Key result
Calcium, calcitriol, hydrocortisone, and fluconazole resolve refractory seizures in an autoimmune polyendocrine syndrome type 1 case.
Case Report (n=1)
A rare case of APS-1 presenting as refractory epilepsy due to hypocalcemia, which completely resolved with calcium, calcitriol, hydrocortisone, and fluconazole therapy.
Does not support practice change; hypothesis-generating for APS-1 evaluation in refractory seizures.
Autoimmune polyendocrine syndrome type 1 (APS-1) is a rare and complex primary immunodeficiency disorder. The classic clinical triad of APS-1 includes chronic mucocutaneous candidiasis, hypoparathyroidism, and adrenal insufficiency. Clinically, APS-1 presents with significant variability and is characterized by autoimmune dysfunction affecting both endocrine organs (including the parathyroids, adrenal glands, thyroid, gonads, and pituitary) as well as non-endocrine tissues (such as the skin, liver, kidneys, lungs, eyes, and intestines). Here we present a 23-year-old female with a history of abnormal body movement associated with posturing and transient loss of consciousness, along with a history of recurrent oral ulceration, itchy patches over intertriginous areas, and pigmentation of skin. Her examination was suggestive of low blood pressure, oral and cutaneous candidiasis, and hyperpigmentation of the skin. Routine investigations showed very low serum calcium, low parathyroid hormone (PTH) levels, and low early morning cortisol levels, and pathological calcification of the basal ganglia was noted on CT brain. As the patient met the diagnostic criteria of APS-1, she was treated accordingly and responded well.
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Dubey et al. (2025) conducted a case report in Autoimmune polyendocrine syndrome type 1 (APS-1) (n=1). Calcium carbonate, calcitriol, hydrocortisone, and fluconazole was evaluated on Resolution of symptoms (seizures, carpal spasms, and syncopal episodes). Targeted treatment with calcium, calcitriol, hydrocortisone, and fluconazole led to complete resolution of refractory seizures and carpal spasms in a patient with autoimmune polyendocrine syndrome type 1.
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