Primary Adrenal Insufficiency due to Cryptococcus With Persistent Adrenal Enlargement and Insufficiency

Q3 Medicine
Catherine E. Price MD, Cynthia Burns MD, Joseph A. Aloi MD
{"title":"Primary Adrenal Insufficiency due to Cryptococcus With Persistent Adrenal Enlargement and Insufficiency","authors":"Catherine E. Price MD,&nbsp;Cynthia Burns MD,&nbsp;Joseph A. Aloi MD","doi":"10.1016/j.aace.2023.09.002","DOIUrl":null,"url":null,"abstract":"<div><h3>Background/Objective</h3><p>Infiltrative fungal infections are an unusual cause of primary adrenal insufficiency (AI). Our objective is to present a long-term follow-up of a patient with AI due to cryptococcal adrenalitis.</p></div><div><h3>Case Report</h3><p>A 47-year-old woman presented in January 2004, with 50-lb weight loss, nausea, emesis, and headache with diplopia. During the 6 months prior to her presentation the patient had multiple admissions for evaluation of recurrent nausea and emesis. Prior to the most recent of these admissions, the patient developed a headache; evaluation of her cerebrospinal fluid revealed the presence of <em>Cryptococcus</em>, and she was treated with a 2-week course of amphotericin B. Physical examination demonstrated a temperature of 101.1 °F, heart rate of 110 bpm, and blood pressure of 94/65 mm Hg. She appeared ill and was underweight with dry mucous membranes and photophobia. Laboratory tests revealed random cortisol of 0.5 μg per dL. CT imaging showed bilateral adrenal gland enlargement and fine needle aspiration of the adrenal gland revealed encapsulated budding yeast. Stress dose intravenous glucocorticoids were administered and switched to oral hydrocortisone and fludrocortisone because the patient clinically improved with a second course of amphotericin B. Further evaluation in 2017 revealed persistently enlarged adrenal glands, positive cryptococcus antigen, and low IgG levels.</p></div><div><h3>Discussion</h3><p>Our literature review noted few publications of AI caused by disseminated cryptococcus with no long-term follow-up of these cases beyond a 1- to 4-year time frame.</p></div><div><h3>Conclusion</h3><p>Patients with AI due to disseminated fungal infection need long-term follow-up to assess for resolution of adrenal enlargement and evaluation of immunocompromised status.</p></div>","PeriodicalId":7051,"journal":{"name":"AACE Clinical Case Reports","volume":"9 6","pages":"Pages 189-192"},"PeriodicalIF":0.0000,"publicationDate":"2023-11-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.sciencedirect.com/science/article/pii/S2376060523001384/pdfft?md5=acdf28154fce382b5bdc781979bb5c2a&pid=1-s2.0-S2376060523001384-main.pdf","citationCount":"1","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"AACE Clinical Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S2376060523001384","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}
引用次数: 1

Abstract

Background/Objective

Infiltrative fungal infections are an unusual cause of primary adrenal insufficiency (AI). Our objective is to present a long-term follow-up of a patient with AI due to cryptococcal adrenalitis.

Case Report

A 47-year-old woman presented in January 2004, with 50-lb weight loss, nausea, emesis, and headache with diplopia. During the 6 months prior to her presentation the patient had multiple admissions for evaluation of recurrent nausea and emesis. Prior to the most recent of these admissions, the patient developed a headache; evaluation of her cerebrospinal fluid revealed the presence of Cryptococcus, and she was treated with a 2-week course of amphotericin B. Physical examination demonstrated a temperature of 101.1 °F, heart rate of 110 bpm, and blood pressure of 94/65 mm Hg. She appeared ill and was underweight with dry mucous membranes and photophobia. Laboratory tests revealed random cortisol of 0.5 μg per dL. CT imaging showed bilateral adrenal gland enlargement and fine needle aspiration of the adrenal gland revealed encapsulated budding yeast. Stress dose intravenous glucocorticoids were administered and switched to oral hydrocortisone and fludrocortisone because the patient clinically improved with a second course of amphotericin B. Further evaluation in 2017 revealed persistently enlarged adrenal glands, positive cryptococcus antigen, and low IgG levels.

Discussion

Our literature review noted few publications of AI caused by disseminated cryptococcus with no long-term follow-up of these cases beyond a 1- to 4-year time frame.

Conclusion

Patients with AI due to disseminated fungal infection need long-term follow-up to assess for resolution of adrenal enlargement and evaluation of immunocompromised status.

隐球菌所致原发性肾上腺功能不全伴持续肾上腺肿大和功能不全
背景/目的浸润性真菌感染是原发性肾上腺功能不全(AI)的罕见病因。我们的目的是对一例隐球菌性肾上腺炎所致AI患者进行长期随访。病例报告:一名47岁女性,2004年1月出现体重减轻50磅,恶心,呕吐,头痛伴复视。在她就诊前的6个月,患者多次入院评估复发性恶心和呕吐。在最近一次入院之前,患者出现头痛;脑脊液检查发现隐球菌,并给予两周两性霉素b疗程。体格检查显示体温101.1°F,心率110 bpm,血压94/65 mmHg,体重过轻,黏膜干燥,畏光。实验室检测随机皮质醇0.5 ug/dL。CT示双侧肾上腺肿大,肾上腺FNA示包被芽孢酵母。患者给予应激剂量静脉注射糖皮质激素后,经第二疗程两性霉素b治疗后临床好转,改为口服氢化可的松和氟化可的松。2017年进一步评估发现肾上腺持续增大,隐球菌抗原阳性,IgG水平低。我们的文献综述注意到很少有关于播散性隐球菌引起的AI的出版物,这些病例没有超过1-4年的长期随访。结论弥散性真菌感染所致AI患者需要长期随访,以评估肾上腺肿大的消退情况和免疫功能低下状况。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 求助全文
来源期刊
AACE Clinical Case Reports
AACE Clinical Case Reports Medicine-Endocrinology, Diabetes and Metabolism
CiteScore
2.30
自引率
0.00%
发文量
61
审稿时长
55 days
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信