https://m2.mtmt.hu/hun2024-03-29 11:33JournalArticle30398712VALIDATEDtrueDepartment of Internal Medicine, Shirakawa Kosei General Hospital, 2-1 Toyochi Kamiyajirou, Shirakawa, Fukushima, 961-0005, Japan
Department of Diabetes, Endocrinology and Metabolism School of Medicine, Fukushima Medical University, Fukushima City, Fukushima, Japan
Export Date: 30 July 2019
CODEN: MEDIA
Correspondence Address: Hirai, H.; Department of Internal Medicine, Shirakawa Kosei General Hospital, 2-1 Toyochi Kamiyajirou, Japan; email: hiroyuki@fmu.ac.jp
Department of Internal Medicine, Shirakawa Kosei General Hospital, 2-1 Toyochi Kamiyajirou, Shirakawa, Fukushima, 961-0005, Japan
Department of Diabetes, Endocrinology and Metabolism School of Medicine, Fukushima Medical University, Fukushima City, Fukushima, Japan
Export Date: 28 March 2021
CODEN: MEDIA
Correspondence Address: Hirai, H.; Department of Internal Medicine, 2-1 Toyochi Kamiyajirou, Japan; email: hiroyuki@fmu.ac.jp
Chemicals/CAS: hydrocortisone, 50-23-7; prednisolone, 50-24-8; steroid 21 monooxygenase, 9029-68-9; corticotropin, 11136-52-0, 9002-60-2, 9061-27-2; hydroxyprogesterone, 68-96-2; 17-alpha-Hydroxyprogesterone; Adrenocorticotropic Hormone; CYP21A2 protein, human; Hydrocortisone; Steroid 21-Hydroxylase0false2021-03-28T11:22:29.535+00002019-02-24T07:22:39.741+00002019-01-17T20:57:58.178+0000true10015206ÁncsánGizella
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REVIEWEDtruefalse3178false3178true0025-79741536-5964JournalFOREIGN9733e11888e1188842018Rationale:Adrenal incidentaloma is sometimes complicated with 21-hydroxylase deficiency (21-OHD). Latent nonclassical 21-OHD in incidentaloma is difficult to diagnose. Although adrenalectomy in 21-OHD has been conducted when malignancy could not be excluded, adrenal insufficiency sometimes occurs, and it might not be observed immediately after operation. Here, we report a case of a 71-year-old man who experienced adrenal insufficiency over 2 decades postadrenalectomy, leading to a diagnosis of latent nonclassical 21-OHD.Patient concerns:A 71-year-old man was admitted to the hospital due to difficulty in movements and a sodium level of 119mEq/L. His medical history revealed precocious puberty and left adrenalectomy because of an incidentaloma at 49 years of age, diagnosed pathologically as an adenoma. He did not attend follow-up visits because he did not have any symptoms. In 2017, 3 months before hospitalization, he experienced general fatigue. A few days before admittance, he complained of difficulty in moving and visual hallucination of small animals.Diagnoses:Laboratory evaluations revealed a high level of adrenocorticotropic hormone (ACTH) and low cortisol level. ACTH-stimulating test revealed a low basal level and low response for cortisol, and a high basal level and low response for 17-hydroxyprogesterone. We analyzed large gene deletion or conversion and the 9 most common micro mutations in the CYP21A2 gene by polymerase chain reaction; micro mutation of I172N and heterozygous large gene deletion or conversion were detected leading to the diagnosis of nonclassical 21-OHD.Interventions:Immediately, 100mg hydrocortisone was administered, followed by daily hydrocortisone and saline. The symptoms and hyponatremia improved in a few days. He was discharged from the hospital on day 34 with a daily dose of 15mg hydrocortisone.Lessons:Clinicians should be aware of late onset of adrenal insufficiency after adrenalectomy. In such cases, clinicians should not overlook the latent nonclassical 21-OHD.2019truetruetruefalsefalseGOLD2021-03-28falsehttp://journals.lww.com/md-journal/toc/1922/1100000000000000011Q2falsefalsefalsefalse2019-04-17T21:33:50.727+00002019-01-17T22:33:50.827+0000821030trueLanguage10002
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<div class="JournalArticle Publication short-list"> <div class="authors"> <span class="author-name" > Hirai, Hiroyuki </span> <span class="author-type"> </span> ; <span class="author-name" > Kuwana, Keisuke </span> <span class="author-type"> </span> ; <span class="author-name" > Kusano, Yoshiro </span> <span class="author-type"> </span> </div ><div class="title"><a href="/gui2/?mode=browse¶ms=publication;30398712" mtid="30398712" target="_blank">Late onset adrenal insufficiency after adrenalectomy due to latent nonclassical 21-hydroxylase deficiency: A case report</a></div> <div class="pub-info"> <span class="journal-title">MEDICINE</span> <span class="journal-volume">97</span> : <span class="journal-issue">33</span> <span class="page"> Paper: e11888 , 4 p. </span> <span class="year">(2018)</span> </div> <div class="pub-end"><div class="identifier-list"> <span class="identifiers"> <span class="id identifier oa_none" title="none"> <a style="color:blue" title="10.1097/MD.0000000000011888" target="_blank" href="https://doi.org/10.1097%2FMD.0000000000011888"> DOI </a> </span> <span class="id identifier oa_none" title="none"> <a style="color:blue" title="000442255600036" target="_blank" href="http://gateway.isiknowledge.com/gateway/Gateway.cgi?&GWVersion=2&SrcAuth=CustomerName&SrcApp=CustomerName&DestLinkType=FullRecord&KeyUT=000442255600036&DestApp=WOS"> WoS </a> </span> <span class="id identifier oa_none" title="none"> <a style="color:blue" title="85052169968" target="_blank" href="http://www.scopus.com/record/display.url?origin=inward&eid=2-s2.0-85052169968"> Scopus </a> </span> <span class="id identifier oa_none" title="none"> <a style="color:blue" title="30113485" target="_blank" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=Retrieve&db=PubMed&list_uids=30113485&dopt=Abstract"> PubMed </a> </span> </span> </div> <span class="short-pub-prop-list"> <span class="short-pub-mtid"> Közlemény:30398712 </span> <span class="status-holder"><span class="status-data status-VALIDATED"> Egyeztetett </span></span> <span class="pub-core"> Idéző </span> <span class="pub-type">Folyóiratcikk (Szakcikk ) </span> <!-- && !record.category.scientific --> <span class="pub-category">Tudományos</span> </span> </div> </div><div class="JournalArticle Publication long-list">
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<div class="autype autype0"> <span class="author-name" >Hirai Hiroyuki
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<span class="author-name" >Kuwana Keisuke
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<span class="author-name" >Kusano Yoshiro
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<div class="title"><a href="/gui2/?mode=browse¶ms=publication;30398712" target="_blank">Late onset adrenal insufficiency after adrenalectomy due to latent nonclassical 21-hydroxylase deficiency: A case report</a></div> <div> <span class="journal-title">MEDICINE</span>
<span class="journal-issn">(<a target="_blank" href="https://portal.issn.org/resource/ISSN/0025-7974">0025-7974</a> <a target="_blank" href="https://portal.issn.org/resource/ISSN/1536-5964">1536-5964</a>)</span>:
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Paper e11888.
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<pre class="comment" style="margin-top: 0; margin-bottom: 0;"><u>Megjegyzés</u>: Department of Internal Medicine, Shirakawa Kosei General Hospital, 2-1 Toyochi Kamiyajirou, Shirakawa, Fukushima, 961-0005, Japan
Department of Diabetes, Endocrinology and Metabolism School of Medicine, Fukushima Medical University, Fukushima City, Fukushima, Japan
Export Date: 30 July 2019
CODEN: MEDIA
...</pre>
</div></div>