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Journal of Pediatric Endocrinology and Metabolism

Editor-in-Chief: Kiess, Wieland

Ed. by Bereket, Abdullah / Darendeliler, Feyza / Dattani, Mehul / Gustafsson, Jan / Luo, Feihong / Mericq, Veronica / Roth, Christian / Toppari, Jorma

Editorial Board Member: Battelino, Tadej / Buyukgebiz, Atilla / Cassorla, Fernando / Chrousos, George P. / Cutfield, Wayne / Fideleff, Hugo L. / Hershkovitz, Eli / Hiort, Olaf / LaFranchi, Stephen H. / Lanes M. D., Roberto / Mohn, Angelika / Root, Allen W. / Rosenfeld, Ron G. / Werther, George / Zadik, Zvi

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2191-0251
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Volume 27, Issue 1-2 (Jan 2014)

Issues

Pediatric idiopathic intracranial hypertension and the underlying endocrine-metabolic dysfunction: a pilot study

Vincenzo Salpietro / Kshitij Mankad
  • Department of Pediatrics, University of Messina, Messina, Italy
  • Great Ormond Street Children’s Hospital, London, UK
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
/ Maria Kinali / Ashok Adams
  • Department of Pediatrics, University of Messina, Messina, Italy
  • Great Ormond Street Children’s Hospital, London, UK
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
/ Mariella Valenzise / Gaetano Tortorella / Eloisa Gitto / Agata Polizzi
  • National Centre for Rare Diseases, Superior Institute of Health, Rome, Italy
  • Institute of Neurological Sciences, National Research Council (CNR), Catania, Italy
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
/ Valeria Chirico / Francesco Nicita / Emanuele David / Anna Claudia Romeo / Carlo Attilio Squeri
  • Ophthalmology Unit, Department of Experimental Medical Surgical Sciences and Odontostomatology, University of Messina, Messina, Italy
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
/ Salvatore Savasta / Gian Luigi Marseglia / Teresa Arrigo / Conrad Earl Johanson
  • Department of Pediatrics, University of Messina, Messina, Italy
  • Department of Neurosurgery, Brown University, Providence, Rhode Island, USA
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
/ Martino Ruggieri
  • Department of Pediatrics, University of Messina, Messina, Italy
  • Department of Educational Sciences, University of Catania, Catania, Italy
  • Unit of Neurosurgery, University of Catania, Catania, Italy
  • Other articles by this author:
  • De Gruyter OnlineGoogle Scholar
Published Online: 2013-09-11 | DOI: https://doi.org/10.1515/jpem-2013-0156

Abstract

Aim: To unravel the potential idiopathic intracranial hypertension (IIH) endocrine-metabolic comorbidities by studying the natural (and targeted drug-modified) history of disease in children. IIH is a disorder of unclear pathophysiology, characterized by raised intracranial pressure without hydrocephalus or space-occupying lesion coupled with normal cerebrospinal fluid (CSF) composition.

Methods: Retrospective study (years 2001–2010) of clinical records and images and prospective follow-up (years 2010–2013) in 15 children (11 girls, 4 boys; aged 5–16 years) diagnosed previously as “IIH”, according to the criteria for pediatric IIH proposed by Rangwala, at four university pediatric centers in northern, central, and southern Italy.

Results: We identified six potential endocrine-metabolic comorbidities including, weight gain and obesity (n=5), recombinant growth hormone therapy (n=3), obesity and metabolic syndrome (n=1), secondary hyperaldosteronism (n=1), hypervitaminosis A (n=1), and corticosteroid therapy (n=1). Response to etiologically targeted treatments (e.g., spironolactone, octreotide) was documented.

Conclusions: IIH is a protean syndrome caused by various potential (risk and) associative factors. Several conditions could influence the pressure regulation of CSF. An endocrine-metabolic altered homeostasis could be suggested in some IIH patients, and in this context, etiologically targeted therapies (spironolactone) should be considered

Keywords: aldosterone; childhood obesity; children; idiopathic intracranial hypertension; magnetic resonance imaging; metabolic syndrome; pseudotumor cerebri; spironolactone

References

  • 1.

    Radhakrishnan K, Ahlskog JE, Cross SA, Kurland LT, O’Fallon WM. Idiopathic intracranial hypertension (pseudotumour cerebri): descriptive epidemiology in Rochester, Minn, 1976 to 1990. Arch Neurol 1993;50:78–80.Google Scholar

  • 2.

    Hacifazlioglu Eldes N, Yilmaz Y. Pseudotumour cerebri in children: etiological, clinical features and treatment modalities. Eur J Paediatr Neurol 2012;16:349–55.CrossrefPubMedWeb of ScienceGoogle Scholar

  • 3.

    Per H, Canpolat M, Gümüş H, Poyrazoğlu HG, Yıkılmaz A, et al. Clinical spectrum of the pseudotumor cerebri in children: etiological, clinical features, treatment and prognosis. Brain Dev 2013;35:561–8.CrossrefPubMedWeb of ScienceGoogle Scholar

  • 4.

    Distelmaier F, Sengler U, Messing-Juenger M, Assmann B, Mayatepek E, et al. Pseudotumor cerebri as an important differential diagnosis of papilledema in children. Brain Dev 2006;28:190–5.Web of SciencePubMedCrossrefGoogle Scholar

  • 5.

    Shapiro I, Shapiro SK. Familial pseudotumor cerebri and the empty sella syndrome. Ann Ophthalmol 1980;12:1045–8.PubMedGoogle Scholar

  • 6.

    Curry WT Jr, Butler WE, Barker FG II. Rapidly rising incidence of cerebrospinal fluid shunting procedures for idiopathic intracranial hypertension in the United States, 1988–2002. Neurosurgery 2005;57:97–108.Google Scholar

  • 7.

    Wall M, George D. Idiopathic intracranial hypertension. A prospective study of 50 patients. Brain 1991;114:155–80.Google Scholar

  • 8.

    Lessell S. Pediatric pseudotumor cerebri (idiopathic intracranial hypertension). Surv Ophthalmol 1992;37:155–66CrossrefPubMedGoogle Scholar

  • 9.

    Wang SJ, Silberstein SD, Patterson S, Young WB. Idiopathic intracranial hypertension without papilledema. A case-control study in a headache center. Neurology 1998;51:245–49.CrossrefGoogle Scholar

  • 10.

    Lipton HL, Michelson PE. Pseudotumor cerebri syndrome without papilledema. J Am Med Assoc 1972;220:1591–2.Google Scholar

  • 11.

    Rush JA. Pseudotumor cerebri: clinical profile and visual outcome in 63 patients. Mayo Clin Proc 1980;55:541–6.PubMedGoogle Scholar

  • 12.

    Headache Classification Subcommittee of the International Headache Society. The international classification of headache disorders, 2nd ed. Cephalalgia 2004;24(Suppl 1):9–160.Google Scholar

  • 13.

    Rangwala LM, Liu GT. Pediatric idiopathic intracranial hypertension. Surv Ophthalmol 2007;52:597–617.CrossrefPubMedGoogle Scholar

  • 14.

    Phillips PH. Pediatric pseudotumor cerebri. Int Ophthalmol Clin 2012;52:51–9.PubMedCrossrefGoogle Scholar

  • 15.

    Traviesa DC, Schwartzman RJ, Glaser JS, Savino P. Familial benign intracranial hypertension. J Neurol Neurosurg Psychiatry 1976;39:420–3.PubMedCrossrefGoogle Scholar

  • 16.

    Malm J, Kristensen B, Markgren P, Ekstedt J. CSF hydrodynamics in idiopathic intracranial hypertension: a long-term study. Neurology 1992;42:851–8.PubMedCrossrefGoogle Scholar

  • 17.

    Salpietro V, Polizzi A, Bertè LF, Chimenz R, Chirico V, et al. Idiopathic intracranial hypertension: a unifying neuroendocrine hypothesis through the adrenal-brain axis. Neuro Endocrinol Lett 2012;33:569–73.PubMedGoogle Scholar

  • 18.

    Weig SG. Asymptomatic idiopathic intracranial hypertension in young children. J Child Neurol 2002;17:239–41.CrossrefPubMedGoogle Scholar

  • 19.

    Biousse V, Bruce BB, Newman NJ. Update on the pathophysiology and management of idiopathic intracranial hypertension. J Neurol Neurosurg Psychiatry 2012;83:488–94.Web of ScienceCrossrefPubMedGoogle Scholar

  • 20.

    Salpietro V, Ruggieri M, Sancetta F, Colavita L, D’Angelo G, et al. New insights on the relationship between pseudotumor cerebri and secondary hyperaldosteronism in children. J Hypertens 2012;30:629–30.PubMedWeb of ScienceCrossrefGoogle Scholar

  • 21.

    Weber KT. Aldosteronism revisited: perspectives on less well recognized actions of aldosterone. J Lab Clin Med 2003; 142:71–82.Google Scholar

  • 22.

    Khan MU, Khalid H, Salpietro V, Weber KT. idiopathic intracranial hypertension associated with either primary or secondary aldosteronism. Am J Med Sci 2012 [Epub ahead of print].Web of ScienceGoogle Scholar

  • 23.

    Malozowski S, Tanner LA, Wysowski D, Fleming GA. Growth hormone, insulin like growth factor 1, and benign intracranial hypertension. N Engl J Med 1993;329:665–6.Google Scholar

  • 24.

    Johansson JO, Larson G, Andersson M, Elmgren A, Hynsjo L, et al. Treatment of growth hormone-deficient adults with recombinant human growth hormone increases the concentration of growth hormone in the cerebrospinal fluid and affects neurotransmitters. Neuroendocrinology 1995;61:57–66.CrossrefPubMedGoogle Scholar

  • 25.

    Ho KY, Weissberger AJ. The antinatriuretic action of biosynthetic human growth hormone in man involves activation of the renin-angiotensin system. Metabolism 1990;39:133–7.CrossrefPubMedGoogle Scholar

  • 26.

    Panagopoulos GN, Deftereos SN, Tagaris GA, Gryllia M, Kounadi T, et al. Octreotide: a therapeutic option for idiopathic intracranial hypertension. Neurol Neurophysiol Neurosci 2007:1.PubMedGoogle Scholar

  • 27.

    Katz SE, Klisovic DD, O’Dorisio MS, Lynch R, Lubow M. Expression of somatostatin receptors 1 and 2 in human choroid plexus and arachnoid granulations: implications for idiopathic intracranial hypertension. Arch Ophthalmol 2002;120:1540–3.PubMedCrossrefGoogle Scholar

  • 28.

    Sugerman H, Windsor A, Bessos M, Wolfe L. Intra-abdominal pressure, sagittal abdominal diameter and obesity comorbidity. J Intern Med 1997;241:71–9.Google Scholar

  • 29.

    Whiteley W, Al-Shahi R, Warlow CP, Zeidler M, Lueck CJ. CSF opening pressure: reference interval and the effect of body mass index. Neurology 2006;67:1690–1.CrossrefWeb of SciencePubMedGoogle Scholar

  • 30.

    Sinclair AJ, Ball AK, Burdon MA, Clarke CE, Stewart PM, et al. Exploring the pathogenesis of IIH: an inflammatory perspective. J Neuroimmunol 2008;201–202:212–20.Web of ScienceGoogle Scholar

  • 31.

    Lampl Y, Eshel Y, Kessler A, Fux A, Gilad R, et al. Serum leptin level in women with idiopathic intracranial hypertension. J Neurol Neurosurg Psychiatry 2002;72:642–3.PubMedCrossrefGoogle Scholar

  • 32.

    Whaley-Connell A, Johnson MS, Sowers JR. Aldosterone: role in the cardiometabolic syndrome and resistant hypertension. Prog Cardiovasc Dis 2010;52:401–9.CrossrefWeb of ScienceGoogle Scholar

  • 33.

    Ehrhart-Bornstein M, Lamounier-Zepter V, Schraven A, Langenbach J, Willenberg HS, et al. Human adipocytes secrete mineralocorticoid-releasing factors. Proc Natl Acad Sci USA 2003;100:14211–6.CrossrefGoogle Scholar

  • 34.

    Guo C, Ricchiuti V, Lian BQ, Yao TM, Coutinho P, et al. Mineralocorticoid receptor blockade reverses obesity-related changes in expression of adiponectin, peroxisome proliferator-activated receptor-gamma, and proinflammatory adipokines. Circulation 2008;117:2253–61.Web of ScienceGoogle Scholar

  • 35.

    Weiss R, Dziura J, Burgert TS, Tamborlane WV, Taksali SE, et al. Obesity and the metabolic syndrome in children and adolescent N Engl J Med 2004;350:2362–74.Google Scholar

  • 36.

    Brara SM, Koebnick C, Porter AH, Langer-Gould A. Pediatric idiopathic intracranial hypertension and extreme childhood obesity. J Pediatr 2012;161:602–7.PubMedCrossrefWeb of ScienceGoogle Scholar

  • 37.

    Salpietro V, Chimenz R, Arrigo T, Ruggieri M. Pediatric idiopathic intracranial hypertension and extreme childhood obesity: a role of weight gain. J Pediatr 2013;162:1084.CrossrefPubMedWeb of ScienceGoogle Scholar

  • 38.

    Zada G, Tirosh A, Kaiser UB, Laws ER, Woodmansee WW. Cushing’s disease and idiopathic intracranial hypertension: case report and review of underlying pathophysiological mechanisms J Clin Endocrinol Metab 2010;95:4850–4.CrossrefWeb of ScienceGoogle Scholar

  • 39.

    Sinclair AJ, Onyimba CU, Khosla P, Vijapurapu N, Tomlinson JW, et al. Corticosteroids, 11beta-hydroxysteroid dehydrogenase isozymes and the rabbit choroid plexus. J Neuroendocrinol 2007;19:614–20.CrossrefWeb of ScienceGoogle Scholar

  • 40.

    Sowers JR, Whaley-Connell A, Epstein M. The emerging clinical implications of the role of aldosterone in the metabolic syndrome and resistant hypertension. Ann Intern Med 2009;150:776–83.Google Scholar

  • 41.

    Kushida A, Tamura H. Retinoic acids induce neurosteroid biosynthesis in human glial GI-1 cells via the induction of steroidogenic genes. J Biochem 2009;146:917–23.Web of ScienceGoogle Scholar

About the article

Corresponding author: Vincenzo Salpietro, MD, Unit of Genetics and Pediatric Immunology, Department of Pediatrics, University of Messina, Policlinico Universitario “Gaetano Martino”, Via Consolare Valeria, 1 98125 Messina, Italy, Phone: +39 090 2213115, Fax: +39 090 2217029, E-mail:


Received: 2013-04-21

Accepted: 2013-08-06

Published Online: 2013-09-11

Published in Print: 2014-01-01


Citation Information: Journal of Pediatric Endocrinology and Metabolism, ISSN (Online) 2191-0251, ISSN (Print) 0334-018X, DOI: https://doi.org/10.1515/jpem-2013-0156.

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