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Plurihormonal Pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue

A 21 year-old woman was found to have a pituitary macroadenoma following an episode of haemophilus meningitis. Biochemical TSH and GH excess was noted, although with no clear clinical correlates. She was treated with a somatostatin analogue (SSA), which restored the euthyroid state and controlled GH...

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Autores principales: Olmedilla, Yoko, Khan, Shoaib, Young, Victoria, Joseph, Robin, Cudlip, Simon, Ansgorge, Olaf, Grossman, Ashley, Pal, Aparna
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Bioscientifica Ltd 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6432976/
https://www.ncbi.nlm.nih.gov/pubmed/30897548
http://dx.doi.org/10.1530/EDM-18-0130
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author Olmedilla, Yoko
Khan, Shoaib
Young, Victoria
Joseph, Robin
Cudlip, Simon
Ansgorge, Olaf
Grossman, Ashley
Pal, Aparna
author_facet Olmedilla, Yoko
Khan, Shoaib
Young, Victoria
Joseph, Robin
Cudlip, Simon
Ansgorge, Olaf
Grossman, Ashley
Pal, Aparna
author_sort Olmedilla, Yoko
collection PubMed
description A 21 year-old woman was found to have a pituitary macroadenoma following an episode of haemophilus meningitis. Biochemical TSH and GH excess was noted, although with no clear clinical correlates. She was treated with a somatostatin analogue (SSA), which restored the euthyroid state and controlled GH hypersecretion, but she re-presented with a further episode of cerebrospinal fluid (CSF) leak and recurrent meningitis. Histology following transsphenoidal adenomectomy revealed a Pit-1 lineage plurihormonal adenoma expressing GH, TSH and PRL. Such plurihormonal pituitary tumours are uncommon and even more unusual to present with spontaneous bacterial meningitis. The second episode of CSF leak and meningitis appears to have been due to SSA therapy-induced tumour shrinkage, which is not a well-described phenomenon in the literature for this type of tumour. LEARNING POINTS: Pit-1 lineage GH/TSH/PRL-expressing plurihormonal pituitary adenomas are uncommon. Moreover, this case is unique as the patient first presented with bacterial meningitis. Inmunohistochemical plurihormonality of pituitary adenomas does not necessarily correlate with biochemical and clinical features of hormonal hypersecretion. Given that plurihormonal Pit-1 lineage adenomas may behave more aggressively than classical pituitary adenomas, accurate pathological characterization of these tumours has an increasing prognostic relevance. Although unusual, a CSF leak and meningitis may be precipitated by SSA therapy of a pituitary macroadenoma via tumour shrinkage.
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spelling pubmed-64329762019-03-27 Plurihormonal Pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue Olmedilla, Yoko Khan, Shoaib Young, Victoria Joseph, Robin Cudlip, Simon Ansgorge, Olaf Grossman, Ashley Pal, Aparna Endocrinol Diabetes Metab Case Rep Unique/Unexpected Symptoms or Presentations of a Disease A 21 year-old woman was found to have a pituitary macroadenoma following an episode of haemophilus meningitis. Biochemical TSH and GH excess was noted, although with no clear clinical correlates. She was treated with a somatostatin analogue (SSA), which restored the euthyroid state and controlled GH hypersecretion, but she re-presented with a further episode of cerebrospinal fluid (CSF) leak and recurrent meningitis. Histology following transsphenoidal adenomectomy revealed a Pit-1 lineage plurihormonal adenoma expressing GH, TSH and PRL. Such plurihormonal pituitary tumours are uncommon and even more unusual to present with spontaneous bacterial meningitis. The second episode of CSF leak and meningitis appears to have been due to SSA therapy-induced tumour shrinkage, which is not a well-described phenomenon in the literature for this type of tumour. LEARNING POINTS: Pit-1 lineage GH/TSH/PRL-expressing plurihormonal pituitary adenomas are uncommon. Moreover, this case is unique as the patient first presented with bacterial meningitis. Inmunohistochemical plurihormonality of pituitary adenomas does not necessarily correlate with biochemical and clinical features of hormonal hypersecretion. Given that plurihormonal Pit-1 lineage adenomas may behave more aggressively than classical pituitary adenomas, accurate pathological characterization of these tumours has an increasing prognostic relevance. Although unusual, a CSF leak and meningitis may be precipitated by SSA therapy of a pituitary macroadenoma via tumour shrinkage. Bioscientifica Ltd 2019-03-21 /pmc/articles/PMC6432976/ /pubmed/30897548 http://dx.doi.org/10.1530/EDM-18-0130 Text en © 2019 The authors http://creativecommons.org/licenses/by-nc-nd/4.0/ This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. (http://creativecommons.org/licenses/by-nc-nd/4.0/) .
spellingShingle Unique/Unexpected Symptoms or Presentations of a Disease
Olmedilla, Yoko
Khan, Shoaib
Young, Victoria
Joseph, Robin
Cudlip, Simon
Ansgorge, Olaf
Grossman, Ashley
Pal, Aparna
Plurihormonal Pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue
title Plurihormonal Pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue
title_full Plurihormonal Pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue
title_fullStr Plurihormonal Pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue
title_full_unstemmed Plurihormonal Pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue
title_short Plurihormonal Pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue
title_sort plurihormonal pit-1 lineage adenoma presenting as meningitis with recurrence after somatostatin analogue
topic Unique/Unexpected Symptoms or Presentations of a Disease
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6432976/
https://www.ncbi.nlm.nih.gov/pubmed/30897548
http://dx.doi.org/10.1530/EDM-18-0130
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