The Impact of Growth Hormone/Insulin-Like Growth Factor-I Axis and Nocturnal Breathing Disorders on Cardiovascular Features of Adult Patients with Prader-Willi Syndrome

Marzullo, Paolo; Marcassa, Claudio; Campini, Riccardo; Eleuteri, Ermanno; Minocci, Alessandro; Priano, Lorenzo; Temporelli, Pier Luigi; Sartório, Alessandro; Vettor, Roberto; Liuzzi, Antonio; Grugni, Graziano

doi:10.1210/jc.2005-0829

Cited by 46 publications

(51 citation statements)

References 43 publications

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“…Twelve months of GH treatment reduced body fat significantly, but we were not able to demonstrate a significant reduction in IL-6, TNF-α or HCRP levels neither after 6 months nor after 1 year. The elevated HCRP in our cohort is not in accordance with the normal level found by Marzullo et al [17], although the study groups otherwise seemed to be similar. The reason for these discordant results is not clear at present.…”

Section: Discussioncontrasting

confidence: 96%

“…As IL-6 and TNF-α are synthesised in adipose tissue and induce HCRP production from the liver [4] the changes in body composition seen during GH therapy could be the reason for the fall in inflammatory markers. In adult PWS body composition and metabolic abnormalities (decreased total lean body mass and subnormal IGF-I levels) resemble a state of partial GHD [9, 17]. In the current study IL-6 and HCRP were clearly increased, while TNF-α was normal.…”

Section: Discussionsupporting

confidence: 47%

“…On the one hand it has been found, that adults with PWS die of diseases related to obesity [10,11,12], but on the other hand despite the obesity, the metabolic profile is usually favourable [9, 17]. The metabolic syndrome is associated with central adiposity and insulin resistance [24], and elevated inflammatory markers have been demonstrated in obesity [1–3, 5, 6].…”

Section: Discussionmentioning

confidence: 99%

“…Except for a recent study, in which high sensitive CRP (HCRP) levels in PWS adults were found to be normal [17] inflammatory markers have not previously been investigated in PWS. Because of the abnormal body composition, the partial GHD and hypogonadism in PWS, we analysed IL-6, TNF-α and HCRP before and during GH treatment in a group of young adult PWS patients.…”

Section: Introductionmentioning

confidence: 99%

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Inflammatory Markers in Adults with Prader-Willi Syndrome before and during 12 Months Growth Hormone Treatment

Höybye

2006

Horm Res Paediatr

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Background: In Prader-Willi syndrome (PWS) obesity and partial growth hormone (GH) deficiency are frequently observed. The risks of cardiovascular diseases and early death are increased. We examined inflammatory markers in adult PWS, before and during 12 months of GH treatment. Method: Twelve PWS adults, median age 23.5 years (17–37) and median BMI 33.8 kg/m² (21.2–50.4), participated. Serum interleukin-6, tumour necrosis factor α, high sensitive protein C-reactive protein (HCRP), cholesterol, triglycerides, leptin, adiponectin, glucose, insulin, insulin-like growth factor I and body composition were measured at baseline and after 6 and 12 months of GH treatment. Results: Median and range at baseline for interleukin-6 was 9.87 ng/l (1.76–10.72), for tumour necrosis factor α 2.39 ng/l (1.00–3.26) and for HCRP 7.64 mg/l (0.41–41.1) (normal values < 5 ng/l, < 8 ng/l and<5 mg/l, respectively). At baseline correlations between inflammatory markers and age, anthropometry, body composition and the metabolic parameters were non-significant; only positive associations were found between tumour necrosis factor α and body weight (r = 0.617, p = 0.033) and between HCRP and BMI (r = 0.594, p = 0.041). GH treatment non-significantly decreased the levels of the inflammatory markers. Conclusion: In this pilot study, levels of interleukin-6 and HCRP were increased, and GH intervention did not significantly reduce the levels. Chronic inflammation might contribute to the increased cardiovascular morbidity and mortality in PWS.

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Section: Discussioncontrasting

confidence: 96%

Section: Discussionsupporting

confidence: 47%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Inflammatory Markers in Adults with Prader-Willi Syndrome before and during 12 Months Growth Hormone Treatment

Höybye

2006

Horm Res Paediatr

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“…Importantly, patients with GHD following cure of acromegaly or Cushing's disease are more prone to develop hypertension, diabetes mellitus and stroke than those with GHD from any other cause (107). The influence of obesity is equally important as seen in studies on adults with Prader-Willi Syndrome, a monogenic disorder associated with morbid obesity and GHD (108). In populations with classical GHD, GH therapy produces positive cardiac effects depending on patient's medical history, multiplicity of pituitary deficiencies and comorbidities.…”

Section: Uptodate On Gh/igf-1 Axis Actionsmentioning

confidence: 99%

Uptodate on GH/IGF-1 axis actions

Baldelli¹,

Rota²,

Aimaretti³

et al. 2013

REM

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SummaryIn December 2010, a workshop was held at "Regina Elena" National Cancer Institute in Rome, Italy, to develop an update on GH/IGF-1 system. The workshop was supported by NoGeRo group and was sponsored by SIE Lazio (Società Italiana di Endocrinologia-Lazio Regional Section) and AME Lazio (Associazione Medici Endocrinologi-Lazio Regional Section). Invited participants included italian endocrinologists skilled in the field of GH and IGF-I system both in basic and clinical research. In the following two years an extensive review has been structured summarizing the most important points achieved in the discussions during the workshop.

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The Italian National Survey for Prader–Willi syndrome: An epidemiologic study

Grugni

Crinò

Bosio

et al. 2008

American J of Med Genetics Pt A

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Twenty-five medical centers and the Prader-Willi Syndrome (PWS) Association collaborated on a study which attempted to identify all people with genetically confirmed diagnosis of PWS living in Italy. Investigators of the participating centers contacted PWS subjects and/or their family, filled in a specially developed form with the required data and forwarded this information by email. The study identified 425 subjects (209 males and 216 females, between the ages of 0.4-46.7). Two hundred thirty-eight patients had del15, 104 had UPD15, 4 demonstrated a translocation affecting chromosome 15 and 79 showed a positive methylation test. There were fewer subjects found over the age of 35, probably due to the low rate of identification of older PWS patients as well as the high mortality rate. There were a greater number of male children and adolescents with PWS whilst, amongst adults, there were more females. As expected, the majority of subjects with PWS were obese, especially in adult life. Nevertheless, it is noteworthy that 26% of patients aged between 6 and 17 were normal weight. A total of 212 subjects had received GH treatment, of which 141 were still receiving therapy, while the remaining 71 had stopped. In children and adolescents (233 cases), 89 subjects had never undergone GH therapy. Eighteen PWS patients had died in the past 20 years. Obesity-related cardiovascular and respiratory diseases were the cause of death, both during childhood and after 18 years of age. Three children died suddenly whilst undergoing GH therapy. Respiratory infection and cardiac illness were the causes of death in two cases. There was no definitive cause of death found in the third case. Overall, there was no increase in number of deaths during GH treatment, suggesting that GH administration in patients with PWS, as a group, does not increase the risk of death. ß

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The Impact of Growth Hormone/Insulin-Like Growth Factor-I Axis and Nocturnal Breathing Disorders on Cardiovascular Features of Adult Patients with Prader-Willi Syndrome

Cited by 46 publications

References 43 publications

Inflammatory Markers in Adults with Prader-Willi Syndrome before and during 12 Months Growth Hormone Treatment

Inflammatory Markers in Adults with Prader-Willi Syndrome before and during 12 Months Growth Hormone Treatment

Uptodate on GH/IGF-1 axis actions

The Italian National Survey for Prader–Willi syndrome: An epidemiologic study

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