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A Case of Glucocorticoid Hypersensitivity Syndrome Associated With Underlying Rubella Virus Infection

OBJECTIVE: The objective of this article is to report a rare case of glucocorticoid hypersensitivity syndrome, which may be associated with an underlying rubella virus infection. CASE REPORT: A 29-year-old man showed progressive weight gain for 16 months accompanied by a moon face, enlarged dorsocer...

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Autores principales: Zhang, Zhen, Feng, Ying, Cao, Yang, Chen, Yan, Li, Fangping
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Association of Clinical Endocrinology 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8573315/
https://www.ncbi.nlm.nih.gov/pubmed/34765733
http://dx.doi.org/10.1016/j.aace.2021.06.010
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author Zhang, Zhen
Feng, Ying
Cao, Yang
Chen, Yan
Li, Fangping
author_facet Zhang, Zhen
Feng, Ying
Cao, Yang
Chen, Yan
Li, Fangping
author_sort Zhang, Zhen
collection PubMed
description OBJECTIVE: The objective of this article is to report a rare case of glucocorticoid hypersensitivity syndrome, which may be associated with an underlying rubella virus infection. CASE REPORT: A 29-year-old man showed progressive weight gain for 16 months accompanied by a moon face, enlarged dorsocervical fat pad, central obesity, and purple striae. His cortisol circadian rhythm was normal, and plasma cortisol levels at 8:00 AM fluctuated between 3.2 and 9.54 μg/dL (reference range, 4.3-22.4 μg/dL). A dexamethasone suppression test with a very low dose (0.25 mg) of dexamethasone showed a marked decrease in plasma cortisol level to 0 μg/dL. Adrenal computed tomography and pituitary magnetic resonance imaging findings were normal. The Z-score of the bone density in the lumbar spine was −4.2. The IgM antibody for the rubella virus was positive. His erythrocyte sedimentation rate was 24 mm/hour (reference range, <15 mm/hour), and the C-reactive protein level was 9.22 mg/L (reference range, <5 mg/L). After 3 months, his symptoms resolved spontaneously. The erythrocyte sedimentation rate and C-reactive protein level returned to normal. The IgM antibody for the rubella virus turned negative, whereas the IgG antibody for the rubella virus was positive. DISCUSSION: According to the paradox between clinical manifestations and laboratory tests exogenous Cushing syndrome, cyclical Cushing syndrome, and glucocorticoid hypersensitivity syndrome all should be considered in the diagnosis. Detailed medical history inquiry, complete endocrine hormone testing, and continuous follow-up are all critical for diagnosis. CONCLUSION: Consequently, the patient was diagnosed with glucocorticoid hypersensitivity syndrome. This case illustrates the need to consider the possibility of glucocorticoid hypersensitivity syndrome in a patient who has the manifestations of Cushing syndrome but paradoxical hypocortisolemia, especially after rubella virus infection.
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spelling pubmed-85733152021-11-10 A Case of Glucocorticoid Hypersensitivity Syndrome Associated With Underlying Rubella Virus Infection Zhang, Zhen Feng, Ying Cao, Yang Chen, Yan Li, Fangping AACE Clin Case Rep Case Report OBJECTIVE: The objective of this article is to report a rare case of glucocorticoid hypersensitivity syndrome, which may be associated with an underlying rubella virus infection. CASE REPORT: A 29-year-old man showed progressive weight gain for 16 months accompanied by a moon face, enlarged dorsocervical fat pad, central obesity, and purple striae. His cortisol circadian rhythm was normal, and plasma cortisol levels at 8:00 AM fluctuated between 3.2 and 9.54 μg/dL (reference range, 4.3-22.4 μg/dL). A dexamethasone suppression test with a very low dose (0.25 mg) of dexamethasone showed a marked decrease in plasma cortisol level to 0 μg/dL. Adrenal computed tomography and pituitary magnetic resonance imaging findings were normal. The Z-score of the bone density in the lumbar spine was −4.2. The IgM antibody for the rubella virus was positive. His erythrocyte sedimentation rate was 24 mm/hour (reference range, <15 mm/hour), and the C-reactive protein level was 9.22 mg/L (reference range, <5 mg/L). After 3 months, his symptoms resolved spontaneously. The erythrocyte sedimentation rate and C-reactive protein level returned to normal. The IgM antibody for the rubella virus turned negative, whereas the IgG antibody for the rubella virus was positive. DISCUSSION: According to the paradox between clinical manifestations and laboratory tests exogenous Cushing syndrome, cyclical Cushing syndrome, and glucocorticoid hypersensitivity syndrome all should be considered in the diagnosis. Detailed medical history inquiry, complete endocrine hormone testing, and continuous follow-up are all critical for diagnosis. CONCLUSION: Consequently, the patient was diagnosed with glucocorticoid hypersensitivity syndrome. This case illustrates the need to consider the possibility of glucocorticoid hypersensitivity syndrome in a patient who has the manifestations of Cushing syndrome but paradoxical hypocortisolemia, especially after rubella virus infection. American Association of Clinical Endocrinology 2021-06-23 /pmc/articles/PMC8573315/ /pubmed/34765733 http://dx.doi.org/10.1016/j.aace.2021.06.010 Text en © 2021 Published by Elsevier Inc. on behalf of the AACE. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Zhang, Zhen
Feng, Ying
Cao, Yang
Chen, Yan
Li, Fangping
A Case of Glucocorticoid Hypersensitivity Syndrome Associated With Underlying Rubella Virus Infection
title A Case of Glucocorticoid Hypersensitivity Syndrome Associated With Underlying Rubella Virus Infection
title_full A Case of Glucocorticoid Hypersensitivity Syndrome Associated With Underlying Rubella Virus Infection
title_fullStr A Case of Glucocorticoid Hypersensitivity Syndrome Associated With Underlying Rubella Virus Infection
title_full_unstemmed A Case of Glucocorticoid Hypersensitivity Syndrome Associated With Underlying Rubella Virus Infection
title_short A Case of Glucocorticoid Hypersensitivity Syndrome Associated With Underlying Rubella Virus Infection
title_sort case of glucocorticoid hypersensitivity syndrome associated with underlying rubella virus infection
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8573315/
https://www.ncbi.nlm.nih.gov/pubmed/34765733
http://dx.doi.org/10.1016/j.aace.2021.06.010
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