{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,13]],"date-time":"2026-04-13T16:08:26Z","timestamp":1776096506298,"version":"3.50.1"},"reference-count":18,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"5","license":[{"start":{"date-parts":[[2026,2,2]],"date-time":"2026-02-02T00:00:00Z","timestamp":1769990400000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Neurosurg Case Lessons"],"published-print":{"date-parts":[[2026,2,2]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Craniopharyngiomas (CPs) usually manifest with symptoms of intracranial hypertension secondary to obstructive hydrocephalus, visual disturbances, or growth failure. Hypoglycemia as the sentinel presentation is exceptionally rare and reflects hypothalamic-pituitary dysfunction.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>A previously healthy 4-year-old girl presented with progressive headache and vomiting for 4 days, followed by coma (Glasgow Coma Scale score 6). Emergency testing revealed profound hypoglycemia (capillary glucose 40 mg\/dL [2.2 mmol\/L]); consciousness returned rapidly after an intravenous dextrose bolus was administered. Because she had been vomiting, the hypoglycemia was initially attributed to malnutrition secondary to poor oral intake. However, recurrence of hypoglycemia within 24 hours prompted further evaluation. Abdominal CT excluded insulinoma and adrenal tumors. Endocrine testing revealed low morning cortisol and free thyroxine with inappropriately low thyroid-stimulating hormone, consistent with central adrenal insufficiency and hypothyroidism, while other hypothalamic-pituitary axes were preserved. Brain MRI showed a cystic sellar-suprasellar mass displacing the optic chiasm. Gross-total resection was achieved via a pterional approach. Histology confirmed an adamantinomatous CP. Postoperatively, hypoglycemia resolved, and the patient remained stable on hydrocortisone and levothyroxine replacement.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>CP, although relatively frequent, may exceptionally present with severe recurrent hypoglycemia and coma. Unexplained pediatric hypoglycemia warrants prompt endocrine evaluation and sellar imaging. Early recognition and multidisciplinary management (neurosurgery and endocrinology teams) are crucial for favorable outcomes.<\/jats:p>\n                    <jats:p>\n                      <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"https:\/\/thejns.org\/doi\/10.3171\/CASE25727\">https:\/\/thejns.org\/doi\/10.3171\/CASE25727<\/jats:ext-link>\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case25727","type":"journal-article","created":{"date-parts":[[2026,2,5]],"date-time":"2026-02-05T13:30:02Z","timestamp":1770298202000},"source":"Crossref","is-referenced-by-count":0,"title":["Pediatric craniopharyngioma: when hypoglycemia reveals a brain tumor. Illustrative case"],"prefix":"10.3171","volume":"11","author":[{"given":"Oualid M.","family":"Hmamouche","sequence":"first","affiliation":[{"name":"Department of Neurosurgery, Hassan II University Hospital, Fez, Morocco"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4720-492X","authenticated-orcid":true,"given":"Marouane","family":"Hammoud","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Hassan II University Hospital, Fez, Morocco"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Faycal","family":"Lakhdar","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Hassan II University Hospital, Fez, Morocco"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mohammed","family":"Benzagmout","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Hassan II University Hospital, Fez, Morocco"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Khalid","family":"Chakour","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Hassan II University Hospital, Fez, Morocco"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mohammed","family":"Chaoui El Faiz","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery, Hassan II University Hospital, Fez, Morocco"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1754","reference":[{"issue":"3","key":"bib1","series-title":"Endocr Rev","first-page":"513","article-title":"Craniopharyngioma","volume":"35","author":"M\u00fcller","year":"2014","unstructured":"M\u00fcller HL.Craniopharyngioma. Endocr Rev. 2014;35(3):513-543."},{"key":"bib2","series-title":"Front Neurol","first-page":"737743","article-title":"Endocrine disorder in patients with craniopharyngioma","volume":"12","author":"Zhou","year":"2021","unstructured":"Zhou ZZhang SHu F.Endocrine disorder in patients with craniopharyngioma. Front Neurol. 2021;12:737743."},{"issue":"2","key":"bib3","series-title":"Neurol India","first-page":"600","article-title":"Long-term outcomes of paediatric-onset craniopharyngioma: a retrospective analysis from a tertiary Care Centre in North India","volume":"70","author":"Harsha","year":"2022","unstructured":"Harsha GSDabadghao PSudhanshu S.Long-term outcomes of paediatric-onset craniopharyngioma: a retrospective analysis from a tertiary Care Centre in North India. Neurol India. 2022;70(2):600-605."},{"issue":"5","key":"bib4","series-title":"J Pediatr Endocrinol Metab","first-page":"695","article-title":"Severe hypoglycemia and reduction of insulin requirement in a girl with insulin-dependent diabetes mellitus: first sign of a craniopharyngioma","volume":"12","author":"Lebl","year":"1999","unstructured":"Lebl JSnajderov\u00e1 MKolouskov\u00e1 S.Severe hypoglycemia and reduction of insulin requirement in a girl with insulin-dependent diabetes mellitus: first sign of a craniopharyngioma. J Pediatr Endocrinol Metab. 1999;12(5):695-697."},{"issue":"12","key":"bib5","series-title":"J Clin Endocrinol Metab","first-page":"5969","article-title":"Defect in epinephrine production in children with craniopharyngioma: functional or organic origin?","volume":"88","author":"Coutant","year":"2003","unstructured":"Coutant RMaurey HRouleau SDefect in epinephrine production in children with craniopharyngioma: functional or organic origin? J Clin Endocrinol Metab. 2003;88(12):5969-5975."},{"issue":"8-9","key":"bib6","series-title":"Childs Nerv Syst","first-page":"640","article-title":"Endocrine manifestations of craniopharyngioma","volume":"21","author":"Halac","year":"2005","unstructured":"Halac IZimmerman D.Endocrine manifestations of craniopharyngioma. Childs Nerv Syst. 2005;21(8-9):640-648."},{"issue":"3","key":"bib7","series-title":"J Clin Med","first-page":"389","article-title":"Neuroendocrine disorders in pediatric craniopharyngioma patients","volume":"4","author":"Daubenb\u00fcchel","year":"2015","unstructured":"Daubenb\u00fcchel AMMM\u00fcller HL.Neuroendocrine disorders in pediatric craniopharyngioma patients. J Clin Med. 2015;4(3):389-413."},{"issue":"5","key":"bib8","series-title":"Pediatr Neonatol","first-page":"466","article-title":"Clinical and endocrinological manifestations of childhood craniopharyngioma","volume":"62","author":"Huang","year":"2021","unstructured":"Huang CCChen YCTsai MCClinical and endocrinological manifestations of childhood craniopharyngioma. Pediatr Neonatol. 2021;62(5):466-474."},{"issue":"3","key":"bib9","series-title":"J Clin Res Pediatr Endocrinol","first-page":"288","article-title":"Comprehensive insights into pediatric craniopharyngioma: endocrine and metabolic profiles, treatment challenges, and long-term outcomes from a multicenter study","volume":"16","author":"\u015e\u0131klar","year":"2024","unstructured":"\u015e\u0131klar Z\u00d6zsu EK\u0131z\u0131lcan \u00c7etin SComprehensive insights into pediatric craniopharyngioma: endocrine and metabolic profiles, treatment challenges, and long-term outcomes from a multicenter study. J Clin Res Pediatr Endocrinol. 2024;16(3):288-296."},{"issue":"3","key":"bib1","series-title":"Endocr Rev","first-page":"513","article-title":"Craniopharyngioma","volume":"35","author":"M\u00fcller","year":"2014","unstructured":"M\u00fcller HL.Craniopharyngioma. Endocr Rev. 2014;35(3):513-543."},{"key":"bib2","series-title":"Front Neurol","first-page":"737743","article-title":"Endocrine disorder in patients with craniopharyngioma","volume":"12","author":"Zhou","year":"2021","unstructured":"Zhou ZZhang SHu F.Endocrine disorder in patients with craniopharyngioma. Front Neurol. 2021;12:737743."},{"issue":"2","key":"bib3","series-title":"Neurol India","first-page":"600","article-title":"Long-term outcomes of paediatric-onset craniopharyngioma: a retrospective analysis from a tertiary Care Centre in North India","volume":"70","author":"Harsha","year":"2022","unstructured":"Harsha GSDabadghao PSudhanshu S.Long-term outcomes of paediatric-onset craniopharyngioma: a retrospective analysis from a tertiary Care Centre in North India. Neurol India. 2022;70(2):600-605."},{"issue":"5","key":"bib4","series-title":"J Pediatr Endocrinol Metab","first-page":"695","article-title":"Severe hypoglycemia and reduction of insulin requirement in a girl with insulin-dependent diabetes mellitus: first sign of a craniopharyngioma","volume":"12","author":"Lebl","year":"1999","unstructured":"Lebl JSnajderov\u00e1 MKolouskov\u00e1 S.Severe hypoglycemia and reduction of insulin requirement in a girl with insulin-dependent diabetes mellitus: first sign of a craniopharyngioma. J Pediatr Endocrinol Metab. 1999;12(5):695-697."},{"issue":"12","key":"bib5","series-title":"J Clin Endocrinol Metab","first-page":"5969","article-title":"Defect in epinephrine production in children with craniopharyngioma: functional or organic origin?","volume":"88","author":"Coutant","year":"2003","unstructured":"Coutant RMaurey HRouleau SDefect in epinephrine production in children with craniopharyngioma: functional or organic origin? J Clin Endocrinol Metab. 2003;88(12):5969-5975."},{"issue":"8-9","key":"bib6","series-title":"Childs Nerv Syst","first-page":"640","article-title":"Endocrine manifestations of craniopharyngioma","volume":"21","author":"Halac","year":"2005","unstructured":"Halac IZimmerman D.Endocrine manifestations of craniopharyngioma. Childs Nerv Syst. 2005;21(8-9):640-648."},{"issue":"3","key":"bib7","series-title":"J Clin Med","first-page":"389","article-title":"Neuroendocrine disorders in pediatric craniopharyngioma patients","volume":"4","author":"Daubenb\u00fcchel","year":"2015","unstructured":"Daubenb\u00fcchel AMMM\u00fcller HL.Neuroendocrine disorders in pediatric craniopharyngioma patients. J Clin Med. 2015;4(3):389-413."},{"issue":"5","key":"bib8","series-title":"Pediatr Neonatol","first-page":"466","article-title":"Clinical and endocrinological manifestations of childhood craniopharyngioma","volume":"62","author":"Huang","year":"2021","unstructured":"Huang CCChen YCTsai MCClinical and endocrinological manifestations of childhood craniopharyngioma. Pediatr Neonatol. 2021;62(5):466-474."},{"issue":"3","key":"bib9","series-title":"J Clin Res Pediatr Endocrinol","first-page":"288","article-title":"Comprehensive insights into pediatric craniopharyngioma: endocrine and metabolic profiles, treatment challenges, and long-term outcomes from a multicenter study","volume":"16","author":"\u015e\u0131klar","year":"2024","unstructured":"\u015e\u0131klar Z\u00d6zsu EK\u0131z\u0131lcan \u00c7etin SComprehensive insights into pediatric craniopharyngioma: endocrine and metabolic profiles, treatment challenges, and long-term outcomes from a multicenter study. 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