Lymphocytic hypophysitis, an autoimmune mediated chronic inflammation of the pituitary gland, is uncommon in children.1 The usual presentation is with symptoms secondary to pressure from the enlargement of the gland itself and or impairment of pituitary hormones of varying degrees, in conjunction with a sellar mass. 2,3 In 80% of cases, partial or panhypopituitarism is present with approximately 75% of cases having multiple hormonal deficiencies.2 We report the case of a prepubertal female who presented with cyclical vomiting, as her initial symptom of lymphocytic hypophysitis which resolved promptly after pituitary biopsy. This case suggests that cyclical vomiting can be a symptom associated with lymphocytic hypophysitis. Therefore, it should be considered in the differential diagnosis of any child presenting with cyclical vomiting.
Maghnie M., Ghirardello S., De Bellis A., et al. Idiopathic central diabetes insipidus in children and young adults in commonly associated with vasopressin cell antibodies and markers of autoimmunity. Clin Endocrinol. 2006;65:470-478.
6.
Hoshimaru M. , Hashimoto N., Kikuchi H.Central diabetes insipidus resulting from a nonneoplastic tiny mass lesion localized in the neurohypophyseal system . Surg Neurol. 1992;38:1-6.
7.
Atkins D., Sanford R., Thomas B., Joyner R.Infundibuloneurohypophysitis in children. Pediatr Neurosurg . 1999;30:267-271.
8.
De Buyst J. , Massa G., Christophe C., Heinrichs C.Clinical, hormonal and imaging findings in 27 children with central diabetes insipidus. Eur J Pediatr . 2006;166:43-46.
9.
Maghnie M., Cosi G., Genovese E., et al. Central diabetes insipidus in children and young adults. N Engl J Med. 2000;343:998-1007.
10.
Collin P., Hakanen M., Salmi J., Mäki M., Kaukinen K.Autoimmune hypopituitarism in patients with coeliac disease: symptoms confusingly similar. Scand J Gastroentrol. 2001;5:558-560.
11.
Bastida Eizaguirre M., Arto Urzainqui MJ, Iturbe Ortizde Urbina R.[Diabetes insipidus and panhypopituitarism in an 8-year-old girl due to probable lymphocytic hypophysitis] . An Esp Pediatr. 1996;44:402-404.
12.
Maghnie M., Genovese E., Sommaruga MG, et al. Evolution of childhood central diabetes insipidus into panhypopituitarism with a large hypothalamic mass: is ‘lymphocytic infundibuloneurohypophysitis’ in children a different entity? Eur J Endocrinol. 1998; 139:635-640.
13.
Bettendorf M., Fehn M., Grulich-Henn J., et al. Lymphocytic hypophysitis with central diabetes insipidus and consequent panhypopituitarism preceding a multifocal, intracranial germinomas in a prepubertal girl. Eur J Pediatr . 1999;158:288-292.
14.
Mootha SL, Barkovich AJ, Grumbach MM, et al. Idiopathic hypothalamic diabetes insipidus, pituitary stalk thickening, and the occult intracranial germinomas in children and adolescents . J Clin Endocrinol Metab. 1997;82:1362-1367.
15.
Beni-Adani L., Sainte-Rose C., Zerah M., et al. Surgical implications of the thickened pituitary stalk accompanied by central diabetes insipidus . J Neurosurg Pediatr. 2005;103:142-147.
16.
Younes JS, Secord EAPanhypopituitarism in a child with common variable immunodeficiency . Ann Allergy Asthma Immunol. 2002;89:322-325.
17.
Sato N., Szes G., Endo K.Hypophysitis: endocrinologic and dynamic MR findings. AJNR Am J Neuroradiol. 1998;19: 439-444.
18.
Gutenberg A. , Buslei R., Fahlbusch R., et al. Immunopathology of primary hypophysitis: implications for pathogenesis. Am J Surg Pathol. 2005;29:329-338.
19.
Scherbaum WA , Czernichow P., Bottazzo GF, Doniach D.Diabetes insipidus in children. IV. A possible autoimmune type with vasopressin cell antibodies . J Pediatr . 1985;107:922-925.
20.
Fehn M., Bettendorf M., Lüdecke DK, et al. Lymphocytic hypophysitis masking a suprasellar germinoma in a 12-year-old girl: a case report. Pituitary. 1999;1:303-307.
21.
Leger J., Velasquez A., Garel C., et al. Thickened pituitary stalk on magnetic resonance imaging in children with central diabetes insipidus. J Clin Endocrinol Metab. 1999;84:1954-1960.
22.
Iughetti L. , De Bellis A., Predieri B., et al. Growth hormone impaired secretion and antipituitary antibodies in patients with celiac disease and poor catch-up growth after a long gluten-free diet period: a causal association ? Eur J Pediatr. 2006;166:897-903.
23.
Papathanasiou A., Kousta E., Skarpa V., et al. Growth hormone deficiency in a patient with autoimmune polyendocrinopathy type 2. Hormones ( Athens). 2007;6:247-250.
Mikami-Terao Y., Akiyama M., Yanagisawa T., et al. Lymphocytic hypophysitis with central diabetes insipidus and subsequent hypopituitarism masking a suprasellar germinoma in a 13-year-old girl. Childs Nerv Syst. 2006 ;22:1338-1343.
26.
Al-Mujainim A., Ganesh A., Al-Zuhaibi S., et al. Lymphocytic infundibulo-neurohypophysitis: an unusual cause of recurrent optic neuropathy in a child. J AAPOS. 2009;13:207-209.
27.
Cemeroglu AP , Blaivas M., Muraszko KM, et al. Lymphocytic hypophysitis presenting with diabetes insipidus in a 14-year-old girl: case report and review of the literature. Eur J Pediatr. 1997;165:684-688.
28.
Giavoli G., Ferrante E., Bergamaschi S., et al. An unusual case of recurrent autoimmune hypophysitis. Exp Clin Endocrinol Diabetes. 2010;118:287-290.
29.
Ahmadi J., Scott Meyers G., Segall HD, et al. Lymphocytic adenohypophysitis: contrast-enhanced MR imaging in five cases. Radiology. 1995;195:30-34.
30.
Mizokami T. , Inokuchi K., Okamura K., et al. Hypopituitarism associated with transient diabetes insipidus followed by an episode of painless thyroiditis in a young man. Intern Med. 1996 ; 35:135-141.
31.
Brandes JC, Cerletty JMPregnancy in lymphocytic hypophysitis: case report and review. Wis Med J. 1989;88:29-32.
32.
Flanagan DEH , Ibrahim AEK, Ellison DW, et al. Inflammatory hypophysitis: the spectrum of disease. Acta Neurochir ( Wein). 2002;144:47-56.
33.
Pérez-Núñez A., Miranda P., Arrese I., et al. Lymphocytic hypophysitis with cystic MRI appearance. Acta Neurochir ( Wein). 2005;147:1297-1300.
34.
Zeller JR, Cerletty JM, Rabinovitch RA, Daniels D.Spontaneous regression of a post-partum pituitary mass demonstrated by computed tomography. Arch Intern Med. 1982 ;142:373-374.
35.
Levine SN, Benzel EC, Fowler MR, et al. Lymphocytic adenohypophysitis: clinical, radiological, and magnetic resonance imaging characterization. Neurosurgery. 1988;22:937-941.
36.
Honegger J. , Fahlbusch R., Bomemann A., et al. Lyphocytic and granulomatous hypophysitis: experience with nine cases (clinical studies) . Neurosurgery . 1997;40:713-723.
37.
Weimann E., Mölenkamp G., Böhles HJDiabetes insipidus due to hypophysitis. Horm Res. 1997;47:81-84.
38.
Catruegli P.Editorial: autoimmune hypophysitis: an underestimated disease in search of its autoantigen(s). J Clin Endocrinol Metab. 2007;92:2038-2040.
