Antunes, A

Espada, F

Guimarães, J

Marques, O

Oliveira, MJ

Portuguese Endocrinology and Diabetes Pediatric Society (SPEDP)

Portuguese Multicentre Group

English

Institutional repository of Hospital de Braga

Treatment of Pediatric Graves Disease:  results of a multicenter survey in Portugal Background and objective Pediatric Graves Disease (GD) is a rare disease and there is currently no evidence-based strategy for its management. Therapy choice is determined by individual consideration of the risks and benefits of the three treatment modalities and varies considerably within and between countries, depending on local traditions and resources and on the different clinical conditions of the patients. In 2011, American Thyroid Association (ATA) and the American Association of Clinical Endocrinologists (AACE) published Guidelines on GD, changing disease`s evaluation. Nevertheless, GD treatment is still a matter of controversy.  The main objective was to know the characteristics of the pediatric Portuguese GD population, how the different therapies were used and their results.  Printed by SPEDP conducted the first nationwide questionnaire survey among all the Endocrinologists and Pediatricians in the Portuguese Public Health System Hospitals about pediatric GD treatment. A questionnaire (Fig. 1) was designed and distributed to all hospitals with pediatric endocrinology, in order to include all GD patients under 18 years at diagnosis and with a minimum of 6 months follow-up. A retrospective assessment of patient`s medical records was performed from May to  August 2013. For the statistical analysis we used Excell® and SPSS®.  We used Kruskal-Wallis Test and Spearman’s correlation and considered a level of significance of 5%. Portuguese Endocrinology and Diabetes Pediatric Society (SPEDP): Marques,O; Oliveira, MJ; Antunes, A; Espada, F; Guimarães, J. and Portuguese Multicentre Group: Mirante A, Monteiro A; Vasconcelos C, Meireles C; Brandão C; Galo E; Fernandes I; Anselmo J; Lopes L; Sampaio L; Fonseca M; Almeida M; Ferreira M; Anselmo M; Campos R A; Bernardo T; Borges T; Pratas S; Sousa S; Silva T; Leite V • 87 hospitals contacted, 67 responders • 25 hospitals reported patients  • Pediatric clinic – 56%; Endocrinology clinic – 44%  • 152 patients were identified, 76% were females • Mean age at diagnosis: 11,3 ±3,4  years [3,3-18,3] Type of ATD Total patients (N/%) Initial dose (mg/Kg) Initial ATD duration (months) Adverse effects (all minor)  (N/%)  Methimazole (MTZ) 105/69.1 0.38+0.23 28.26+21.27 7/4.6  Propylthiouracil (PTU) 47/30.9 3.74+2.65 41.51+38.55 3/1.9  Initial treatment with ATD N=152 Stopped  ATD                 N=92 Remission and relapse N=26 Surgery  N=2 Iodine 131  N=5 ATD N=19 Remission, no relapse N=39  No remission N=27 Iodine 131 N=14 Surgery N=13 Ongoing ATD N=60                                 PTU                       N=8 MTZ                N=52 Remission, no relapse Remission and Relapse No remission p Number of patients (%) 39 (42,4%) 26 (28,3%) 27 (29,3%) NS Age at diagnosis (years) 10.57+3.36 12.02+3.32 11.46+3.60 NS Type ATD  (PTU/TMZ) 23/16 7/19* 9/18* 0.02* ATD duration (months) 35.59+22.24 26.85+14.89 41.67+47.61 NS MTZ dose (mg/Kg) 0.36+0.14 0.42+0.19 0.40+0.18 NS PTU dose (mg/Kg) 3.96+2.07 2.33+2.46 5.71+3.16** 0.024** Combination therapy (ATD+LT4) (N/%) 20 (51,3%) 17 (65,4%) 10 (37,0%) NS *p<0.05 vs remission, no relapse; **p<0.05 vs remission and relapse Initial and final TRABs and remission status (N=92)  A statistically significant positive association between final, (but not inicial (p=0.272)) TRABs and remission/relapse category was found. Patients without remission had higher final TRABs (Spearman correlation  r=0.264; p=0.011) 19 11 9 3 4 1 2 3 1 11 8 15 4 0 1 02468101214161820Remission, norelapseRemission andrelapseNo remissionNumber of patients 35 24 17 1 2 1 1 0 5 2 0 1 0510152025303540Remission, norelapseRemission andrelapseNo remission1-3 times4-5 times6-10 times11-50 times> 50 timesThis first multicenter portuguese survey is representative of the national pediatric GD population as it included 77% of all hospitals in our country. ATD therapy was the first choice for all patients, MTZ being preferred. Only minor adverse effects of the drugs were found. Despite a long treatment duration, only 25% patients achieved long term remission. Definitive therapy was a second choice in few patients (22.4%) and more frequent in recent years. Few significant predictive factors of remission were found between groups that finished first course of ATD, namely lower final TRABs and the use of MTZ instead of PTU.  Our population results are conform with literature and reflect a tendency to change GD therapy, introducing definite therapy sooner in the course of treatment. 4 5 3 4 9 15 7 10 13 15 16 28 17 6 05101520253035401 to 6 7 to 12 13-18 19-24 25-36 37-60 >60Number of patients MethimazolePropylthiouracilTreatment evolution 1 2 3 2 5 9 8 8 15 4 3 0246810121416181 to 6 7 to 12 13-18 19-24 25-36 37-60 >60Number of patients MethimazolePropylthiouracilPatients that stopped ATD (N=92) Methods Results Conclusion TRABs evolution (N=152) 63 14 18 50 7 128 7 8 6 3 0204060801001201401-3 times 4-5 times 6-10 times 11-50 times > 50 timesNumber of patients InitialFinal3,9 32,3 63,9 010203040506070< 4 years 5-10 years 11-18 yearsPercentage of patients Figure 2 – Age at dianosis Figure 3 – Duration of firts course of ATD (months) Table 1 – Initial treatment with ATD and adverse effects Table 2 –  Caracterization of patients that stopped ATD Figure 4 – Comparison of TRABs before and after treatment (increased number of times) Figure 5 – Treatment evolution Figure 6 – Duration of treatment  in patients  ongoing first ATD course (months) Figure 7 – Initial and final TRABs (increased number of times) and remission/relapse status Figure 1 – Questionnaire Patients ongoing first course ATD (N=60) Bibliography: 1. Bahn Chair RS, Burch HB, Cooper DS, et al. Hyperthyroidism and other causes of thyrotoxicosis: management guidelines of the American Thyroid Association and American Association of Clinical Endocrinologists. Thyroid 2011; 612-617; 2. Léger J, Gelwane G, Kaguelidou F, et al. Positive impact of long-term antithyroid drug treatment on the outcome of children with Graves' disease: national long-term cohort study. J Clin Endocrinol Metab 2012; 97:110; 3. Ohye H, Minagawa A, Noh JY, et al. Antithyroid drug treatment for graves' disease in children: a long-term retrospective study at a single institution. Thyroid 2014; 24:200; 4. Kaguelidou F, Alberti C, Castanet M, et al. Predictors of autoimmune hyperthyroidism relapse in children after discontinuation of antithyroid drug treatment. J Clin Endocrinol Metab 2008; 93:3817; 5. Stephen LaFranchi, Treatment and prognosis of Graves`disease in children and adolescentes. Uptodate, may 2013; 6. Leger J.et al. Graves`disease in children.  Best Practice & Research Clinical Endocrinology & Metabolism 28 (2014) 233–243  

Treatment of Pediatric Graves Disease:  results of a multicenter survey in Portugal

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Treatment of Pediatric Graves Disease: results of a multicenter survey in Portugal

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