Growth hormone therapy and respiratory disorders: Long-term follow-up in PWS children

Jenny Berini, Valeria Spica Russotto, Paolo Castelnuovo, Stefania Di Candia, Luigi Gargantini, Graziano Grugni, Lorenzo Iughetti, Luigi Nespoli, Luana Nosetti, Giovanni Padoan, Alba Pilotta, Giuliana Trifiro, Giuseppe Chiumello, Alessandro Salvatoni

Research output: Contribution to journalArticle

Abstract

Context: Adenotonsillar tissue hypertrophy and obstructive sleep apnea have been reported during short-term GH treatment in children with Prader-Willi syndrome (PWS). Objective: We conducted an observational study to evaluate the effects of long-term GH therapy on sleep-disordered breathing and adenotonsillar hypertrophy in children with PWS. Design: This was a longitudinal observational study. PatientsandMethods:Weevaluated 75 children with genetically confirmedPWS,ofwhom50 fulfilled the criteria and were admitted to our study. The patients were evaluated before treatment (t0), after 6 weeks (t1), after 6 months (t2), after 12 months (t3), and yearly (t4-t6) thereafter, for up to 4 years of GH therapy. The central apnea index, obstructive apnea hypopnea index (OAHI), respiratory disturbance index, and minimal blood oxygen saturation were evaluated overnight using polysomnography. We evaluated the adenotonsillar size using a flexible fiberoptic endoscope. Results: The percentage of patients with an OAHI of 1 increased from 3 to 22, 36, and 38 at t1, t4, and t6, respectively (2 12.2; P .05). We observed a decrease in the respiratory disturbance indexfrom1.4 (t0) to 0.8 (t3) (P.05)andthe centralapneaindexfrom1.2 (t0) to 0.1 (t4) (P.0001). We had to temporarily suspend treatment for 3 patients at t1, t4, and t5 because of severe obstructive sleep apnea. The percentage of patients with severe adenotonsillar hypertrophy was significantly higher at t4 and t5 than at t0. The OAHI directly correlated with the adenoid size (adjusted for age) (P .01) but not with the tonsil size and IGF-1 levels. Conclusion: Long-termGHtreatment in patients withPWSis safe; however,werecommend annual polysomnography and adenotonsillar evaluation.

Original languageEnglish
JournalJournal of Clinical Endocrinology and Metabolism
Volume98
Issue number9
DOIs
Publication statusPublished - Sep 2013

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Prader-Willi Syndrome
Respiratory Therapy
Growth Hormone
Apnea
Hypertrophy
Polysomnography
Obstructive Sleep Apnea
Endoscopy
Observational Studies
Insulin-Like Growth Factor I
Central Sleep Apnea
Blood
Therapeutics
Adenoids
Tissue
Oxygen
Endoscopes
Palatine Tonsil
Sleep Apnea Syndromes
Longitudinal Studies

ASJC Scopus subject areas

  • Biochemistry
  • Clinical Biochemistry
  • Endocrinology
  • Biochemistry, medical
  • Endocrinology, Diabetes and Metabolism

Cite this

Growth hormone therapy and respiratory disorders : Long-term follow-up in PWS children. / Berini, Jenny; Spica Russotto, Valeria; Castelnuovo, Paolo; Di Candia, Stefania; Gargantini, Luigi; Grugni, Graziano; Iughetti, Lorenzo; Nespoli, Luigi; Nosetti, Luana; Padoan, Giovanni; Pilotta, Alba; Trifiro, Giuliana; Chiumello, Giuseppe; Salvatoni, Alessandro.

In: Journal of Clinical Endocrinology and Metabolism, Vol. 98, No. 9, 09.2013.

Research output: Contribution to journalArticle

Berini, J, Spica Russotto, V, Castelnuovo, P, Di Candia, S, Gargantini, L, Grugni, G, Iughetti, L, Nespoli, L, Nosetti, L, Padoan, G, Pilotta, A, Trifiro, G, Chiumello, G & Salvatoni, A 2013, 'Growth hormone therapy and respiratory disorders: Long-term follow-up in PWS children', Journal of Clinical Endocrinology and Metabolism, vol. 98, no. 9. https://doi.org/10.1210/jc.2013-1831
Berini, Jenny ; Spica Russotto, Valeria ; Castelnuovo, Paolo ; Di Candia, Stefania ; Gargantini, Luigi ; Grugni, Graziano ; Iughetti, Lorenzo ; Nespoli, Luigi ; Nosetti, Luana ; Padoan, Giovanni ; Pilotta, Alba ; Trifiro, Giuliana ; Chiumello, Giuseppe ; Salvatoni, Alessandro. / Growth hormone therapy and respiratory disorders : Long-term follow-up in PWS children. In: Journal of Clinical Endocrinology and Metabolism. 2013 ; Vol. 98, No. 9.
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abstract = "Context: Adenotonsillar tissue hypertrophy and obstructive sleep apnea have been reported during short-term GH treatment in children with Prader-Willi syndrome (PWS). Objective: We conducted an observational study to evaluate the effects of long-term GH therapy on sleep-disordered breathing and adenotonsillar hypertrophy in children with PWS. Design: This was a longitudinal observational study. PatientsandMethods:Weevaluated 75 children with genetically confirmedPWS,ofwhom50 fulfilled the criteria and were admitted to our study. The patients were evaluated before treatment (t0), after 6 weeks (t1), after 6 months (t2), after 12 months (t3), and yearly (t4-t6) thereafter, for up to 4 years of GH therapy. The central apnea index, obstructive apnea hypopnea index (OAHI), respiratory disturbance index, and minimal blood oxygen saturation were evaluated overnight using polysomnography. We evaluated the adenotonsillar size using a flexible fiberoptic endoscope. Results: The percentage of patients with an OAHI of 1 increased from 3 to 22, 36, and 38 at t1, t4, and t6, respectively (2 12.2; P .05). We observed a decrease in the respiratory disturbance indexfrom1.4 (t0) to 0.8 (t3) (P.05)andthe centralapneaindexfrom1.2 (t0) to 0.1 (t4) (P.0001). We had to temporarily suspend treatment for 3 patients at t1, t4, and t5 because of severe obstructive sleep apnea. The percentage of patients with severe adenotonsillar hypertrophy was significantly higher at t4 and t5 than at t0. The OAHI directly correlated with the adenoid size (adjusted for age) (P .01) but not with the tonsil size and IGF-1 levels. Conclusion: Long-termGHtreatment in patients withPWSis safe; however,werecommend annual polysomnography and adenotonsillar evaluation.",
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AU - Berini, Jenny

AU - Spica Russotto, Valeria

AU - Castelnuovo, Paolo

AU - Di Candia, Stefania

AU - Gargantini, Luigi

AU - Grugni, Graziano

AU - Iughetti, Lorenzo

AU - Nespoli, Luigi

AU - Nosetti, Luana

AU - Padoan, Giovanni

AU - Pilotta, Alba

AU - Trifiro, Giuliana

AU - Chiumello, Giuseppe

AU - Salvatoni, Alessandro

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N2 - Context: Adenotonsillar tissue hypertrophy and obstructive sleep apnea have been reported during short-term GH treatment in children with Prader-Willi syndrome (PWS). Objective: We conducted an observational study to evaluate the effects of long-term GH therapy on sleep-disordered breathing and adenotonsillar hypertrophy in children with PWS. Design: This was a longitudinal observational study. PatientsandMethods:Weevaluated 75 children with genetically confirmedPWS,ofwhom50 fulfilled the criteria and were admitted to our study. The patients were evaluated before treatment (t0), after 6 weeks (t1), after 6 months (t2), after 12 months (t3), and yearly (t4-t6) thereafter, for up to 4 years of GH therapy. The central apnea index, obstructive apnea hypopnea index (OAHI), respiratory disturbance index, and minimal blood oxygen saturation were evaluated overnight using polysomnography. We evaluated the adenotonsillar size using a flexible fiberoptic endoscope. Results: The percentage of patients with an OAHI of 1 increased from 3 to 22, 36, and 38 at t1, t4, and t6, respectively (2 12.2; P .05). We observed a decrease in the respiratory disturbance indexfrom1.4 (t0) to 0.8 (t3) (P.05)andthe centralapneaindexfrom1.2 (t0) to 0.1 (t4) (P.0001). We had to temporarily suspend treatment for 3 patients at t1, t4, and t5 because of severe obstructive sleep apnea. The percentage of patients with severe adenotonsillar hypertrophy was significantly higher at t4 and t5 than at t0. The OAHI directly correlated with the adenoid size (adjusted for age) (P .01) but not with the tonsil size and IGF-1 levels. Conclusion: Long-termGHtreatment in patients withPWSis safe; however,werecommend annual polysomnography and adenotonsillar evaluation.

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