title: Refractory hypercalcemia in an infant secondary to talc pleurodesis resolving after renal transplantation.
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-dihydroxyvitamin D(3) (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.
authors.affiliation: Division of Pulmonary Medicine, Allergy and Immunology, Department of Pediatrics, Children's Hospital of Pittsburgh, The University of Pittsburgh School of Medicine, Pittsburgh, PA, USA.
authors.forename: S J
authors.initials: SJ
authors.lastname: Aujla
authors.forename: P
authors.initials: P
authors.lastname: Michelson
authors.forename: C B
authors.initials: CB
authors.lastname: Langman
authors.forename: R
authors.initials: R
authors.lastname: Shapiro
authors.forename: D
authors.initials: D
authors.lastname: Ellis
authors.forename: M L
authors.initials: ML
authors.lastname: Moritz
mesh_terms: D000287:Administration, Topical; D015745:Granuloma, Foreign-Body / Q000209:etiology; D006801:Humans; D006876:Hydrothorax / Q000209:etiology / Q000628:therapy*; D006934:Hypercalcemia / Q000209:etiology* / Q000601:surgery; D007223:Infant; D007668:Kidney / Q000002:abnormalities; D016030:Kidney Transplantation; D008297:Male; D010530:Peritoneal Dialysis / Q000009:adverse effects; D018700:Pleurodesis; D012074:Remission Induction; D012597:Sclerosing Solutions / Q000008:administration & dosage / Q000009:adverse effects*; D013627:Talc / Q000008:administration & dosage / Q000009:adverse effects*; D014564:Urogenital Abnormalities / Q000601:surgery*
chemical_list: D012597:Sclerosing Solutions; D013627:Talc