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A 15-year-old adolescent with a rare pituitary lesion

Cystic sellar lesions are a rare cause of hypopituitarism and extremely rare in the pediatric age group. The differential diagnosis is large and includes both primary pituitary abscesses and cystic components on pre-existing lesions, such as adenoma, craniopharyngioma, Rathke's cleft cyst, leuk...

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Autores principales: Manousaki, Despoina, Deal, Cheri, De Bruycker, Jean Jacques, Ovetchkine, Philippe, Mercier, Claude, Alos, Nathalie
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Bioscientifica Ltd 2014
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4027854/
https://www.ncbi.nlm.nih.gov/pubmed/24851183
http://dx.doi.org/10.1530/EDM-14-0010
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author Manousaki, Despoina
Deal, Cheri
De Bruycker, Jean Jacques
Ovetchkine, Philippe
Mercier, Claude
Alos, Nathalie
author_facet Manousaki, Despoina
Deal, Cheri
De Bruycker, Jean Jacques
Ovetchkine, Philippe
Mercier, Claude
Alos, Nathalie
author_sort Manousaki, Despoina
collection PubMed
description Cystic sellar lesions are a rare cause of hypopituitarism and extremely rare in the pediatric age group. The differential diagnosis is large and includes both primary pituitary abscesses and cystic components on pre-existing lesions, such as adenoma, craniopharyngioma, Rathke's cleft cyst, leukemia, granulomatous disease and lymphocytic hypophysitis. In the absence of a definitive diagnosis, treatment can be challenging. We report a case of a 15-year-old female, who presented with headaches, altered consciousness and diplopia after a molar extraction, for which she had received oral antibiotics. Broad-spectrum i.v. antibiotics were given for presumed meningitis. Blood cultures failed to identify pathogens. Cerebral magnetic resonance imaging showed a pituitary cystic lesion. Endocrine studies revealed abnormal pituitary function. In the absence of a therapeutic response, the patient underwent a transsphenoidal biopsy of the pituitary gland, which yielded a purulent liquid, but cultures were negative. Histopathology showed lymphocytic infiltrates but no neutrophils, compatible with an inflammation of autoimmune or infectious origin. High-dose glucocorticoid therapy was started and pursued, along with i.v. antibiotics, for 6 weeks, leading to clinical and radiological improvement but with persistence of endocrine deficits. In conclusion, this is a case of secondary panhypopituitarism due to a cystic pituitary lesion, with a differential diagnosis of lymphocytic hypophysitis vs abscess in a context of decapitated meningitis. Combination therapy with antibiotics and glucocorticoids is a legitimate approach in the face of diagnostic uncertainty, given the morbidity, and even mortality, associated with these lesions. LEARNING POINTS: It is not always easy to differentiate primary cystic sellar lesions (such as a primary infectious pituitary abscess) from cystic components on pre-existing lesions (such as adenoma, craniopharyngioma, Rathke's cleft cyst, leukemia or lymphocytic hypophysitis). Because of the absence of specific symptoms and of immunohistochemical and serum markers, response to glucocorticoids can be the only way to differentiate lymphocytic hypophysitis from pituitary lesions of another origin. In addition, microbiological cultures are negative in 50% of cases of primary infectious sellar abscesses, thus the response to antibiotic treatment is often the key element to this diagnosis. A short course of high-dose glucocorticoids combined with antibiotics is not harmful in cases where there is no diagnostic certainty as to the origin of a cystic sellar mass, given the morbidity and mortality associated with these lesions. This approach may also diminish inflammation of either infectious or autoimmune origin while ensuring that the most likely pathogens are being targeted.
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spelling pubmed-40278542014-05-21 A 15-year-old adolescent with a rare pituitary lesion Manousaki, Despoina Deal, Cheri De Bruycker, Jean Jacques Ovetchkine, Philippe Mercier, Claude Alos, Nathalie Endocrinol Diabetes Metab Case Rep Novel Treatment Cystic sellar lesions are a rare cause of hypopituitarism and extremely rare in the pediatric age group. The differential diagnosis is large and includes both primary pituitary abscesses and cystic components on pre-existing lesions, such as adenoma, craniopharyngioma, Rathke's cleft cyst, leukemia, granulomatous disease and lymphocytic hypophysitis. In the absence of a definitive diagnosis, treatment can be challenging. We report a case of a 15-year-old female, who presented with headaches, altered consciousness and diplopia after a molar extraction, for which she had received oral antibiotics. Broad-spectrum i.v. antibiotics were given for presumed meningitis. Blood cultures failed to identify pathogens. Cerebral magnetic resonance imaging showed a pituitary cystic lesion. Endocrine studies revealed abnormal pituitary function. In the absence of a therapeutic response, the patient underwent a transsphenoidal biopsy of the pituitary gland, which yielded a purulent liquid, but cultures were negative. Histopathology showed lymphocytic infiltrates but no neutrophils, compatible with an inflammation of autoimmune or infectious origin. High-dose glucocorticoid therapy was started and pursued, along with i.v. antibiotics, for 6 weeks, leading to clinical and radiological improvement but with persistence of endocrine deficits. In conclusion, this is a case of secondary panhypopituitarism due to a cystic pituitary lesion, with a differential diagnosis of lymphocytic hypophysitis vs abscess in a context of decapitated meningitis. Combination therapy with antibiotics and glucocorticoids is a legitimate approach in the face of diagnostic uncertainty, given the morbidity, and even mortality, associated with these lesions. LEARNING POINTS: It is not always easy to differentiate primary cystic sellar lesions (such as a primary infectious pituitary abscess) from cystic components on pre-existing lesions (such as adenoma, craniopharyngioma, Rathke's cleft cyst, leukemia or lymphocytic hypophysitis). Because of the absence of specific symptoms and of immunohistochemical and serum markers, response to glucocorticoids can be the only way to differentiate lymphocytic hypophysitis from pituitary lesions of another origin. In addition, microbiological cultures are negative in 50% of cases of primary infectious sellar abscesses, thus the response to antibiotic treatment is often the key element to this diagnosis. A short course of high-dose glucocorticoids combined with antibiotics is not harmful in cases where there is no diagnostic certainty as to the origin of a cystic sellar mass, given the morbidity and mortality associated with these lesions. This approach may also diminish inflammation of either infectious or autoimmune origin while ensuring that the most likely pathogens are being targeted. Bioscientifica Ltd 2014-05-01 2014 /pmc/articles/PMC4027854/ /pubmed/24851183 http://dx.doi.org/10.1530/EDM-14-0010 Text en © 2014 The authors This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivs 3.0 Unported License (http://creativecommons.org/licenses/by/3.0/deed.en_GB) .
spellingShingle Novel Treatment
Manousaki, Despoina
Deal, Cheri
De Bruycker, Jean Jacques
Ovetchkine, Philippe
Mercier, Claude
Alos, Nathalie
A 15-year-old adolescent with a rare pituitary lesion
title A 15-year-old adolescent with a rare pituitary lesion
title_full A 15-year-old adolescent with a rare pituitary lesion
title_fullStr A 15-year-old adolescent with a rare pituitary lesion
title_full_unstemmed A 15-year-old adolescent with a rare pituitary lesion
title_short A 15-year-old adolescent with a rare pituitary lesion
title_sort 15-year-old adolescent with a rare pituitary lesion
topic Novel Treatment
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4027854/
https://www.ncbi.nlm.nih.gov/pubmed/24851183
http://dx.doi.org/10.1530/EDM-14-0010
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