Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case
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 unusua...
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Bioscientifica
2021-07-01
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Series: | Endocrinology, Diabetes & Metabolism Case Reports |
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doaj-928e4f3e024746e8bd942553452df1482021-07-09T04:44:31ZengBioscientificaEndocrinology, Diabetes & Metabolism Case Reports2052-05732052-05732021-07-01111510.1530/EDM-21-0037Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported caseSimon Ryder0Jed Robusto1Thomas Robertson2Hamish Alexander3Emma L Duncan4Department of Endocrinology and Diabetes, Faculty of Medicine, Royal Brisbane and Women’s Hospital, University of Queensland, Queensland, AustraliaKenneth J Jamison Neurosurgery Department, Royal Brisbane and Woman’s Hospital, Queensland, AustraliaPathology Queensland, Faculty of Medicine, Royal Brisbane and Women’s Hospital, University of Queensland, Queensland, AustraliaKenneth J Jamison Neurosurgery Department, Faculty of Medicine, Royal Brisbane and Woman’s Hospital, University of Queensland, Queensland, AustraliaDepartment of Twin Research and Genetic Epidemiology, Faculty of Life Sciences and Medicine, King’s College London, London, UK; Faculty of Health and Medicine, Institute of Health and Biomedical Innovation, Queensland University of Technology, University of Queensland, Queensland, AustraliaAlthough 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.https://edm.bioscientifica.com/view/journals/edm/2021/1/EDM21-0037.xml |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Simon Ryder Jed Robusto Thomas Robertson Hamish Alexander Emma L Duncan |
spellingShingle |
Simon Ryder Jed Robusto Thomas Robertson Hamish Alexander Emma L Duncan Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case Endocrinology, Diabetes & Metabolism Case Reports |
author_facet |
Simon Ryder Jed Robusto Thomas Robertson Hamish Alexander Emma L Duncan |
author_sort |
Simon Ryder |
title |
Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case |
title_short |
Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case |
title_full |
Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case |
title_fullStr |
Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case |
title_full_unstemmed |
Unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case |
title_sort |
unilateral hydrocephalus from a gangliocytoma-somatotrophinoma: first reported case |
publisher |
Bioscientifica |
series |
Endocrinology, Diabetes & Metabolism Case Reports |
issn |
2052-0573 2052-0573 |
publishDate |
2021-07-01 |
description |
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. |
url |
https://edm.bioscientifica.com/view/journals/edm/2021/1/EDM21-0037.xml |
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