Reversible primary adrenal insufficiency related to anti-programmed cell-death 1 protein active immunotherapy: Insight into an unforeseen outcome of a rare immune-related adverse event

Deligiorgi, Maria V; Trafalis, Dimitrios T.

doi:10.1016/j.intimp.2020.107050

Cited by 7 publications

(3 citation statements)

References 45 publications

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“…Twenty-seven studies with detailed patients’ features have been published from 2013 to 2022 and are included in Table 1 [ 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 ]. These studies describe a total of 29 patients who experienced PAI following ICI therapy for various types of malignancy.…”

Section: Literature Review Resultsmentioning

confidence: 99%

“…The majority of these reported cases (20/27, 74.1%) presented with severe or life-threatening symptoms (grade III/IV). Follow-up data were available for only nine patients from whom the vast majority (7/9, 77.8%) presented permanent PAI requiring lifelong steroid substitution, whereas only two cases experienced remission of their PAI confirmed with physiological response of cortisol in the synacthen test [ 26 , 34 ].…”

Section: Literature Review Resultsmentioning

confidence: 99%

“…The assessment of antibodies was inconsistently reported throughout the studies. Six cases out of 10 [ 13 , 17 , 23 , 26 , 28 , 39 ] presented positive 21-hydroxylase antibodies (21-OHAbs), while antibodies were negative in four cases [ 20 , 29 , 35 ]. The 21-OHAbs and adrenal cortex antibodies (ACA) were both positive in two cases [ 13 , 23 ].…”

Section: Literature Review Resultsmentioning

confidence: 99%

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The Uncharted Landscape of Rare Endocrine Immune-Related Adverse Events

Mytareli

Ziogas

Karampela

et al. 2023

Cancers

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Immune checkpoint inhibitors (ICIs) have been approved for the treatment of many cancers, either in adjuvant or metastatic settings. Regarding safety, endocrine adverse events (AEs) are some of the most common AEs in ICI-treated patients, with thyroid dysfunction and hypophysitis being the most frequent disorders. However, there are also some rare and very rare immune-related (ir) endocrine complications (incidence between ≥1/10,000 to <1/1000 and <1/10,000, respectively, according to the established classification) that have been reported in isolated case reports, with limited data about their management. In this systematic review, we summarize all published cases with primary adrenal insufficiency, central diabetes insipidus, primary hypoparathyroidism, lipodystrophy, osteoporosis, hypergonadotrophic hypogonadism, or Cushing disease and discuss their diagnostic and therapeutic approaches as well as the current knowledge on their pathophysiology. In these ICI-treated cancer patients, the presentation of symptoms unrelated to their underlying malignancy has led to further diagnostic tests, including hormonal profile and functional assays which subsequently confirmed endocrinopathy, while the assessment of autoantibodies was rarely available. In most of these cases, the exact pathogenesis remained unknown, and the endocrine dysfunction was permanent, requiring lifelong supplementation. Although endrocine irAEs are rare, physicians must be aware of these irAEs to recognize them on time and treat them appropriately.

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