ABSTRACT
Objectives From the beginning of the COVID-19 pandemic, parents have been exposed to multiple alternative over-the-counter remedies, claiming low risk and effectiveness in preventing or combating COVID-19 illness. These remedies lack robust safety and efficacy data, are frequently unregulated lacking dosing consistency and are usually encouraged without physician supervision. The objective of this care is to raise awareness about the risks from vitamin D over-supplementation for COVID-19 prophylaxis and to outline the successful treatment with Pamidronate of hypercalcemia from vitamin D toxicity. Methods A previously healthy 2-year-old male, presented with one week of intermittent vomiting, constipation, polyuria, and fatigue after receiving daily 3000 IU liquid vitamin D3 and a multivitamin for the past year for COVID-19 prophylaxis. He had elevated serum calcium (17.6 mg/dL), ionized calcium (2.40 mmol/L), lipase (2386 U/L), 25-hydroxyvitamin D (>120 ng/mL) and 1,25-dihydroxyvitamin D (154.0 pg/mL), with low intact PTH (3.3 pg/mL), marked hypercalciuria (3250 mg/g Cr) and nephrocalcinosis. He was diagnosed with hypercalcemia and pancreatitis from Vitamin D toxicity that was only mildly responsive to hydration, furosemide, calcitonin, and calcium restriction. Results One dose of the bisphosphonate pamidronate (2mg/kg) led to the gradual normalization of his calcium within 3 days on a low calcium diet. Vitamin D 25 OH levels remain high (>120 ng/mL) six months later with normal calcium. Conclusions The consistent daily intake of over 3000 IU vitamin D3 [recommended daily intake for 1-13 years old is 600 IU (15 mcg);risk for toxicity over 2000 IU/day] of an unregulated vitamin D3 supplement, led to the patient's toxicity. Pamidronate was instrumental in correcting severe hypercalcemia and should be considered early in its treatment. Considering vaccinehesitancy and continuation of the pandemic, providers should counsel parents who try alternative treatments for their children about the limited efficacy data and risks from vitamin D oversupplementation. Physicians should consider Pamidronate treatment early in the treatment of severe pediatric hypercalcemia.
ABSTRACT
Background: Severe presentation of ANCA vasculitis is a life-threatening disease despite aggressive immunodepression therapy. Complement hyperactivation is involved in pathogenesis;thus, the effect of the C5 inhibitor (eculizumab) used in severe forms of ANCA vasculitis may be a treatment option. Method(s): This is a retrospective study. Nine patients were included. Period of study: from May 2017 to May 2022. All patients received at least 3 drugs (steroids, rituximab and mycophenolate or cyclophosphamide) before eculizumab. Eculizumab was indicated as an off-label indication due to lack of improvement or clinical worsening. Result(s): Mean (SD) age: 62 (15) years. Female: 4. Three patients showed serum antiproteinase 3 antineutrophil cytoplasmic antibody (PR3-ANCA) and 5 (myeloperoxidase: MPO-ANCA) and one was ANCA-negative. Six patients had an estimated glomerular filtration rate (eGFR) < 10 ml/min/1.73 m2 at presentation. Five patients had pulmonary involvement. The mean (min-max) time of follow-up after the onset of eculizumab was 27 (1-60) months. One patient ANCA-negative microscopic polyangiitis with diffuse alveolar hemorrhage needed orotracheal intubation and had a satisfactory evolution after eculizumab;however, 20 days after, the patient developed a COVID-19 infection and died. One patient who needed urgent dialysis at presentation did not recover renal function and showed a complement factor H mutation. The evolution of the other 7 patients was as follows: the median (p25-p75) eGFR increased from baseline to the end of the follow-up: 9.1(4.8-21.7)ml/min/1.73m2 to 31(13-45)ml/min/1.73m2, respectively (P=0.018) and the mild proteinuria disappeared in all patients. Alveolar hemorrhage improved in all patients within seven days after the first eculizumab administration. The median (p25-p75) doses of eculizumab administered were 1800(1800-3600) mg. One patient required eculizumab for two different periods. Conclusion(s): One patient died due to a COVID-19 infection, and one remained in chronic renal replacement therapy. Alveolar hemorrhage was well controlled in all patients. The eGFR increased significantly in 7/9 patients, and in 4/6 patients, dialysis could be withdrawn. In severe ANCA vasculitis, eculizumab should be considered for improving outcomes.