Single Dose Of Bisphosphonate To Treat Infantile Hypercalcemia

ABSTRACT: 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...

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Main Authors: Neha S. Patel, DO, Thomas O. Carpenter, MD, Myron Genel, MD
Format: Article
Language:English
Published: Elsevier 2017-01-01
Series:AACE Clinical Case Reports
Online Access:http://www.sciencedirect.com/science/article/pii/S2376060520301875
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spelling doaj-5f26fff85e1c4271a92a6f5c8736172b2021-04-30T07:23:52ZengElsevierAACE Clinical Case Reports2376-06052017-01-0133e246e250Single Dose Of Bisphosphonate To Treat Infantile HypercalcemiaNeha S. Patel, DO0Thomas O. Carpenter, MD1Myron Genel, MD2Address correspondence to Dr. Neha S. Patel, Yale Pediatric Endocrinology, One Long Wharf Drive, Suite 503, New Haven, CT 06510.; From Yale School of Medicine, New Haven, Connecticut.From Yale School of Medicine, New Haven, Connecticut.From Yale School of Medicine, New Haven, Connecticut.ABSTRACT: 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 necrosishttp://www.sciencedirect.com/science/article/pii/S2376060520301875
collection DOAJ
language English
format Article
sources DOAJ
author Neha S. Patel, DO
Thomas O. Carpenter, MD
Myron Genel, MD
spellingShingle Neha S. Patel, DO
Thomas O. Carpenter, MD
Myron Genel, MD
Single Dose Of Bisphosphonate To Treat Infantile Hypercalcemia
AACE Clinical Case Reports
author_facet Neha S. Patel, DO
Thomas O. Carpenter, MD
Myron Genel, MD
author_sort Neha S. Patel, DO
title Single Dose Of Bisphosphonate To Treat Infantile Hypercalcemia
title_short Single Dose Of Bisphosphonate To Treat Infantile Hypercalcemia
title_full Single Dose Of Bisphosphonate To Treat Infantile Hypercalcemia
title_fullStr Single Dose Of Bisphosphonate To Treat Infantile Hypercalcemia
title_full_unstemmed Single Dose Of Bisphosphonate To Treat Infantile Hypercalcemia
title_sort single dose of bisphosphonate to treat infantile hypercalcemia
publisher Elsevier
series AACE Clinical Case Reports
issn 2376-0605
publishDate 2017-01-01
description ABSTRACT: 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
url http://www.sciencedirect.com/science/article/pii/S2376060520301875
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