Document Detail


Resorptive hypercalcemia in post-essential thrombocythemia myelofibrosis: treatment with denosumab.
MedLine Citation:
PMID:  22730513     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
CONTEXT: Hypercalcemia associated with myelofibrosis is rare, and its pathogenesis and treatment are not known.
OBJECTIVE: We report a unique case of hypercalcemia associated with post-essential thrombocythemia myelofibrosis and review the clinical and laboratory features, pathogenesis, and responsiveness to treatment with the bone antiresorptive agent, denosumab.
RESULTS: A 62-yr-old woman with essential thrombocythemia presented with progression to myelofibrosis with lytic skull lesions and symptomatic hypercalcemia. Other causes of hypercalcemia were excluded. Her disturbance in calcium homeostasis was not PTH- or vitamin D-mediated, although this has been postulated in cases of hypercalcemia with the related entity of primary myelofibrosis. Her hypercalcemia was refractory to aggressive iv saline administration, furosemide, calcitonin, and pamidronate, but promptly improved after one 120-mg sc dose of the anti-receptor activator of nuclear factor κB (RANK) ligand monoclonal antibody, denosumab, with sustained normocalcemia for approximately 2 months. She died 6 months later from complications due to the leukemic transformation of her hematological disease.
CONCLUSION: The pathogenesis of myelofibrosis-related hypercalcemia could be due to multiple factors, particularly changes in the RANK ligand-RANK-osteoprotegerin system that lead to increased osteoclast activity. Although we did not measure these factors, denosumab holds promise in the treatment of malignancy-associated hypercalcemia and specifically that related to myelofibrosis. Hypercalcemia associated with myelofibrosis is rare, and its pathogenesis and treatment are not known.
Authors:
Nadia Khoury; Julietta Chang; Alejandro A Gru; Michael P Whyte
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't     Date:  2012-06-22
Journal Detail:
Title:  The Journal of clinical endocrinology and metabolism     Volume:  97     ISSN:  1945-7197     ISO Abbreviation:  J. Clin. Endocrinol. Metab.     Publication Date:  2012 Sep 
Date Detail:
Created Date:  2012-09-10     Completed Date:  2012-11-26     Revised Date:  2013-06-03    
Medline Journal Info:
Nlm Unique ID:  0375362     Medline TA:  J Clin Endocrinol Metab     Country:  United States    
Other Details:
Languages:  eng     Pagination:  3051-5     Citation Subset:  AIM; IM    
Affiliation:
Department of Endocrinology and Diabetes, Washington University School of Medicine at Barnes-Jewish Hospital, St. Louis, Missouri 63110, USA.
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MeSH Terms
Descriptor/Qualifier:
Antibodies, Monoclonal, Humanized / therapeutic use*
Autopsy
Biopsy
Bone Marrow / pathology
Calcium / blood
Fatal Outcome
Female
Humans
Hypercalcemia / drug therapy*,  etiology,  pathology
Leukemia / etiology
Middle Aged
Osteoprotegerin / physiology
Primary Myelofibrosis / complications,  drug therapy*,  pathology
RANK Ligand / physiology
Thrombocytosis / complications,  drug therapy*,  pathology
Chemical
Reg. No./Substance:
0/Antibodies, Monoclonal, Humanized; 0/Osteoprotegerin; 0/RANK Ligand; 4EQZ6YO2HI/denosumab; 7440-70-2/Calcium

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