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Pituitary function and the response to GH therapy in patients with Langerhans Cell Histiocytosis: Analysis of the KIMS Database

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Touraine, P, Sagna, Y, Mattson, AF, Burman, P, Van Beek, AP, Carlsson, MO, Aydin, F, Feldt-Rasmussen, U & Camacho-Hübner, C 2022, 'Pituitary function and the response to GH therapy in patients with Langerhans Cell Histiocytosis: Analysis of the KIMS Database', European Journal of Endocrinology, bind 187, nr. 3, s. 373-381. https://doi.org/10.1530/EJE-22-0160

APA

Touraine, P., Sagna, Y., Mattson, A. F., Burman, P., Van Beek, A. P., Carlsson, M. O., Aydin, F., Feldt-Rasmussen, U., & Camacho-Hübner, C. (2022). Pituitary function and the response to GH therapy in patients with Langerhans Cell Histiocytosis: Analysis of the KIMS Database. European Journal of Endocrinology, 187(3), 373-381. https://doi.org/10.1530/EJE-22-0160

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MLA

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Author

Touraine, Philippe ; Sagna, Yempabou ; Mattson, Anders F ; Burman, Pia ; Van Beek, André P ; Carlsson, Martin Ove ; Aydin, Ferah ; Feldt-Rasmussen, Ulla ; Camacho-Hübner, Cecilia. / Pituitary function and the response to GH therapy in patients with Langerhans Cell Histiocytosis : Analysis of the KIMS Database. I: European Journal of Endocrinology. 2022 ; Bind 187, Nr. 3. s. 373-381.

Bibtex

@article{5b21b13ef1b549bfa210c7d4e4f7be7e,
title = "Pituitary function and the response to GH therapy in patients with Langerhans Cell Histiocytosis: Analysis of the KIMS Database",
abstract = "Objective: To analyze the effectiveness and safety of growth hormone (GH) replacement treatment in adult patients with Langerhans cell histiocytosis (LCH) and GH deficiency (GHD) enrolled in KIMS (Pfizer International Metabolic Database).Patients and methods: Patients with LCH and GHD were studied at baseline and some of them after 1 year of GH treatment. The effectiveness of GH is presented as change after 1 year of treatment (mean, 95% CI). The LCH population was compared to two other groups of patients enrolled in KIMS, granulomatous and lymphocytic hypophysitis.Results: At baseline, 81 adults with LCH (27 with childhood onset, 56% females), mean age at GHD onset of 29 (15) years were studied. Diabetes insipidus was diagnosed in 86% of patients. Analysis of 1 year of GH treatment was possible in 37 patients. One-year cross-sectional values for the GH dose were 0.39 (s.d.± 0.21) mg and -0.5 (-1.2 to 0.2) for insulin-like growth factor-1 s.d. Total cholesterol decreased 0.9 (-1.5 to -0.3 (mmol/L); P < 0.05); AGHDA-QoL-score (n = 20) was improved by 2.8 points (-5.6 to 0.0; P < 0.05), while mean BMI increased 0.6 ± 3 kg/m2 (95% CI: -0.2 to 1.4). All these effects did not differ from the two other groups after adjusting for age, gender, and baseline values. In 20 of 77 patients included in the safety analysis, 36 serious adverse events were reported during 435 patient-years (82.8/1000); no new safety signals were reported.Conclusion: After 1 year of GH treatment in patients with LCH, metabolic variables and quality of life improved, with no new safety signals.",
keywords = "Adult, Child, Cross-Sectional Studies, Dwarfism, Pituitary, Female, Histiocytosis, Langerhans-Cell/drug therapy, Hormone Replacement Therapy, Human Growth Hormone/therapeutic use, Humans, Hypopituitarism/drug therapy, Male, Quality of Life",
author = "Philippe Touraine and Yempabou Sagna and Mattson, {Anders F} and Pia Burman and {Van Beek}, {Andr{\'e} P} and Carlsson, {Martin Ove} and Ferah Aydin and Ulla Feldt-Rasmussen and Cecilia Camacho-H{\"u}bner",
year = "2022",
month = sep,
day = "1",
doi = "10.1530/EJE-22-0160",
language = "English",
volume = "187",
pages = "373--381",
journal = "European Journal of Endocrinology",
issn = "0804-4643",
publisher = "BioScientifica Ltd.",
number = "3",

}

RIS

TY - JOUR

T1 - Pituitary function and the response to GH therapy in patients with Langerhans Cell Histiocytosis

T2 - Analysis of the KIMS Database

AU - Touraine, Philippe

AU - Sagna, Yempabou

AU - Mattson, Anders F

AU - Burman, Pia

AU - Van Beek, André P

AU - Carlsson, Martin Ove

AU - Aydin, Ferah

AU - Feldt-Rasmussen, Ulla

AU - Camacho-Hübner, Cecilia

PY - 2022/9/1

Y1 - 2022/9/1

N2 - Objective: To analyze the effectiveness and safety of growth hormone (GH) replacement treatment in adult patients with Langerhans cell histiocytosis (LCH) and GH deficiency (GHD) enrolled in KIMS (Pfizer International Metabolic Database).Patients and methods: Patients with LCH and GHD were studied at baseline and some of them after 1 year of GH treatment. The effectiveness of GH is presented as change after 1 year of treatment (mean, 95% CI). The LCH population was compared to two other groups of patients enrolled in KIMS, granulomatous and lymphocytic hypophysitis.Results: At baseline, 81 adults with LCH (27 with childhood onset, 56% females), mean age at GHD onset of 29 (15) years were studied. Diabetes insipidus was diagnosed in 86% of patients. Analysis of 1 year of GH treatment was possible in 37 patients. One-year cross-sectional values for the GH dose were 0.39 (s.d.± 0.21) mg and -0.5 (-1.2 to 0.2) for insulin-like growth factor-1 s.d. Total cholesterol decreased 0.9 (-1.5 to -0.3 (mmol/L); P < 0.05); AGHDA-QoL-score (n = 20) was improved by 2.8 points (-5.6 to 0.0; P < 0.05), while mean BMI increased 0.6 ± 3 kg/m2 (95% CI: -0.2 to 1.4). All these effects did not differ from the two other groups after adjusting for age, gender, and baseline values. In 20 of 77 patients included in the safety analysis, 36 serious adverse events were reported during 435 patient-years (82.8/1000); no new safety signals were reported.Conclusion: After 1 year of GH treatment in patients with LCH, metabolic variables and quality of life improved, with no new safety signals.

AB - Objective: To analyze the effectiveness and safety of growth hormone (GH) replacement treatment in adult patients with Langerhans cell histiocytosis (LCH) and GH deficiency (GHD) enrolled in KIMS (Pfizer International Metabolic Database).Patients and methods: Patients with LCH and GHD were studied at baseline and some of them after 1 year of GH treatment. The effectiveness of GH is presented as change after 1 year of treatment (mean, 95% CI). The LCH population was compared to two other groups of patients enrolled in KIMS, granulomatous and lymphocytic hypophysitis.Results: At baseline, 81 adults with LCH (27 with childhood onset, 56% females), mean age at GHD onset of 29 (15) years were studied. Diabetes insipidus was diagnosed in 86% of patients. Analysis of 1 year of GH treatment was possible in 37 patients. One-year cross-sectional values for the GH dose were 0.39 (s.d.± 0.21) mg and -0.5 (-1.2 to 0.2) for insulin-like growth factor-1 s.d. Total cholesterol decreased 0.9 (-1.5 to -0.3 (mmol/L); P < 0.05); AGHDA-QoL-score (n = 20) was improved by 2.8 points (-5.6 to 0.0; P < 0.05), while mean BMI increased 0.6 ± 3 kg/m2 (95% CI: -0.2 to 1.4). All these effects did not differ from the two other groups after adjusting for age, gender, and baseline values. In 20 of 77 patients included in the safety analysis, 36 serious adverse events were reported during 435 patient-years (82.8/1000); no new safety signals were reported.Conclusion: After 1 year of GH treatment in patients with LCH, metabolic variables and quality of life improved, with no new safety signals.

KW - Adult

KW - Child

KW - Cross-Sectional Studies

KW - Dwarfism, Pituitary

KW - Female

KW - Histiocytosis, Langerhans-Cell/drug therapy

KW - Hormone Replacement Therapy

KW - Human Growth Hormone/therapeutic use

KW - Humans

KW - Hypopituitarism/drug therapy

KW - Male

KW - Quality of Life

UR - http://www.scopus.com/inward/record.url?scp=85135375980&partnerID=8YFLogxK

U2 - 10.1530/EJE-22-0160

DO - 10.1530/EJE-22-0160

M3 - Journal article

C2 - 35830276

VL - 187

SP - 373

EP - 381

JO - European Journal of Endocrinology

JF - European Journal of Endocrinology

SN - 0804-4643

IS - 3

ER -

ID: 79393384