Refractory Hypercalcemia in an Infant Secondary to Talc Pleurodesis Resolving After Renal Transplantation

Authors

  • S. J. Aujla,

    1. Division of Pulmonary Medicine, Allergy and Immunology, Department of Pediatrics, Children's Hospital of Pittsburgh, The University of Pittsburgh School of Medicine, Pittsburgh, PA
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  • P. Michelson,

    1. Division of Pulmonary Medicine, Allergy and Immunology, Department of Pediatrics, Children's Hospital of Pittsburgh, The University of Pittsburgh School of Medicine, Pittsburgh, PA
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  • C. B. Langman,

    1. Division of Kidney Diseases, Children's Memorial Hospital, Feinberg School of Medicine, Northwestern University, Chicago, IL
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  • R. Shapiro,

    1. University of Pittsburgh,Thomas E Starzl Transplant Institute, Surgery Division of Transplantation, Pittsburgh, PA
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  • D. Ellis,

    1. Division of Nephrology, Department of Pediatrics, Children's Hospital of Pittsburgh, The University of Pittsburgh School of Medicine, Pittsburgh, PA
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  • M. L. Moritz

    Corresponding author
    1. Division of Nephrology, Department of Pediatrics, Children's Hospital of Pittsburgh, The University of Pittsburgh School of Medicine, Pittsburgh, PA
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* Corresponding author: Michael L. Moritz, Michael.Moritz@CHP.edu

Abstract

Talc pleurodesis is the definitive therapy of recurrent pneumothorax and has not been associated with metabolic complications. We report an anephric male infant who developed severe hypercalcemia 6 months following talc pleurodesis for recurrent peritoneal dialysis-related hydrothorax. The etiology of hypercalcemia was related to persistently elevated 1,25-dihyroxyvitamin D3 (1,25[OH]2D) levels. The source appeared to be the extrarenal production of 1,25(OH)2D from macrophages in a large thoracic talc granuloma. Hypercalcemia was controlled with a combination of a low calcium diet, low calcium dialysis, ketoconazole and hydroxychloroquine, but elevated 1,25(OH)2D levels persisted. At 32 months of age the child underwent renal transplantation with alemtuzumab pre-conditioning. The hypercalcemia resolved immediately, with normalization of serum 1,25(OH)2D levels and without hypercalciuria. This case demonstrates that hypercalcemia is a potential complication of talc pleurodesis from the extrarenal production of 1,25(OH)2D and that alemtuzumab, a monoclonal antibody directed against the CD52 antigen (which is expressed on almost all macrophages), may have a role in the treatment of hypercalcemia associated with granulomatous conditions.

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