{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:17:53Z","timestamp":1784557073929,"version":"3.55.0"},"reference-count":0,"publisher":"Bioscientifica","license":[{"start":{"date-parts":[[2021,7,1]],"date-time":"2021-07-01T00:00:00Z","timestamp":1625097600000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["edm.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2021,7,1]]},"abstract":"\n Summary<\/jats:title>\n Although pituitary macroadenomas often cause mass effects on surrounding structures, it is extremely rare for pituitary lesions to disturb cerebrospinal fluid circulation. Sellar gangliocytoma-pituitary adenomas (SGPAs) are also extremely rare. Here we report the unique case of a man with the unusual combination of acromegaly from an SGPA, who presented with unilateral hydrocephalus. A 60-year-old man presented with rapid neurological deterioration, bitemporal hemianopia, and acromegalic features. Neuroimaging revealed a large sellar lesion extending superiorly into the left foramen of Monro, causing acute obstructive unilateral hydrocephalus. External ventricular drain placement improved consciousness immediately. Biochemical assessment confirmed acromegaly. Following trans-sphenoidal debulking, histology revealed a mixed gangliocytoma\/sparsely-granulated somatotrophinoma. Despite the residual disease, his vision recovered remarkably, low-dose cabergoline controlled residual excess growth hormone (GH) secretion, and the residual tumour has remained extremely stable over 2 years. Hydrocephalus is an extremely rare complication of pituitary lesions, and unilateral hydrocephalus has never been reported previously. GH secretion in SGPAs is more common than for pituitary adenomas in general, raising questions regarding the aetiology and therapeutic approach to this rare combination tumour.<\/jats:p>\n <\/jats:sec>\n \n Learning points<\/jats:title>\n \n \n Pituitary tumours most commonly present with symptoms related to endocrine disturbance or mass effects upon visual pathways (e.g. optic chiasm, nerves in the lateral cavernous sinus). However, extremely rarely, pituitary masses may disrupt cerebrospinal fluid (CSF) circulation resulting in hydrocephalus.<\/jats:p>\n <\/jats:list-item>\n \n Sellar gangliocytomas are very rare tumours and most of them are hybrid tumours with pituitary adenomas (SGPAs).<\/jats:p>\n <\/jats:list-item>\n \n SGPAs are typically indolent and may be functioning or non-functioning tumours.<\/jats:p>\n <\/jats:list-item>\n \n Growth hormone (GH)-producing SGPAs are less likely to respond to somatostatin agonists than classic somatotrophinomas.<\/jats:p>\n <\/jats:list-item>\n \n Primary surgical debulking via a trans-sphenoidal approach was effective in this individual, leading to the restoration of CSF circulation and improvement in visual disturbance, while also negating the need for permanent CSF diversion despite the residual tumour burden.<\/jats:p>\n <\/jats:list-item>\n <\/jats:list>\n <\/jats:sec>","DOI":"10.1530\/edm-21-0037","type":"journal-article","created":{"date-parts":[[2021,7,8]],"date-time":"2021-07-08T06:07:41Z","timestamp":1625724461000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-2","source":"Crossref","is-referenced-by-count":0,"title":["Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case"],"prefix":"10.1530","volume":"2021","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2694-8612","authenticated-orcid":true,"given":"Simon","family":"Ryder","sequence":"first","affiliation":[{"name":"1Department of Endocrinology and Diabetes, Faculty of Medicine, Royal Brisbane and Women\u2019s Hospital, University of Queensland, Queensland, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jed","family":"Robusto","sequence":"additional","affiliation":[{"name":"2Kenneth J Jamison Neurosurgery Department, Royal Brisbane and Woman\u2019s Hospital, Queensland, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Robertson","sequence":"additional","affiliation":[{"name":"3Pathology Queensland, Faculty of Medicine, Royal Brisbane and Women\u2019s Hospital, University of Queensland, Queensland, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hamish","family":"Alexander","sequence":"additional","affiliation":[{"name":"4Kenneth J Jamison Neurosurgery Department, Faculty of Medicine, Royal Brisbane and Woman\u2019s Hospital, University of Queensland, Queensland, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emma L","family":"Duncan","sequence":"additional","affiliation":[{"name":"5Department of Twin Research and Genetic Epidemiology, Faculty of Life Sciences and Medicine, King\u2019s College London, London, UK"},{"name":"6Faculty of Health and Medicine, Institute of Health and Biomedical Innovation, Queensland University of Technology, University of Queensland, Queensland, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","container-title":["Endocrinology, Diabetes & Metabolism Case Reports"],"original-title":[],"link":[{"URL":"https:\/\/edm.bioscientifica.com\/view\/journals\/edm\/2021\/1\/EDM21-0037.xml","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/edm.bioscientifica.com\/downloadpdf\/journals\/edm\/2021\/1\/EDM21-0037.xml","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T13:47:39Z","timestamp":1784555259000},"score":1,"resource":{"primary":{"URL":"http:\/\/journals.bioscientifica.com\/edm\/article\/3369"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,7,1]]},"references-count":0,"URL":"https:\/\/doi.org\/10.1530\/edm-21-0037","relation":{},"ISSN":["2052-0573"],"issn-type":[{"value":"2052-0573","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,7,1]]}}}