BACKGROUND: Children initially diagnosed with isolated GH deficiency (IGHD) have a variable rate to progress to combined pituitary hormone deficiency (CPHD) during follow-up. OBJECTIVE: To evaluate the development of CPHD in a group of childhood-onset IGHD followed at a single tertiary center over a long period of time. PATIENTS AND METHODS: We retrospectively analyzed data from 83 patients initially diagnosed as IGHD with a mean follow-up of 15.2 years. The Kaplan-Meier method and Cox regression analysis was used to estimate the temporal progression and to identify risk factors to development of CPHD over time. RESULTS: From 83 patients initially with IGHD, 37 (45%) developed CPHD after a median time of follow up of 5.4 years (range from 1.2 to 21 years). LH and FSH deficiencies were the most common pituitary hormone (38%) deficiencies developed followed by TSH (31%), ACTH (12%) and ADH deficiency (5%). ADH deficiency (3.1 ± 1 years from GHD diagnosis) presented earlier and ACTH deficiency (9.3 ± 3.5 years) presented later during follow up compared to LH/FSH (8.3 ± 4 years) and TSH (7.5 ± 5.6 years) deficiencies. In a Cox regression model, pituitary stalk abnormalities was the strongest risk factor for the development of CPHD (hazard ratio of 3.28; p = 0.002). CONCLUSION: Our study indicated a high frequency of development of CPHD in patients initially diagnosed as IGHD at childhood. Half of our patients with IGHD developed the second hormone deficiency after 5 years of diagnosis, reinforcing the need for lifelong monitoring of pituitary function in these patients.
BACKGROUND:Children initially diagnosed with isolated GH deficiency (IGHD) have a variable rate to progress to combined pituitary hormone deficiency (CPHD) during follow-up. OBJECTIVE: To evaluate the development of CPHD in a group of childhood-onset IGHD followed at a single tertiary center over a long period of time. PATIENTS AND METHODS: We retrospectively analyzed data from 83 patients initially diagnosed as IGHD with a mean follow-up of 15.2 years. The Kaplan-Meier method and Cox regression analysis was used to estimate the temporal progression and to identify risk factors to development of CPHD over time. RESULTS: From 83 patients initially with IGHD, 37 (45%) developed CPHD after a median time of follow up of 5.4 years (range from 1.2 to 21 years). LH and FSH deficiencies were the most common pituitary hormone (38%) deficiencies developed followed by TSH (31%), ACTH (12%) and ADH deficiency (5%). ADH deficiency (3.1 ± 1 years from GHD diagnosis) presented earlier and ACTH deficiency (9.3 ± 3.5 years) presented later during follow up compared to LH/FSH (8.3 ± 4 years) and TSH (7.5 ± 5.6 years) deficiencies. In a Cox regression model, pituitary stalk abnormalities was the strongest risk factor for the development of CPHD (hazard ratio of 3.28; p = 0.002). CONCLUSION: Our study indicated a high frequency of development of CPHD in patients initially diagnosed as IGHD at childhood. Half of our patients with IGHD developed the second hormone deficiency after 5 years of diagnosis, reinforcing the need for lifelong monitoring of pituitary function in these patients.
Authors: Maria Geralda F Osorio; Suemi Marui; Alexander A L Jorge; Ana C Latronico; Leonard S S Lo; Claudia C Leite; Vivian Estefan; Berenice B Mendonca; Ivo J P Arnhold Journal: J Clin Endocrinol Metab Date: 2002-11 Impact factor: 5.958
Authors: Marcela M França; Alexander A L Jorge; Luciani R S Carvalho; Everlayny F Costalonga; Gabriela A Vasques; Claudia C Leite; Berenice B Mendonca; Ivo J P Arnhold Journal: J Clin Endocrinol Metab Date: 2010-08-04 Impact factor: 5.958
Authors: R J Kuczmarski; C L Ogden; L M Grummer-Strawn; K M Flegal; S S Guo; R Wei; Z Mei; L R Curtin; A F Roche; C L Johnson Journal: Adv Data Date: 2000-06-08
Authors: Eva Fernandez-Rodriguez; Celsa Quinteiro; Jesus Barreiro; Mónica Marazuela; Inmaculada Pereiro; Roberto Peinó; Jose Manuel Cabezas-Agrícola; Fernando Dominguez; Felipe F Casanueva; Ignacio Bernabeu Journal: Neuroendocrinology Date: 2011-02-08 Impact factor: 4.914
Authors: Eveline G P Silva; Natasha Slhessarenko; Ivo J P Arnhold; Marcelo C Batista; Vivian Estefan; Maria G F Osorio; Suemi Marui; Berenice B Mendonca Journal: Horm Res Date: 2003
Authors: Cheri Deal; Caroline Hasselmann; Roland W Pfäffle; Alan G Zimmermann; Charmian A Quigley; Christopher J Child; Elena P Shavrikova; Gordon B Cutler; Werner F Blum Journal: Horm Res Paediatr Date: 2013-05-16 Impact factor: 2.852
Authors: Qing Fang; Akima S George; Michelle L Brinkmeier; Amanda H Mortensen; Peter Gergics; Leonard Y M Cheung; Alexandre Z Daly; Adnan Ajmal; María Ines Pérez Millán; A Bilge Ozel; Jacob O Kitzman; Ryan E Mills; Jun Z Li; Sally A Camper Journal: Endocr Rev Date: 2016-11-09 Impact factor: 19.871
Authors: María I Pérez Millán; Sebastian A Vishnopolska; Alexandre Z Daly; Juan P Bustamante; Adriana Seilicovich; Ignacio Bergadá; Débora Braslavsky; Ana C Keselman; Rosemary M Lemons; Amanda H Mortensen; Marcelo A Marti; Sally A Camper; Jacob O Kitzman Journal: Mol Genet Genomic Med Date: 2018-05-08 Impact factor: 2.183
Authors: Marilena Nakaguma; Fernanda A Correa; Lucas S Santana; Anna F F Benedetti; Ricardo V Perez; Martha K P Huayllas; Mirta B Miras; Mariana F A Funari; Antonio M Lerario; Berenice B Mendonca; Luciani R S Carvalho; Alexander A L Jorge; Ivo J P Arnhold Journal: Endocr Connect Date: 2019-05-01 Impact factor: 3.335
Authors: Sebastian Alexis Vishnopolska; Maria Florencia Mercogliano; Maria Andrea Camilletti; Amanda Helen Mortensen; Debora Braslavsky; Ana Keselman; Ignacio Bergadá; Federico Olivieri; Lucas Miranda; Roxana Marino; Pablo Ramírez; Natalia Pérez Garrido; Helen Patiño Mejia; Marta Ciaccio; Maria Isabel Di Palma; Alicia Belgorosky; Marcelo Adrian Martí; Jacob Otto Kitzman; Sally Ann Camper; Maria Ines Pérez-Millán Journal: J Clin Endocrinol Metab Date: 2021-06-16 Impact factor: 6.134