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Development of a novel score to predict probability of growth without growth hormone after resection of paediatric craniopharyngiomas: relative to tumour growth pattern

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Abstract

Objective

Some patients with paediatric craniopharyngiomas (PCs) showed normal growth despite growth hormone deficiency, which is known as growth without GH (GWGH); however, its mechanism remains unclear. We aimed to develop a novel clinical score to predict the probability of GWGH in PCs.

Methods

A total of 708 PC patients were prospectively enrolled from six hospitals, among which 431 patients were finally included. Data from four of the six hospitals (n = 325) were used to develop the innovative clinical score (ICS), which was further validated using the data from the other two hospitals (n = 106). To establish and validate the ICS, sequential logistic regression was used to analyse the clinical characteristics including tumour growth pattern and tumour size and so on. Furthermore, C-statistic was employed to calibrate the discriminatory ability of the established clinical score, while a calibration plot was adopted for further assessment.

Results

The overall incidence of GWGH was 16.9% (73/431). The ICS ranged from 2 to 23, with an optimism-corrected C-statistic of 0.820, Furthermore, the optimism-corrected C-statistic of external validation was 0.835, indicating good discriminatory power and robustness of the clinical score. Additionally, no apparent overestimation or underestimation was observed in the calibration plots, which showed excellent calibration power of the clinical score.

Conclusions

Based on tumour growth patterns and PC patients’ clinical characteristics, individualized surgical strategies were promising to achieve long-term effective management of PC patients. The ICS is valuable for the evaluation of probability of developing postoperative GWGH.

Clinical trial registration

ClinicalTrials.gov ID: NCT00949156.

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References

  1. Qi S, Lu Y, Pan J, Zhang X, Long H, Fan J (2011) Anatomic relations of the arachnoidea around the pituitary stalk: relevance for surgical removal of craniopharyngiomas. Acta Neurochir (Wien) 153:785–796

    Article  Google Scholar 

  2. Müller HL (2019) Management of endocrine disease: childhood-onset craniopharyngioma: state of the art of care in 2018. Eur J Endocrinol. https://doi.org/10.1530/EJE-18-1021

    Article  PubMed  Google Scholar 

  3. Qi S, Pan J, Lu Y, Gao F, Cao Y, Peng J, Zhang X (2012) The impact of the site of origin and rate of tumour growth on clinical outcome in children with craniopharyngiomas. Clin Endocrinol (Oxf) 76:103–110

    Article  Google Scholar 

  4. Bucher H, Zapf J, Torresani T, Prader A, Froesch ER, Illig R (1983) Insulin-like growth factors I and II, prolactin, and insulin in 19 growth hormone-deficient children with excessive, normal, or decreased longitudinal growth after operation for craniopharyngioma. N Engl J Med 309:1142–1146

    Article  CAS  Google Scholar 

  5. Lazar L, Dan S, Phillip M (2003) Growth without growth hormone: growth pattern and final height of five patients with idiopathic combined pituitary hormone deficiency. Clin Endocrinol (Oxf) 59:82–88

    Article  CAS  Google Scholar 

  6. Iwayama H, Kamijo T, Ueda N (2011) Hyperinsulinemia may promote growth without GH in children after resection of suprasellar brain tumors. Endocrine 40:130–133

    Article  CAS  Google Scholar 

  7. Pavlou M, Tsatsoulis A, Efstathiadou Z, Bitsis S, Papadopoulou ZL (2001) A study of the growth-promoting and metabolic effects of growth hormone (GH) in a patient with the “growth without GH” syndrome. Growth Horm IGF Res 11:225–230

    Article  CAS  Google Scholar 

  8. Lee SS, Han AL, Ahn MB, Kim SH, Cho WK, Cho KS, Park SH, Jung MH, Suh BK (2017) Growth without growth hormone in combined pituitary hormone deficiency caused by pituitary stalk interruption syndrome. Ann Pediatr Endocrinol Metab 22:55–59

    Article  Google Scholar 

  9. Liu Y, Qi ST, Wang CH, Pan J, Fan J, Peng JX, Zhang X, Bao Y, Liu YW (2018) Pathological relationship between adamantinomatous craniopharyngioma and adjacent structures based on QST classification. J Neuropathol Exp Neurol 77:1017–1023

    Article  Google Scholar 

  10. Pan J, Qi S, Liu Y, Lu Y, Peng J, Zhang X, Xu Y, Huang GL, Fan J (2016) Growth patterns of craniopharyngiomas: clinical analysis of 226 patients. J Neurosurg Pediatr 17:418–433

    Article  Google Scholar 

  11. Grimberg A, DiVall SA, Polychronakos C, Allen DB, Cohen LE, Quintos JB, Rossi WC, Feudtner C, Murad MH (2016) Guidelines for growth hormone and insulin-like growth factor-I Treatment in children and adolescents: growth hormone deficiency, idiopathic short stature, and primary insulin-like growth factor-I deficiency. Horm Res Paediatr 86:361–397

    Article  CAS  Google Scholar 

  12. Murray RO (1976) Assessment of skeletal maturity and prediction of adult height (TW2 method). Proc R Soc Med 69:542

    PubMed Central  Google Scholar 

  13. Oto Y, Matsubara K, Ayabe T, Shiraishi M, Murakami N, Ihara H, Matsubara T, Nagai T (2018) Delayed peak response of cortisol to insulin tolerance test in patients with Prader–Willi syndrome. Am J Med Genet A 176:1369–1374

    Article  CAS  Google Scholar 

  14. Koulouri O, Auldin MA, Agarwal R, Kieffer V, Robertson C, Falconer Smith J, Levy MJ, Howlett TA (2011) Diagnosis and treatment of hypothyroidism in TSH deficiency compared to primary thyroid disease: pituitary patients are at risk of under-replacement with levothyroxine. Clin Endocrinol (Oxf) 74:744–749

    Article  CAS  Google Scholar 

  15. Zong XN, Li H (2013) Construction of a new growth references for China based on urban Chinese children: comparison with the WHO growth standards. PLoS One 8:e5969

    Google Scholar 

  16. Xu S, Gu X, Pan H, Zhu H, Gong F, Li Y, Xing Y (2010) Reference ranges for serum IGF-1 and IGFBP-3 levels in Chinese children during childhood and adolescence. Endocr J 57:221–228

    Article  CAS  Google Scholar 

  17. Halbreich U, Kinon BJ, Gilmore JA, Kahn LS (2003) Elevated prolactin levels in patients with schizophrenia: mechanisms and related adverse effects. Psychoneuroendocrinol 28:53–67

    Article  CAS  Google Scholar 

  18. Collins GS, Reitsma JB, Altman DG, Moons KG (2015) Transparent reporting of a multivariable prediction model for individual prognosis or diagnosis (TRIPOD): the TRIPOD statement. Br J Surg 102:148–158

    Article  CAS  Google Scholar 

  19. Sullivan LM, Massaro JM, D’Agostino RB (2004) Presentation of multivariate data for clinical use: the Framingham Study risk score functions. Stat Med 23:1631–1660

    Article  Google Scholar 

  20. Cook NR (2008) Statistical evaluation of prognostic versus diagnostic models: beyond the ROC curve. Clin Chem 54:17–23

    Article  CAS  Google Scholar 

  21. Hosmer DW Jr, Lemeshow S, Sturdivant RX (2013) The multiple logistic regression model. Applied logistic regression, 3rd edn. Wiley, New York

    Book  Google Scholar 

  22. Olsson DS, Andersson E, Bryngelsson IL, Nilsson AG, Johannsson G (2015) Excess mortality and morbidity in patients with craniopharyngioma, especially in patients with childhood onset: a population-based study in Sweden. J Clin Endocrinol Metab 100:467–474

    Article  CAS  Google Scholar 

  23. Qi S, Peng J, Pan J, Zhang X, Lu Y, Fan J, Huang G (2013) Growth and weight of children with craniopharyngiomas based on the tumour location and growth pattern. J Clin Neurosci 20:1702–1708

    Article  Google Scholar 

  24. Müller HL, Emser A, Faldum A, Bruhnken G, Etavard-Gorris N, Gebhardt U, Oeverink R, Kolb R, Sörensen N (2004) Longitudinal study on growth and body mass index before and after diagnosis of childhood craniopharyngioma. J Clin Endocrinol Metab 89:3298–3305

    Article  Google Scholar 

  25. Yaşargil MG, Curcic M, Kis M, Siegenthaler G, Teddy PJ, Roth P (1990) Total removal of craniopharyngiomas. Approaches and long-term results in 144 patients. J Neurosurg 73:3–11

    Article  Google Scholar 

  26. Steno J, Malácek M, Bízik I (2004) Tumor-third ventricular relationships in supradiaphragmatic craniopharyngiomas: correlation of morphological, magnetic resonance imaging, and operative findings. Neurosurgery 54:1051–1058

    Article  Google Scholar 

  27. Kassam AB, Gardner PA, Snyderman CH, Carrau RL, Mintz AH, Prevedello DM (2008) Expanded endonasal approach, a fully endoscopic transnasal approach for the resection of midline suprasellar craniopharyngiomas: a new classification based on the infundibulum. J Neurosurg 108:715–728

    Article  Google Scholar 

  28. Pinto G, Bussières L, Recasens C, Souberbielle JC, Zerah M, Brauner R (2000) Hormonal factors influencing weight and growth pattern in craniopharyngioma. Horm Res 53:163–169

    CAS  PubMed  Google Scholar 

  29. Geffner ME (1996) The growth without growth hormone syndrome. Endocrinol Metab Clin North Am 25:649–663

    Article  CAS  Google Scholar 

  30. Giustina A, Mazziotti G, Canalis E (2008) Growth hormone, insulin-like growth factors, and the skeleton. Endocr Rev 29:535–559

    Article  CAS  Google Scholar 

  31. Weinzimer SA, Homan SA, Ferry RJ, Moshang T (1999) Serum IGF-I and IGFBP-3 concentrations do not accurately predict growth hormone deficiency in children with brain tumours. Clin Endocrinol (Oxf) 51:339–345

    Article  CAS  Google Scholar 

  32. Hibi I, Tanaka T, Tanae A, Kagawa J, Hashimoto N, Yoshizawa A, Shizume K (1989) The influence of gonadal function and the effect of gonadal suppression treatment on final height in growth hormone (GH)-treated GH-deficient children. J Clin Endocrinol Metab 69:221–226

    Article  CAS  Google Scholar 

  33. Arroyo A, Pernasetti F, Vasilyev VV, Amato P, Yen SS, Mellon PL (2002) A unique case of combined pituitary hormone deficiency caused by a PROP1 gene mutation (R120C) associated with normal height and absent puberty. Clin Endocrinol (Oxf) 57:283–291

    Article  Google Scholar 

Download references

Acknowledgements

The authors thank Duan Chongyang, MD, Xu Hai-yan, RN, Qiu xiao-yu, RN, Wang Hai, MD, and Fan Jun, MD for the collection of data and care of patients enrolled in this study.

Funding

This research received grant from four fund projects in the public, including Science and Technology Program of Guangdong (No. 2017A020215048) and the Natural Science Foundation of Guangdong Province (No. 2019A1515012140), and Clinical Research Startup Program of Southern Medical University by High-level University Construction Funding of Guangdong Provincial Department of Education (LC2016YM001).

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Author notes

  1. J.X. Peng, L. Yang, G.L. Huang, Y. Liu and S.C. Zhang contributes equally to this work.

Authors and Affiliations

  1. Department of Neurosurgery, Nanfang Hospital, Southern Medical University, Guangzhou, 510515, Guangdong, China

    J. X. Peng, L. Yang, G. L. Huang, Y. Liu, S. C. Zhang, J. Pan & S. T. Qi

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  1. J. X. Peng
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Correspondence to J. X. Peng, J. Pan or S. T. Qi.

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Conflict of interest

We declare that we have no other financial and personal relationships with other people or organizations that can inappropriately influence our work, there is no professional or other personal interest of any nature or kind in any product, service and/or company that could be construed as influencing the position presented in, or the review of, the manuscript entitled “Development of a Novel Score to Predict Probability of Growth without Growth Hormone after Resection of Pediatric Craniopharyngiomas: Relative to Tumor Growth Pattern”. All the work is original, Neither the entire paper nor any part of its content has been published, submitted or accepted elsewhere, which has been approved by all authors.

Ethical approval

All procedures performed in studies involving human participants were approved by the ethics committee at the Nanfang Hospital, Southern Medical University (SMU) and its later amendments or comparable ethical standards.

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Informed consent was obtained from all individual participants included in the study.

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Peng, J.X., Yang, L., Huang, G.L. et al. Development of a novel score to predict probability of growth without growth hormone after resection of paediatric craniopharyngiomas: relative to tumour growth pattern. J Endocrinol Invest 43, 737–747 (2020). https://doi.org/10.1007/s40618-019-01154-y

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