We report on the case of a patient with dysgerminoma, a rare germ cell tumor, which showed hypercalcemia with an elevation of 1α,25-dihydroxycholecalciferol (calcitriol). A 27-year-old nulliparous woman presented with hypercalcemia during the examination of a right ovarian tumor with an elevation of calcitriol, lactate dehydrogenase, and alkaline phosphatase. Fractional excretion of calcium was elevated, and intact parathyroid hormone was suppressed. After undergoing right salpingo-oophorectomy, the patient’s serum calcium and calcitriol returned to the normal range within a week. A literature search was conducted on the topic by reviewing databases for dysgerminoma showing hypercalcemia. We identified 14 patients from the literature and performed a pooled analysis, including the results of our case. However, most cases lack data that can help investigate the potential association between parathyroid hormone, parathyroid hormone-related protein, calcitriol, and phosphorus in hypercalcemia. Thus, more case reports that include additional information are required to fully elucidate the mechanism of hypercalcemia associated with dysgerminoma.
Purpose Immune checkpoint inhibitor (ICI)-induced type 1 diabetes and pituitary dysfunction are life-threatening adverse events, yet there is little clinical data available. We aimed to investigate the clinical characteristics of patients with these adverse events and report their human leukocyte antigen (HLA) profile to determine its relevance. Methods This is a single-center prospective study. We collected clinical and biochemical data and extracted DNA from blood samples. HLA typing was performed using next-generation sequencing. We compared our results with those previously reported in healthy controls and investigated the correlation between HLA and the occurrence of ICI-induced type 1 diabetes and pituitary dysfunction. Results We identified 914 patients treated with ICI in our facility from 1st September, 2017 to 30th June, 2022. Six of these patients developed type 1 diabetes and 14 developed pituitary dysfunction. The duration from the initiation of ICI treatment to the onset of type 1 diabetes or pituitary dysfunction averaged 492 ± 196 days and 191 ± 169 days, respectively. Among the six patients with type 1 diabetes, two were positive for anti-GAD antibody. The frequencies of HLA-DR11, -Cw10, -B61, -DRB1*11:01, and -C*03:04 were significantly higher in patients with ICI-induced type 1 diabetes than in controls. The frequencies of HLA-DR15 and -DRB*15:02 were significantly higher in patients with ICI-induced pituitary dysfunction than in controls. Conclusion This study revealed the clinical characteristics of type 1 diabetes and pituitary dysfunction induced by ICI and the association between specific HLAs and these adverse events.
Immune checkpoint inhibitor (ICI)-induced type 1 diabetes and pituitary dysfunction are life-threatening adverse events, yet there is little clinical data available. We aimed to investigate the clinical characteristics of patients with these adverse events and report their human leukocyte antigen (HLA) pro le to determine its relevance. MethodsThis is a single-center prospective study. We collected clinical and biochemical data and extracted DNA from blood samples. HLA typing was performed using next-generation sequencing. We compared our results with those previously reported in healthy controls and investigated the correlation between HLA and the occurrence of ICI-induced type 1 diabetes and pituitary dysfunction. ResultsWe identi ed 914 patients treated with ICI in our facility from 1st September, 2017 to 30th June, 2022. Six of these patients developed type 1 diabetes and 14 developed pituitary dysfunction. The duration from the initiation of ICI treatment to the onset of type 1 diabetes or pituitary dysfunction averaged 492 ± 196 days and 191 ± 169 days, respectively. Among the six patients with type 1 diabetes, two were positive for anti-GAD antibody. The frequencies of HLA-DR11, -Cw10, -B61, -DRB1*11:01, and -C*03:04 were signi cantly higher in patients with ICI-induced type 1 diabetes than in controls. The frequencies of HLA-DR15 and -DRB*15:02 were signi cantly higher in patients with ICI-induced pituitary dysfunction than in controls. ConclusionThis study revealed the clinical characteristics of type 1 diabetes and pituitary dysfunction induced by ICI and the association between speci c HLAs and these adverse events.
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