Title:Adrenocortical Crisis Triggered by Levothyroxine in an Unrecognized Autoimmune Polyglandular Syndrome Type-2: A Case Report with Review of the Literature
Volume: 16
Issue: 1
Author(s): Dhruvkumar M. Patel, Jayanti K. Gurumikhani, Mukundkumar V. Patel*, Maitri M. Patel, Suyog Y. Patel and Prathna N. Patel
Affiliation:
- Department of General Medicine, Zydus Medical College and Hospital, Dahod, Gujarat,India
Keywords:
Autoimmune polyglandular syndrome type-2, autoimmune thyroiditis, hypothyroidism, levothyroxine, addison’s
crisis, adrenocortical insufficiency, Schmidt’s syndrome.
Abstract: Background: Autoimmune polyglandular syndrometype-2 (APS-2) is an uncommon endocrine
disorder of Addison’s disease with an autoimmune thyroid disorder and/or type 1 diabetes
mellitus. The diagnosis is more challenging when a patient presents with nonspecific neuropsychiatric
features with hypothyroidism in the setting of unrecognized Addison’s disease.
Case Report: We report a case of subclinical autoimmune hypothyroidism presented with nonspecific
neuropsychiatric symptoms precipitated by stress. Despite levothyroxine treatment, her symptoms
deteriorated and she was admitted with persistent vomiting and hypovolemic shock. Clinical
features and laboratory parameters were suggestive of underlying adrenocortical insufficiency. Preexisting
autoimmune hypothyroidism combined with Addison's disease confirmed the diagnosis of
unrecognized APS-2. She remarkably improved and her thyroid function tests also normalized with
the treatment of corticosteroids only.
Review of the Literature: We identified only five published case reports of our title by searching
the database. Neufeld and Betterle have reported their data of APS-2 and concluded that a full-
blown clinical picture of two or more components of the syndrome is like the tip of the iceberg.
Conclusion: The patients of one major component of APS-2 should be screened for other components
of the disease to pick up latent cases. Addison’s disease should be ruled out in patients of hypothyroidism
who are intolerant to levothyroxine.
