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Article ; Online: Near adult height and BMI changes in growth hormone treated short children with Noonan syndrome: the Belgian experience.

De Schepper, Jean / Thomas, Muriel / Huysentruyt, Koen / Becker, Marianne / Boros, Emese / Casteels, Kristina / Chivu, Olimpia / De Waele, Kathleen / Dotremont, Hilde / Lysy, Philippe A / Massa, Guy / Parent, Anne-Simone / Rochtus, Anne / Gies, Inge

Hormone research in paediatrics

2024

Abstract: Introduction A variable near adult height (NAH) outcome after growth hormone (GH) therapy in Noonan syndrome (NS) patients with short stature has been reported. The main objective of this study was to evaluate NAH and body mass index (BMI) evolution in a ...

Abstract	Introduction A variable near adult height (NAH) outcome after growth hormone (GH) therapy in Noonan syndrome (NS) patients with short stature has been reported. The main objective of this study was to evaluate NAH and body mass index (BMI) evolution in a large Belgian cohort of NS patients treated for short stature. The secondary objectives were to investigate whether sex, genotype, the presence of a thoracic deformity and/or a heart anomaly might affect NAH and to validate the recently developed NAH prediction model by Ranke et al. Methods Clinical and auxological data of GH treated short NS patients born before 2001 were extracted from the national Belgrow registry. NAH was available in 54 (35 male) genotyped NS using a gene panel of 9 genes, showing pathogenic variants in PTPN11 in 32 and in SOS1 in 5 patients, while in 17 patients gene panel analysis was inconclusive (no mutation group). Results After a median (P10; P90) duration of 5.4 (2.2-10.3) years of GH therapy with a median dose of 0.05 mg/kg/day NS patients reached a median NAH of -1.7 (-3.4; -0.8) SDS. Median total height gain was 1.1 (0.1; 2.3) SDS. Sex, genotype and the presence of a thoracic or cardiac malformation did not correlate with NAH or total height gain. Linear regression modelling revealed that height SDS at start (beta=0.90, p<0.001), mid-parental height SDS (beta =0.27; p=0.005), birth weight SDS (beta=0.15; p=0.051), age at start (beta=0.07; p=0032) were independently associated with NAH SDS. Median BMI SDS increased significantly (p<0.001) from -1.0 (-2.5; 0.0) at start to -0.2 (-1.5; 0.9) at NAH. The observed NAH in a subgroup of 44 patients with more than 3 years of GH treatment was not statistically different from the predicted NAH by the Noonan NAH prediction model of Ranke. Conclusion Long-term GH therapy at a dose of 0.05 mg/kg/day in short NS patients is effective in improving adult height and BMI, irrespective of the genotype and presence or absence of cardiac and or thoracic anomalies.
Language	English
Publishing date	2024-03-01
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	2537278-6
ISSN	1663-2826 ; 1663-2818
ISSN (online)	1663-2826
ISSN	1663-2818
DOI	10.1159/000538034
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Article: Efficacy and safety of a 4-year combination therapy of growth hormone and gonadotropin-releasing hormone analogue in pubertal girls with short predicted adult height.

Dotremont, Hilde / France, Annick / Heinrichs, Claudine / Tenoutasse, Sylvie / Brachet, Cécile / Cools, Martine / De Waele, Kathleen / Massa, Guy / Lebrethon, Marie-Christine / Gies, Inge / Van Besien, Jesse / Derycke, Christine / Ziraldo, Mathieu / De Schepper, Jean / Beauloye, Véronique / Verhulst, Stijn / Rooman, Raoul / den Brinker, Marieke

Frontiers in endocrinology

2023 Volume 14, Page(s) 1113750

Abstract: Objectives: To improve adult height in pubertal girls with a poor height prediction, treatment with growth hormone (GH) can be used in combination with a gonadotropin releasing hormone agonist (GnRHa), to delay closure of the growth plates. However, ... ...

Abstract	Objectives: To improve adult height in pubertal girls with a poor height prediction, treatment with growth hormone (GH) can be used in combination with a gonadotropin releasing hormone agonist (GnRHa), to delay closure of the growth plates. However, there are few studies to support this practice, and they show conflicting results. The objective of this trial is to assess the safety and efficacy of this combination treatment in early pubertal girls with a short predicted height, in comparison with matched controls. Design patients and methods: We designed an open-label, multicenter, interventional case-control study. Early pubertal girls with predicted adult height (PAH) below -2.5 SDS, were recruited in tertiary care centers in Belgium. They were treated for four years with GH and GnRHa. The girls were followed until adult height (AH) was reached. AH Results: Sixteen girls with mean age ( ± SD) at start of 11.0 years (± 1.3) completed the study protocol and follow-up. Their mean height ( ± SD) increased from 131.3 ± 4.1 cm (-2.3 ± 0.7 SDS) at start of treatment to 159.8 ± 4.7 cm (-1.1 ± 0.7 SDS) at AH. In matched controls, height increased from 132.3 ± 4.2 cm (-2.4 ± 0.5 SDS) to 153.2 ± 3.4 cm (-2.1 ± 0.6 SDS) (p<0.001). AH surpassed initial PAH by 12.0 ± 2.6 cm in treated girls; and by 4.2 ± 3.6 cm in the controls (p<0.001). Most treated girls reached normal adult height (>-2SD) (87.5%) and 68.7% reached or superseded the target height (TH), which was the case in only a minority of the controls (37.5% and 6.2%, respectively) (p= 0.003 and 0.001). A serious adverse event possibly related to the treatment, was a fracture of the metatarsals. Conclusion: A four-year GH/GnRHa treatment in early pubertal girls with a poor PAH seems safe and results in a clinically relevant and statistically significant increase in AH compared with matched historical controls. Clinical trial registration: ClinicalTrials.gov, identifier NCT00840944.
MeSH term(s)	Female ; Humans ; Adult ; Child ; Growth Hormone ; Gonadotropin-Releasing Hormone ; Case-Control Studies ; Body Height ; Human Growth Hormone/therapeutic use ; Puberty, Precocious/drug therapy
Chemical Substances	Growth Hormone (9002-72-6) ; Gonadotropin-Releasing Hormone (33515-09-2) ; Human Growth Hormone (12629-01-5)
Language	English
Publishing date	2023-03-17
Publishing country	Switzerland
Document type	Multicenter Study ; Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	2592084-4
ISSN	1664-2392
ISSN	1664-2392
DOI	10.3389/fendo.2023.1113750
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Article: Comparison of SARS-CoV-2 seroconversion in children with chronic diseases with healthy children and adults during the first waves of the COVID-19 pandemic.

Hoste, Levi / Prytula, Agnieszka / Dehoorne, Jo / De Bruyne, Ruth / Van Biervliet, Stephanie / De Waele, Kathleen / Maes, Evelyn / Bordon, Victoria / Vanlander, Arnaud / Claes, Karlien / Vande Walle, Johan / Schelstraete, Petra / Van Daele, Sabine / Haerynck, Filomeen

Frontiers in pediatrics

2023 Volume 11, Page(s) 1210181

Abstract: Background: Infection with severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is clinically diverse, and children have a low risk of developing severe coronavirus disease 2019 (COVID-19). However, children with chronic diseases have a ... ...

Abstract	Background: Infection with severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) is clinically diverse, and children have a low risk of developing severe coronavirus disease 2019 (COVID-19). However, children with chronic diseases have a potentially increased risk. Methods: We performed a prospective surveillance study with longitudinal serum SARS-CoV-2 anti-nucleocapsid antibody quantification and questionnaires in pediatric tertiary care patients during the first waves of the COVID-19 pandemic (November 2020-September 2021). The results were compared with those of healthy children and adults from the same geographic area. Results: We obtained 525 samples from 362 patients (M/F ratio of 1.3:1; median age of 11.1 years) comprising children with immune-suppressive or immune-modulating drugs (32.9%), inborn errors of immunity (23.5%), type 1 diabetes mellitus (15.2%), and rheumatic diseases (11.9%). A total of 51 (9.7%) samples were seropositive among 37/351 children (10.5%). Seropositivity increased from 5.8% in November-December 2020 to 21.6% in July-September 2021. Compared with adults, a longitudinal analysis revealed reduced seroprevalence but similar kinetics as in children from the same country. Demographic or social variables and disease characteristics did not correlate with seropositivity. Being obese and household contact with COVID-19-infected individuals significantly increased the odds of infection. The majority of seropositive patients had mild symptoms (21/37). One-third were asymptomatic and/or unaware of having COVID-19 (10/37). Four patients (4/37) needed hospitalization, with good clinical outcomes. Conclusions: Although harboring a chronic disease, we observed a low SARS-CoV-2 incidence in a cohort of pediatric tertiary care patients, comparable with healthy children during the first year of the pandemic. Infection was mostly associated with mild symptoms.
Language	English
Publishing date	2023-08-07
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	2711999-3
ISSN	2296-2360
ISSN	2296-2360
DOI	10.3389/fped.2023.1210181
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Article ; Online: Hypotonia and delayed motor development as an early presentation of Lowe syndrome: case report and literature review.

David, Sara / De Waele, Kathleen / De Wilde, Bram / Faes, Franny / Vanakker, Olivier / Walraedt, Sophie / Prytuła, Agnieszka

Acta clinica Belgica

2018 Volume 74, Issue 6, Page(s) 460–464

Abstract: We describe a boy who presented with neonatal hypotonia, followed by delayed motor development and growth impairment. Further evaluation revealed rickets caused by proximal renal tubular dysfunction. At age 3, the boy exhibited dysmorphic features and ... ...

Abstract	We describe a boy who presented with neonatal hypotonia, followed by delayed motor development and growth impairment. Further evaluation revealed rickets caused by proximal renal tubular dysfunction. At age 3, the boy exhibited dysmorphic features and bilateral cataract. Genetic analysis of the
MeSH term(s)	Cataract/diagnosis ; Cataract/etiology ; Child, Preschool ; Developmental Disabilities/diagnosis ; Developmental Disabilities/etiology ; Early Diagnosis ; Early Medical Intervention ; Fanconi Syndrome/diagnosis ; Fanconi Syndrome/etiology ; Genetic Testing/methods ; Humans ; Male ; Motor Disorders/diagnosis ; Motor Disorders/etiology ; Muscle Hypotonia/diagnosis ; Muscle Hypotonia/genetics ; Muscle Hypotonia/urine ; Mutation ; Oculocerebrorenal Syndrome/diagnosis ; Oculocerebrorenal Syndrome/genetics ; Oculocerebrorenal Syndrome/physiopathology ; Phosphoric Monoester Hydrolases/genetics
Chemical Substances	Phosphoric Monoester Hydrolases (EC 3.1.3.2) ; OCRL protein, human (EC 3.1.3.36)
Language	English
Publishing date	2018-12-03
Publishing country	England
Document type	Journal Article
ZDB-ID	390201-8
ISSN	2295-3337 ; 0001-5512 ; 1784-3286
ISSN (online)	2295-3337
ISSN	0001-5512 ; 1784-3286
DOI	10.1080/17843286.2018.1551743
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Article: A synchronous papillary and follicular thyroid carcinoma presenting as a large toxic nodule in a female adolescent.

Van Vlaenderen, Joke / Logghe, Karl / Schiettecatte, Eva / Vermeersch, Hubert / Huvenne, Wouter / De Waele, Kathleen / Van Beveren, Hanne / Van Dorpe, Jo / Creytens, David / De Schepper, Jean

International journal of pediatric endocrinology

2020 Volume 2020, Page(s) 14

Abstract: Case presentation: We report for the first time a synchronous papillary and follicular thyroid carcinoma in a 12-year-old girl presenting with a large (5 cm diameter) left thyroid nodule, an increased left and right upper pole technetium tracer uptake ... ...

Abstract	Case presentation: We report for the first time a synchronous papillary and follicular thyroid carcinoma in a 12-year-old girl presenting with a large (5 cm diameter) left thyroid nodule, an increased left and right upper pole technetium tracer uptake at scintigraphy and hyperthyroidism. The uptake at the right lobe was explained by the crossing of the left nodule to the right site of the neck at Computed Tomography (CT) scanning. Background: Although thyroid nodules are less common in children than in adults, there is more vigilance required in children because of the higher risk of malignancy. According to literature, about 5% of the thyroid nodules in adults are malignant versus 20-26% in children. The characteristics of 9 other pediatric cases with a differentiated thyroid carcinoma presenting with a toxic nodule, which have been reported during the last 20 years, are summarized. A nodular size of more than 3.5 cm and female predominance was a common finding. Conclusions: The presence of hyperthyroidism in association with a hyperfunctioning thyroid nodule does not rule out thyroid cancer and warrants careful evaluation, even in the absence of cervical lymph node invasion.
Language	English
Publishing date	2020-07-21
Publishing country	England
Document type	Case Reports
ZDB-ID	2528691-2
ISSN	1687-9856 ; 1687-9848
ISSN (online)	1687-9856
ISSN	1687-9848
DOI	10.1186/s13633-020-00084-4
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Article ; Online: Efficacy and safety of setmelanotide, an MC4R agonist, in individuals with severe obesity due to LEPR or POMC deficiency: single-arm, open-label, multicentre, phase 3 trials.

Clément, Karine / van den Akker, Erica / Argente, Jesús / Bahm, Allison / Chung, Wendy K / Connors, Hillori / De Waele, Kathleen / Farooqi, I Sadaf / Gonneau-Lejeune, Julie / Gordon, Gregory / Kohlsdorf, Katja / Poitou, Christine / Puder, Lia / Swain, James / Stewart, Murray / Yuan, Guojun / Wabitsch, Martin / Kühnen, Peter

The lancet. Diabetes & endocrinology

2020 Volume 8, Issue 12, Page(s) 960–970

Abstract: Background: The melanocortin 4 receptor (MC4R), a component of the leptin-melanocortin pathway, plays a part in bodyweight regulation. Severe early-onset obesity can be caused by biallelic variants in genes that affect the MC4R pathway. We report the ... ...

Abstract	Background: The melanocortin 4 receptor (MC4R), a component of the leptin-melanocortin pathway, plays a part in bodyweight regulation. Severe early-onset obesity can be caused by biallelic variants in genes that affect the MC4R pathway. We report the results from trials of the MC4R agonist setmelanotide in individuals with severe obesity due to either pro-opiomelanocortin (POMC) deficiency obesity or leptin receptor (LEPR) deficiency obesity. Methods: These single-arm, open-label, multicentre, phase 3 trials were done in ten hospitals across Canada, the USA, Belgium, France, Germany, the Netherlands, and the UK. Participants aged 6 years or older with POMC or LEPR deficiency obesity received open-label setmelanotide for 12 weeks. Participants with at least 5 kg weight loss (or ≥5% if weighing <100 kg at baseline) entered an 8-week placebo-controlled withdrawal sequence (including 4 weeks each of blinded setmelanotide and placebo treatment) followed by 32 additional weeks of open-label treatment. The primary endpoint, which was assessed in participants who received at least one dose of study medication and had a baseline assessment (full analysis set), was the proportion of participants with at least 10% weight loss compared with baseline at approximately 1 year. A key secondary endpoint was mean percentage change in the most hunger score of the 11-point Likert-type scale at approximately 1 year on the therapeutic dose, which was assessed in a subset of participants aged 12 years or older in the full analysis set who demonstrated at least 5 kg weight loss (or ≥5% in paediatric participants if baseline bodyweight was <100 kg) over the 12-week open-label treatment phase and subsequently proceeded into the placebo-controlled withdrawal sequence, regardless of later disposition. These studies are registered with ClinicalTrials.gov, NCT02896192 and NCT03287960. Findings: Between Feb 14, 2017, and Sept 7, 2018, ten participants were enrolled in the POMC trial and 11 participants were enrolled in the LEPR trial, and included in the full analysis and safety sets. Eight (80%) participants in the POMC trial and five (45%) participants in the LEPR trial achieved at least 10% weight loss at approximately 1 year. The mean percentage change in the most hunger score was -27·1% (n=7; 90% CI -40·6 to -15·0; p=0·0005) in the POMC trial and -43·7% (n=7; -54·8 to -29·1; p<0·0001) in the LEPR trial. The most common adverse events were injection site reaction and hyperpigmentation, which were reported in all ten participants in the POMC trial; nausea was reported in five participants and vomiting in three participants. In the LEPR trial, the most commonly reported treatment-related adverse events were injection site reaction in all 11 participants, skin disorders in five participants, and nausea in four participants. No serious treatment-related adverse events occurred in both trials. Interpretation: Our results support setmelanotide for the treatment of obesity and hyperphagia caused by POMC or LEPR deficiency. Funding: Rhythm Pharmaceuticals.
MeSH term(s)	Adolescent ; Adrenal Insufficiency/complications ; Adult ; Anti-Obesity Agents/therapeutic use ; Child ; Double-Blind Method ; Female ; Follow-Up Studies ; Humans ; Male ; Obesity/complications ; Obesity/drug therapy ; Obesity/etiology ; Obesity/pathology ; Pro-Opiomelanocortin/deficiency ; Prognosis ; Receptor, Melanocortin, Type 4/agonists ; Receptors, Leptin/deficiency ; Young Adult ; alpha-MSH/analogs & derivatives ; alpha-MSH/therapeutic use
Chemical Substances	Anti-Obesity Agents ; MC4R protein, human ; Receptor, Melanocortin, Type 4 ; Receptors, Leptin ; setmelanotide ; alpha-MSH (581-05-5) ; Pro-Opiomelanocortin (66796-54-1)
Language	English
Publishing date	2020-10-30
Publishing country	England
Document type	Clinical Trial, Phase III ; Journal Article ; Multicenter Study ; Research Support, Non-U.S. Gov't
ISSN	2213-8595
ISSN (online)	2213-8595
DOI	10.1016/S2213-8587(20)30364-8
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Article: Consensus Recommendations for the Diagnosis and Management of X-Linked Hypophosphatemia in Belgium.

Laurent, Michaël R / De Schepper, Jean / Trouet, Dominique / Godefroid, Nathalie / Boros, Emese / Heinrichs, Claudine / Bravenboer, Bert / Velkeniers, Brigitte / Lammens, Johan / Harvengt, Pol / Cavalier, Etienne / Kaux, Jean-François / Lombet, Jacques / De Waele, Kathleen / Verroken, Charlotte / van Hoeck, Koenraad / Mortier, Geert R / Levtchenko, Elena / Vande Walle, Johan

Frontiers in endocrinology

2021 Volume 12, Page(s) 641543

Abstract: X-linked hypophosphatemia (XLH) is the most common genetic form of hypophosphatemic rickets and osteomalacia. In this disease, mutations in ... ...

Abstract	X-linked hypophosphatemia (XLH) is the most common genetic form of hypophosphatemic rickets and osteomalacia. In this disease, mutations in the
MeSH term(s)	Alkaline Phosphatase/metabolism ; Antibodies, Monoclonal, Humanized/administration & dosage ; Belgium ; Consensus ; Familial Hypophosphatemic Rickets/complications ; Familial Hypophosphatemic Rickets/diagnosis ; Familial Hypophosphatemic Rickets/genetics ; Familial Hypophosphatemic Rickets/therapy ; Fibroblast Growth Factor-23/metabolism ; Humans ; Hypophosphatemia/complications ; Hypophosphatemia/genetics ; Interdisciplinary Communication ; Mutation ; Osteomalacia/complications ; Osteomalacia/genetics ; PHEX Phosphate Regulating Neutral Endopeptidase/genetics ; Severity of Illness Index ; Societies, Medical/organization & administration ; Treatment Outcome ; Vitamin D
Chemical Substances	Antibodies, Monoclonal, Humanized ; FGF23 protein, human ; Vitamin D (1406-16-2) ; Fibroblast Growth Factor-23 (7Q7P4S7RRE) ; Alkaline Phosphatase (EC 3.1.3.1) ; PHEX Phosphate Regulating Neutral Endopeptidase (EC 3.4.24.-) ; PHEX protein, human (EC 3.4.24.-) ; burosumab (G9WJT6RD29)
Language	English
Publishing date	2021-03-19
Publishing country	Switzerland
Document type	Journal Article ; Research Support, Non-U.S. Gov't ; Review
ZDB-ID	2592084-4
ISSN	1664-2392
ISSN	1664-2392
DOI	10.3389/fendo.2021.641543
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Article: Corrigendum: Consensus Recommendations for the Diagnosis and Management of X-Linked Hypophosphatemia in Belgium.

Frontiers in endocrinology

2021 Volume 12, Page(s) 686401

Abstract: This corrects the article DOI: 10.3389/fendo.2021.641543.]. ...

Abstract	[This corrects the article DOI: 10.3389/fendo.2021.641543.].
Language	English
Publishing date	2021-05-25
Publishing country	Switzerland
Document type	Published Erratum
ZDB-ID	2592084-4
ISSN	1664-2392
ISSN	1664-2392
DOI	10.3389/fendo.2021.686401
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Article ; Online: In vitro and in vivo effect of acylated and unacylated ghrelin on neonatal glucose homeostasis.

Ni, Hehong / De Waele, Kathleen / Walia, Pallavi / Chanoine, Jean-Pierre

Pediatric research

2010 Volume 67, Issue 6, Page(s) 609–613

Abstract: Maintenance of normal glucose homeostasis is crucial for survival during the perinatal period. Acylated ghrelin (AG) but not unacylated ghrelin (UAG) inhibits insulin release from pancreatic islets in adult rats. Circulating AG concentrations are low in ... ...

Abstract	Maintenance of normal glucose homeostasis is crucial for survival during the perinatal period. Acylated ghrelin (AG) but not unacylated ghrelin (UAG) inhibits insulin release from pancreatic islets in adult rats. Circulating AG concentrations are low in the fetus and progressively increase in the postnatal period. We tested the hypothesis that AG has insulinostatic effects in vitro and in vivo during the perinatal period. In vitro, AG (10(-10)-10(-8) M) caused a 25-53% decrease in insulin secretion by islets from 5-d-old rat pups under normo- and hyperglycemic conditions, an effect that was mediated through the growth hormone secretagogue receptor (GHSR- 1a). Ghrelin (1-5) amide, [Dap3]-octanoyl, a pentapeptide that is resistant to deacylation and binds the GHSR-1a, had similar effects at 10(-8) M. In vivo, AG, UAG, or GHRP-6 [D-Lys3], a GHSR-1a antagonist, did not affect insulin or glucagon concentrations during the first 3 h of life. In 6-d-old pups, AG, UAG, or ghrelin (1-5) amide, [Dap3]-octanoyl did not affect glucose-induced insulin or C-peptide concentrations. In summary, AG has insulinostatic effects in vitro as early as during the perinatal period. These effects could not be confirmed in vivo, possibly due to the short half-life of AG in rat neonates.
MeSH term(s)	Acylation ; Animals ; Animals, Newborn ; Blood Glucose/metabolism ; C-Peptide/metabolism ; Cesarean Section ; Dose-Response Relationship, Drug ; Female ; Ghrelin/metabolism ; Glucose/metabolism ; Homeostasis ; Hyperglycemia/metabolism ; Insulin/blood ; Insulin/metabolism ; Islets of Langerhans/drug effects ; Islets of Langerhans/metabolism ; Male ; Oligopeptides/pharmacology ; Protein Processing, Post-Translational ; Rats ; Rats, Sprague-Dawley ; Receptors, Ghrelin/metabolism ; Tissue Culture Techniques
Chemical Substances	Blood Glucose ; C-Peptide ; GHRP-6, Lys(3)- ; Ghrelin ; Insulin ; Oligopeptides ; Receptors, Ghrelin ; Glucose (IY9XDZ35W2)
Language	English
Publishing date	2010-06
Publishing country	United States
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	4411-8
ISSN	1530-0447 ; 0031-3998
ISSN (online)	1530-0447
ISSN	0031-3998
DOI	10.1203/PDR.0b013e3181da463a
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Zs.A 564: Show issues			Location: Je nach Verfügbarkeit (siehe Angabe bei Bestand) bis Jg. 1994: Bestellungen von Artikeln über das Online-Bestellformular Jg. 1995 - 2021: Lesesall (1.OG) ab Jg. 2022: Lesesaal (EG)
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Article ; Online: Evaluation of cardiopulmonary exercise testing, heart function, and quality of life in children after allogenic hematopoietic stem cell transplantation.

Vandekerckhove, Kristof / De Waele, Kathleen / Minne, Aurelie / Coomans, Ilse / De Groote, Katya / Panzer, Joseph / Dhooge, Catherine / Bordon, Victoria / De Wolf, Daniel / Boone, Jan

Pediatric blood & cancer

2018 Volume 66, Issue 1, Page(s) e27499

Abstract: Background: Physical fitness is an important determinant of quality of life (QOL) after hematopoietic stem cell transplantation. Cardiac function can influence exercise performance. The aim of this study was to assess these factors and their ... ...

Abstract	Background: Physical fitness is an important determinant of quality of life (QOL) after hematopoietic stem cell transplantation. Cardiac function can influence exercise performance. The aim of this study was to assess these factors and their interrelationship. Procedure: Children underwent cardiopulmonary exercise testing (CPET) at least 1 year after hematopoietic stem cell transplantation (HSCT) and were compared with healthy controls. Systolic and diastolic heart function and left ventricle (LV) wall dimensions were measured. Health-related QOL (HR-QOL) was evaluated using PedsQL questionnaires. Results: Forty-three patients performed CPET (26 boys, 13.6 ± 3.4 years, weight 45.5 ± 13.3 kg, length 152.9 ± 17.5 cm, body surface area 1.35 ± 0.28). HSCT patients had lower maximal oxygen consumption (VO2peak/kg, 34.7 ± 8.4 vs 46.3 ± 7.1 mL/kg/min, P < 0.001), shorter exercise duration (9.1 ± 2.5 vs 12.9 ± 2.6 min, P < 0.001), and lower maximal load (%Ppeak 70.8 ± 19.7 vs 102.4% ± 15.9%, P < 0.001). Echocardiography demonstrated decreased interventricular septal wall thickness (interventricular septum in diastole [IVSd] Z-value -0.64 ± 0.69, P < 0.001), and more systolic (11% of patients) and diastolic dysfunction (high E/E' Z-value 1.06 ± 1.13, P < 0.001). LV dilatation correlates with VO2max/kg (r = -0.364, P = 0.017). HR-QOL showed lower overall and emotional functioning scores (respectively, P = 0.016 and P = 0.001). Patients after anthracycline therapy have the lowest maximal exercise performance, but have no difference in QOL. Diminished exercise performance is not encountered as a QOL limitation. Total body irradiation influences the domain of psychosocial functioning. Conclusions: LV (systolic and diastolic) and right ventricle dysfunctions justify the need for thorough cardiac follow-up in children after HSCT. Lower physical fitness levels and lower HR-QOL emphasize the importance of CPET and fitness programs.
MeSH term(s)	Adolescent ; Cardiorespiratory Fitness ; Case-Control Studies ; Child ; Child, Preschool ; Echocardiography/methods ; Exercise Test/methods ; Female ; Follow-Up Studies ; Hematopoietic Stem Cell Transplantation/methods ; Humans ; Infant ; Male ; Neoplasms/pathology ; Neoplasms/therapy ; Prognosis ; Prospective Studies ; Quality of Life ; Transplantation, Homologous ; Ventricular Function, Left
Language	English
Publishing date	2018-10-14
Publishing country	United States
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	2131448-2
ISSN	1545-5017 ; 1545-5009
ISSN (online)	1545-5017
ISSN	1545-5009
DOI	10.1002/pbc.27499
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