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Incidence of Growth Hormone Deficiency in Pediatric-Onset Langerhans Cell Histiocytosis: Efficacy and Safety of Growth Hormone Treatment

Service d’Hémato-Oncologie Pédiatrique (J.D.), Hopital Trousseau, 75012 Paris, France; Service d’endocrinologie pédiatrique Hopital Necker (M.-A.R., M.P.), 75015 Paris, France; Laboratoire Pharmacia (I.P.), Guyancourt 78200, France; Service d’Hémato-oncologie Pédiatrique (C.T.), CHU de Nantes, Nantes 44000, France; Service de Pédiatrie (F.D.), Institut Curie, 75005, Paris, France; Délégation à la recherche clinique (M.B.), Hopital St. Louis, 75010 Paris, France; Service d’hémato-Oncologie Pédiatrique (A.R.), Hopital Purpan, Toulouse, France; Service d’hémato-Oncologie Pédiatrique (A.D.), Fondation Lenval, Nice, France; Service d’hémato-Oncologie Pédiatrique (F.M.), Hopital Jeanne De Flandres, Lilles, France; Service d’endocrinologie pédiatrique (M.D.), Hopital Lyon-sud, Lyon, France; Service d’endocrinologie pédiatrique (R.B.), Hopital St. Joseph, 75014 Paris, France; Service d’endocrinologie pédiatrique (S.C.), Hopital Trousseau, 75012 Paris, France; and Service de Radiologie (C.G.), Hopital Robert Debré, 75019 Paris, France

Address all correspondence and requests for reprints to: Michel Polak, Service d’endocrinologie Pédiatrique, Hopital Necker, 149 rue de Sèvres, 75015 Paris, France. E-mail: michel.polak{at}nck.ap-hop-paris.fr.

We retrospectively studied 61 patients with GH deficiency (GHD), identified among 589 patients with Langerhans cell histiocytosis (LCH) enrolled in a nationwide survey between 1993 and 2001. Overall, 141 patients in the survey developed diabetes insipidus. The median follow-up of the 61 patients with GHD was 12 yr. The 5- and 10-yr risks of GHD among patients with diabetes insipidus were 34.7 ± 4.5% and 53.7 ± 5.2%, respectively. Growth velocity decreased soon after LCH diagnosis in patients who developed GHD, and anterior pituitary height, estimated by magnetic resonance imaging, was significantly reduced relative to patients who remained free of GHD. GH replacement therapy was administered to 47 of the 61 patients with GHD. Among GH-treated patients, median final height (-0.8 SD) was significantly greater than median height at GHD diagnosis (-1.6 SD) but remained below midparental (target) height. Among patients with pituitary involvement, the number of LCH disease episodes appeared not significantly influenced by GHD or GH administration, suggesting an absence of deleterious effect of GH therapy on LCH disease activity.

The French Langerhans Cell Study Group is composed of N. Brousse, F. Bernard, J. Donadieu, J. F. Emile, T. Genereau, O. Hermine, K. Hoang-Xuan, M. Polak, A. Tazi, and C. Thomas.

These data have already been presented in part at the European Society for Pediatric Endocrinology, Lawson Wilkins Pediatric Endocrine Society, VIth Joint Meeting; Montreal, Canada, 2001.

This work was supported by Grant PHRC 96/AP HP/AOM 96301-96302; Grant PHRC 2001/CHU de Nantes, Grant INSERM/AFM GIS Maladies Rares, a grant from Histiocytose France; and an educational grant from Pharmacia SAS, a branch of Pfizer. We thank Dr. O. de Beco, Dr. C. Pasik, and Mrs. C. Barbara (Pharmacia SAS, a branch of Pfizer) for their support.

Abbreviations: DI, Diabetes insipidus; GHD, GH deficiency; LCH, Langerhans cell histiocytosis; MRI, magnetic resonance imaging; SDS, SD score.

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