Literature DB >> 31782110

Fatal adrenal crisis due to Addison's disease arising in the context of autoimmune polyglandular syndrome type 1.

Kran Suknuntha1,2,3, Qiqi Yu3, Paul S Weisman3, Robert F Corliss4.   

Abstract

The autoimmune polyglandular syndromes (APS) are rare immune-mediated endocrinopathies causing destruction of multiple endocrine and non-endocrine organs. Involvement of adrenal glands associated with any type of APS results in Addison's disease. While patients with Addison's disease often suffer from symptoms of neuroglycopenia, lethal hypotension and hypoglycemia are uncommon. Here, we report a fatal case of APS type 1 with hypotension and profound hypoglycemia in a 24-year-old man who was found unconsciousness at home and progressively evolved into pulseless electrical activity. Although his condition was initially considered to be possibly due to drug toxicity, subsequent drug screening tests failed to detect alcohol or any other substances. Emergent medical evaluation revealed severe hypotension (51/30 mm/Hg) and profound hypoglycemia (blood glucose of 20-30 mg/dl). Despite vigorous supportive care, the patient died following 3 days of respiratory dependency due to irreversible anoxic brain injury. Postmortem examination revealed severely atrophic adrenal glands with lymphocytic infiltration. Subsequent review of the patient's medical history and correlation with autopsy findings confirmed the presence of multiple organ involvement, consistent with APS type 1. This case serves as a reminder for forensic pathologists that death from an acute adrenal (Addisonian) crisis, while uncommon, should remain a differential diagnostic consideration. Furthermore, it underscores the fact that Addison's disease can occur as part of a constellation of autoimmune manifestations within the context of an underlying polyglandular syndrome, such as APS type 1.

Entities:  

Keywords:  APS; Addison; Adrenal insufficiency; Arrhythmia; Autoimmune polyglandular syndromes; Hypoglycemia

Mesh:

Year:  2019        PMID: 31782110     DOI: 10.1007/s12024-019-00204-4

Source DB:  PubMed          Journal:  Forensic Sci Med Pathol        ISSN: 1547-769X            Impact factor:   2.007


  8 in total

1.  Nocturnal hypoglycemia identified by a continuous glucose monitoring system in patients with primary adrenal insufficiency (Addison's Disease).

Authors:  Gesine Meyer; Annika Hackemann; Juergen Reusch; Klaus Badenhoop
Journal:  Diabetes Technol Ther       Date:  2012-01-13       Impact factor: 6.118

Review 2.  Clinical manifestations and management of patients with autoimmune polyendocrine syndrome type I.

Authors:  E S Husebye; J Perheentupa; R Rautemaa; O Kämpe
Journal:  J Intern Med       Date:  2009-05       Impact factor: 8.989

3.  Vital signs, QT prolongation, and newly diagnosed cardiovascular disease during severe hypoglycemia in type 1 and type 2 diabetic patients.

Authors:  Tetsuro Tsujimoto; Ritsuko Yamamoto-Honda; Hiroshi Kajio; Miyako Kishimoto; Hiroshi Noto; Remi Hachiya; Akio Kimura; Masafumi Kakei; Mitsuhiko Noda
Journal:  Diabetes Care       Date:  2013-08-12       Impact factor: 19.112

4.  Polyglandular autoimmune syndrome type I - a novel AIRE mutation in a North American patient.

Authors:  Kelly Egan Huibregtse; Peter Wolfgram; Karen K Winer; Ellen L Connor
Journal:  J Pediatr Endocrinol Metab       Date:  2014-11       Impact factor: 1.634

5.  QT interval prolongation during spontaneous episodes of hypoglycaemia in type 1 diabetes: the impact of heart rate correction.

Authors:  T F Christensen; L Tarnow; J Randløv; L E Kristensen; J J Struijk; E Eldrup; O K Hejlesen
Journal:  Diabetologia       Date:  2010-05-23       Impact factor: 10.122

6.  QTc interval prolongation is independently associated with severe hypoglycemic attacks in type 1 diabetes from the EURODIAB IDDM complications study.

Authors:  Gabriella Gruden; Sara Giunti; Federica Barutta; Nish Chaturvedi; Daniel R Witte; Marinella Tricarico; John H Fuller; Paolo Cavallo Perin; Graziella Bruno
Journal:  Diabetes Care       Date:  2011-11-28       Impact factor: 19.112

7.  Severe hypoglycemia-induced lethal cardiac arrhythmias are mediated by sympathoadrenal activation.

Authors:  Candace M Reno; Dorit Daphna-Iken; Y Stefanie Chen; Jennifer VanderWeele; Krishan Jethi; Simon J Fisher
Journal:  Diabetes       Date:  2013-07-08       Impact factor: 9.461

8.  A Longitudinal Follow-up of Autoimmune Polyendocrine Syndrome Type 1.

Authors:  Øyvind Bruserud; Bergithe E Oftedal; Nils Landegren; Martina M Erichsen; Eirik Bratland; Kari Lima; Anders P Jørgensen; Anne G Myhre; Johan Svartberg; Kristian J Fougner; Åsne Bakke; Bjørn G Nedrebø; Bjarne Mella; Lars Breivik; Marte K Viken; Per M Knappskog; Mihaela C Marthinussen; Kristian Løvås; Olle Kämpe; Anette B Wolff; Eystein S Husebye
Journal:  J Clin Endocrinol Metab       Date:  2016-06-02       Impact factor: 5.958

  8 in total
  1 in total

Review 1.  Adrenal crises in adolescents and young adults.

Authors:  R Louise Rushworth; Georgina L Chrisp; Suzannah Bownes; David J Torpy; Henrik Falhammar
Journal:  Endocrine       Date:  2022-05-18       Impact factor: 3.925

  1 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.