fukuoka, Japan

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glucose consumption rate ( 12 ) in the muscle. However, it remained unclear whether SGLT2 inhibitor administration can improve insulin sensitivity and rapidly reduce plasma glucose concentrations in humans during the early phase of treatment initiation

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Selma Flora Nordqvist Department of Medical Endocrinology and Metabolism, Copenhagen University Hospital Rigshospitalet, Copenhagen, Denmark

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Victor Brun Boesen Department of Medical Endocrinology and Metabolism, Copenhagen University Hospital Rigshospitalet, Copenhagen, Denmark

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Åse Krogh Rasmussen Department of Medical Endocrinology and Metabolism, Copenhagen University Hospital Rigshospitalet, Copenhagen, Denmark

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Ulla Feldt-Rasmussen Department of Medical Endocrinology and Metabolism, Copenhagen University Hospital Rigshospitalet, Copenhagen, Denmark
Institute of Clinical Medicine, University of Copenhagen, Copenhagen, Denmark

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Laszlo Hegedüs Department of Endocrinology and Metabolism, Odense University Hospital, Odense, Denmark

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Steen Joop Bonnema Department of Endocrinology and Metabolism, Odense University Hospital, Odense, Denmark

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Per Karkov Cramon Department of Clinical Physiology and Nuclear Medicine, Bispebjerg and Frederiksberg Hospital, Copenhagen, Denmark

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Torquil Watt Institute of Clinical Medicine, University of Copenhagen, Copenhagen, Denmark
Department of Internal Medicine, Herlev Gentofte Hospital, Copenhagen, Denmark

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Mogens Groenvold Department of Public Health, University of Copenhagen, Copenhagen, Denmark
Department of Palliative Medicine, Bispebjerg Hospital, Copenhagen, Denmark

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Jakob Bue Bjorner Department of Public Health, University of Copenhagen, Copenhagen, Denmark
National Research Centre for the Working Environment, Copenhagen, Denmark
QualityMetric Incorporated, LLC, Johnston, Rhode Island, USA

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University Hospital), were invited to complete ThyPRO prior to and 6 weeks after treatment initiation. At follow-up, patients also rated their change since baseline; both overall and for each of 13 specific domains measured by ThyPRO. Aiming at samples around

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Jaafar Jaafar Division of Endocrinology and Diabetology, Geneva University Hospitals and Faculty of Medicine, Geneva, Switzerland

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Eugenio Fernandez Department of Oncology, Geneva University Hospitals and Faculty of Medicine, Geneva, Switzerland

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Heba Alwan Division of Endocrinology and Diabetology, Geneva University Hospitals and Faculty of Medicine, Geneva, Switzerland

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Jacques Philippe Division of Endocrinology and Diabetology, Geneva University Hospitals and Faculty of Medicine, Geneva, Switzerland

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onset of hypothyroidism and hyperT in patients treated with durvalumab was 42 and 43 days, respectively. In fact, the range of time to the occurrence of a thyroid-related iAE was very large and varied from 1 day after treatment initiation to 31 months

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Xi Wang Department of Obstetrics and Gynaecology, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China

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Qi Yu Department of Obstetrics and Gynaecology, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China

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Objective

To evaluate the safety and efficacy of letrozole in girls with progressive precocious puberty (PP) associated with McCune–Albright syndrome (MAS).

Design

Monocentric retrospective cross-sectional and longitudinal study of consecutive patients.

Patients

Ten MAS patients treated at Peking Union Medical College Hospital between September 1999 and December 2017 were retrospectively reviewed; those with complications due to PP were followed.

Results

The mean age at letrozole initiation was 4.5 ± 2.6 years, while the mean duration of treatment was 3.3 ± 2.4 years. Letrozole was highly effective at decreasing the rate of skeletal maturation, with a significant decrease in the bone age-to-chronological age (BA/CA) ratio from 1.9 ± 1.1 pre-treatment to 1.5 ± 1.2 on letrozole treatment (P = 0.016). Moreover, growth velocity Z-scores declined from 0.41 ± 0.5 to −0.2 ± 0.31 with treatment (P < 0.001). Predicted adult height Z-scores increased significantly from −2.03 ± 2.33 at baseline to 1.13 ± 0.84 following treatment initiation (P = 0.029). Moreover, vaginal bleeding declined significantly on letrozole.

Conclusions

Our findings suggest that letrozole may be an effective therapy in some girls with MAS, as treatment results in improved BA/CA ratio, growth velocity and predicted adult height. Possible adverse effects include nettle rash.

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Hanna Karhapää Medical Faculty, University of Helsinki, Helsinki, Finland
Department of Oncology, Comprehensive Cancer Centre, Helsinki University Hospital, Helsinki, Finland

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Siru Mäkelä Medical Faculty, University of Helsinki, Helsinki, Finland
Department of Oncology, Comprehensive Cancer Centre, Helsinki University Hospital, Helsinki, Finland

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Hanna Laurén Medical Faculty, University of Helsinki, Helsinki, Finland
Department of Radiology, HUS Medical Imaging Centre, University of Helsinki and Helsinki University Hospital, Helsinki, Finland

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Marjut Jaakkola Medical Faculty, University of Helsinki, Helsinki, Finland
Department of Radiology, HUS Medical Imaging Centre, University of Helsinki and Helsinki University Hospital, Helsinki, Finland

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Camilla Schalin-Jäntti Medical Faculty, University of Helsinki, Helsinki, Finland
Endocrinology, Abdominal Centre, University of Helsinki and HUS, Helsinki, Finland

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Micaela Hernberg Medical Faculty, University of Helsinki, Helsinki, Finland
Department of Oncology, Comprehensive Cancer Centre, Helsinki University Hospital, Helsinki, Finland

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multidisciplinary meeting before treatment initiation. We collected the following data from the patient records: patient age at treatment initiation, sex, and ECOG performance status. Furthermore, we listed the characteristics of metastatic disease, including

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Tristan Avril Pediatric Endocrinology Department, CHU Bicetre, Assistance Publique-Hôpitaux de Paris, Reference Center for Rare Disease CRMR DevGen, Le Kremlin Bicêtre, Paris, France

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Quentin Hennocq Datascience platform, Imagine Institute, Université Paris Cité, Paris, France

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Anne-Sophie Lambert Pediatric Endocrinology Department, CHU Bicetre, Assistance Publique-Hôpitaux de Paris, Reference Center for Rare Disease CRMR DevGen, Le Kremlin Bicêtre, Paris, France

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Juliane Leger Pediatric Endocrinology Department, CHU Robert Debré, Assistance Publique-Hôpitaux de Paris, Reference Center for Rare Disease CRMR de la Croissance et du Développement, Paris, France
Université Paris Cité, Faculté de Santé, UFR de Médecine, Paris, France

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Dominique Simon Pediatric Endocrinology Department, CHU Robert Debré, Assistance Publique-Hôpitaux de Paris, Reference Center for Rare Disease CRMR de la Croissance et du Développement, Paris, France

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Laetitia Martinerie Pediatric Endocrinology Department, CHU Robert Debré, Assistance Publique-Hôpitaux de Paris, Reference Center for Rare Disease CRMR de la Croissance et du Développement, Paris, France
Université Paris Cité, Faculté de Santé, UFR de Médecine, Paris, France
Inserm UMR1185, Le Kremlin Bicetre, Paris, France

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Claire Bouvattier Pediatric Endocrinology Department, CHU Bicetre, Assistance Publique-Hôpitaux de Paris, Reference Center for Rare Disease CRMR DevGen, Le Kremlin Bicêtre, Paris, France
Inserm UMR1185, Le Kremlin Bicetre, Paris, France
Paris-Saclay University, Paris, France

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Helsinki. Results Patient characteristics At treatment initiation, there was no significant clinical difference in the two groups concerning the following assessment: patient age (5.1 ± 3.5 months in the pump group vs 13 ± 17.7 months in the

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Alicia Romano Department of Pediatrics, New York Medical College, Valhalla, New York, USA

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Juan Pablo Kaski Centre for Inherited Cardiovascular Diseases, Great Ormond Street Hospital & UCL Institute of Cardiovascular Science, London, UK

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Jovanna Dahlgren Department of Paediatrics, Region Västra Götaland, Sahlgrenska University Hospital, Gothenburg, Sweden

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Nicky Kelepouris US Medical Affairs, Novo Nordisk Inc., Plainsboro, New Jersey, USA

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Alberto Pietropoli Global Medical Affairs, Novo Nordisk Health Care AG, Zurich, Switzerland

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Tilman R Rohrer Department of Pediatric Endocrinology, University Children’s Hospital, Saarland University Medical Center, Homburg, Germany

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Michel Polak Paediatric Endocrinology, Diabetology and Gynaecology Department, Hôpital Universitaire Necker Enfants-Malades, AP-HP, Université de Paris, Imagine Institute, Paris, France

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aneurysm. The first four patients had no reported SAEs. The abdominal aortic aneurysm was diagnosed in a patient with a PTPN11 mutation 2.2 years after GH treatment initiation. This patient was also diagnosed with Crohn’s disease (1.8 years after

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Mark J C van Treijen Department of Endocrine Oncology, University Medical Center Utrecht, Utrecht, The Netherlands
Center for Neuroendocrine Tumors, ENETS Center of Excellence, Netherlands Cancer Institute, University Medical Center Utrecht, Utrecht, The Netherlands

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Catharina M Korse Center for Neuroendocrine Tumors, ENETS Center of Excellence, Netherlands Cancer Institute, University Medical Center Utrecht, Utrecht, The Netherlands
Department of Clinical Chemistry, The Netherlands Cancer Institute, Amsterdam, The Netherlands

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Wieke H Verbeek Center for Neuroendocrine Tumors, ENETS Center of Excellence, Netherlands Cancer Institute, University Medical Center Utrecht, Utrecht, The Netherlands
Department of Gastroenterology, The Netherlands Cancer Institute, Amsterdam, The Netherlands

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Margot E T Tesselaar Center for Neuroendocrine Tumors, ENETS Center of Excellence, Netherlands Cancer Institute, University Medical Center Utrecht, Utrecht, The Netherlands
Department of Medical Oncology, The Netherlands Cancer Institute, Amsterdam, The Netherlands

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Gerlof D Valk Department of Endocrine Oncology, University Medical Center Utrecht, Utrecht, The Netherlands
Center for Neuroendocrine Tumors, ENETS Center of Excellence, Netherlands Cancer Institute, University Medical Center Utrecht, Utrecht, The Netherlands

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received sunitinib (2%). Treatment response was defined as PFS ≥ 12 months after treatment initiation. A total of 34 patients showed response to treatment (69%). The median TTP after the start of treatment was 20 months (2–66 months). Significant

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