Eur J Endocrinol
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DOI: 10.1530/eje.0.1390635
European Journal of Endocrinology, Vol 139, Issue 6, 635-640
Copyright © 1998 by European Society of Endocrinology
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Case Reports

Evolution of childhood central diabetes insipidus into panhypopituitarism with a large hypothalamic mass: is 'lymphocytic infundibuloneurohypophysitis' in children a different entity?

M Maghnie, E Genovese, MG Sommaruga, M Arico, D Locatelli, E Arbustini, S Pezzotta, and F Severi

Department of Pediatrics, University of Pavia, IRCCS Policlinico S. Matteo, Italy.

We report on a 15-year-old girl who had presented with acute onset central diabetes insipidus at the age of 8 years; this was followed by growth failure due to acquired growth hormone deficiency. Initial magnetic resonance imaging showed a uniformly enlarged pituitary stalk and absence of posterior pituitary hyperintensity. Frequent patient examination and magnetic resonance imaging gave unchanged results until after 5 years a large hypothalamic mass and panhypopituitarism were found. Dynamic magnetic resonance imaging documented hypothalamic-pituitary vasculopathy. Histopathological examination revealed perivascular inflammatory lymphoplasmic infiltrates with no granulomatosis or necrosis and negative staining for S-100 protein, suggesting autoimmune inflammatory disease (lymphocytic infundibuloneurohypophysitis?). The response to glucocorticoid pulses (30 mg/kg per day for 3 days i.v.) was favorable. the hypothalamic mass being halved and partial anterior pituitary function recovery maintained for 2 years after the start of treatment. We suggest that long-term surveillance is needed for isolated and chronic thickening of the pituitary stalk and that dynamic magnetic resonance imaging can contribute to the demonstration of hypothalamic-pituitary vascular impairment associated with local vasculitis.


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M. Maghnie, M. Altobelli, N. di Iorgi, E. Genovese, G. Meloni, M. L. Manca-Bitti, A. Cohen, and S. Bernasconi
Idiopathic Central Diabetes Insipidus Is Associated with Abnormal Blood Supply to the Posterior Pituitary Gland Caused by Vascular Impairment of the Inferior Hypophyseal Artery System
J. Clin. Endocrinol. Metab., April 1, 2004; 89(4): 1891 - 1896.
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 NEJMHome page
M. Maghnie, G. Cosi, E. Genovese, M. L. Manca-Bitti, A. Cohen, S. Zecca, C. Tinelli, M. Gallucci, S. Bernasconi, B. Boscherini, et al.
Central Diabetes Insipidus in Children and Young Adults
N. Engl. J. Med., October 5, 2000; 343(14): 998 - 1007.
[Abstract] [Full Text] [PDF]


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