TY - JOUR
T1 - Single Dose Of Bisphosphonate To Treat Infantile Hypercalcemia
AU - Patel, Neha S.
AU - Carpenter, Thomas O.
AU - Genel, Myron
N1 - Funding Information:
The authors thank the participants and their families, the health care professionals and staff of the Yale Pediatric Endocrinology Service, and the dedicated Yale New Haven Children's Hospital nursing staff. N.S.P. wrote the first draft of this manuscript and she has not received any honorarium, grant, or other form of payment to produce this manuscript. M.G. and T.O.C. contributed to the discussion and reviewed and edited the manuscript. N.S.P. is the guarantor of this work, and takes full responsibility for the integrity and accuracy of the report.
Publisher Copyright:
© 2017 Elsevier Inc.
PY - 2017/6/1
Y1 - 2017/6/1
N2 - Objective: To describe a case of subcutaneous fat necrosis (SCFN) treated with a single dose of bisphosphonate. Methods: We present the clinical, laboratory, and radiologic findings in an infant with severe hypercalcemia secondary to SCFN and the unique treatment course, as well as a review of relevant literature. Results: A term neonate with history of hypoxic ischemic encephalopathy status post therapeutic cooling was referred for severe hypercalcemia. At 2 weeks of life, he developed pink, indurated confluent plaques on his back and posterior upper arms, consistent with SCFN. Laboratory data showed a calcium level of 17.6 mg/dL and ionized calcium of 8.98 mg/dL. Parathyroid hormone was adequately suppressed at <3 pg/mL. Serum 1,25-dihydroxyvitamin D was elevated at 106 pg/mL and 25-hydroxyvitamin D appropriate at 31 ng/mL. Hypercalcemia persisted at 14.5 mg/dL despite aggressive hydration and furosemide, and the infant developed tachypnea. Pamidronate 0.5 mg/kg was given once and calcium declined to 12.8 mg/dL 12 hours later. The infant was kept on a low calcium diet for a few months with no subsequent hypercalcemia. Conclusion: SCFN is a potentially life-threatening complication due to development of unrecognized hypercalcemia. A single dose of bisphosphonate can be optimal for treatment of hypercalcemia. Abbreviations: 1,25-(OH)2D 1,25-dihydroxyvitamin D; 25-OHD 25-hydroxyvitamin D; HIE hypoxic ischemic encephalopathy; PTH parathyroid hormone; SCFN subcutaneous fat necrosis
AB - Objective: To describe a case of subcutaneous fat necrosis (SCFN) treated with a single dose of bisphosphonate. Methods: We present the clinical, laboratory, and radiologic findings in an infant with severe hypercalcemia secondary to SCFN and the unique treatment course, as well as a review of relevant literature. Results: A term neonate with history of hypoxic ischemic encephalopathy status post therapeutic cooling was referred for severe hypercalcemia. At 2 weeks of life, he developed pink, indurated confluent plaques on his back and posterior upper arms, consistent with SCFN. Laboratory data showed a calcium level of 17.6 mg/dL and ionized calcium of 8.98 mg/dL. Parathyroid hormone was adequately suppressed at <3 pg/mL. Serum 1,25-dihydroxyvitamin D was elevated at 106 pg/mL and 25-hydroxyvitamin D appropriate at 31 ng/mL. Hypercalcemia persisted at 14.5 mg/dL despite aggressive hydration and furosemide, and the infant developed tachypnea. Pamidronate 0.5 mg/kg was given once and calcium declined to 12.8 mg/dL 12 hours later. The infant was kept on a low calcium diet for a few months with no subsequent hypercalcemia. Conclusion: SCFN is a potentially life-threatening complication due to development of unrecognized hypercalcemia. A single dose of bisphosphonate can be optimal for treatment of hypercalcemia. Abbreviations: 1,25-(OH)2D 1,25-dihydroxyvitamin D; 25-OHD 25-hydroxyvitamin D; HIE hypoxic ischemic encephalopathy; PTH parathyroid hormone; SCFN subcutaneous fat necrosis
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U2 - 10.4158/EP161536.CR
DO - 10.4158/EP161536.CR
M3 - Article
AN - SCOPUS:85124184538
SN - 2376-0605
VL - 3
SP - e246-e250
JO - AACE Clinical Case Reports
JF - AACE Clinical Case Reports
IS - 3
ER -