Hungry bone syndrome and normalisation of renal phosphorus threshold after total parathyroidectomy for tertiary hyperparathyroidism in X-linked hypophosphataemia: a case report

Hdl Handle:
http://hdl.handle.net/10147/315458
Title:
Hungry bone syndrome and normalisation of renal phosphorus threshold after total parathyroidectomy for tertiary hyperparathyroidism in X-linked hypophosphataemia: a case report
Authors:
Crowley, Rachel K; Kilbane, Mark; King, Thomas FJ; Morrin, Michelle; O’Keane, Myra; McKenna, Malachi J
Citation:
Journal of medical case reports. 2014 Mar 04;8(1):84
Journal:
Journal of medical case reports
Issue Date:
4-Mar-2014
URI:
http://dx.doi.org/10.1186/1752-1947-8-84; http://hdl.handle.net/10147/315458
Abstract:
Abstract Introduction This is the first report of which the authors are aware to describe this c.2166delinsGG mutation in X-linked hypophosphataemia and to describe normalisation of renal threshold for phosphate excretion after parathyroidectomy for tertiary hyperparathyroidism in X-linked hypophosphataemia. Case presentation We present the case of a 34-year-old Caucasian woman with X-linked hypophosphataemia. She developed tertiary hyperparathyroidism with markedly high bone turnover requiring total parathyroidectomy and had prolonged requirement for intravenous calcium infusion after surgery. She had a novel mutation in her phosphate-regulating gene with homologies to endopeptidases on the X-chromosome and had an unusual degree of dependence on phosphate supplementation. Prior to operative intervention she had a trial of cinacalcet that improved bone turnover markers when used in isolation but which led to a paradoxical rise in parathyroid hormone levels when given with phosphate supplementation. After correction of hungry bone syndrome, the renal phosphorus threshold normalised as a manifestation of hypoparathyroid state despite marked elevation in level of fibroblast growth factor 23. Conclusions This case illustrates the risk of tertiary hyperparathyroidism as a complication of treatment for hypophosphataemia; it highlights the morbidity associated with hungry bone syndrome and provides novel insight into renal handling of phosphorus.
Item Type:
Article
Language:
en
Keywords:
MUSCULOSKELETAL DISORDERS
Local subject classification:
BONE DISORDERS

Full metadata record

DC FieldValue Language
dc.contributor.authorCrowley, Rachel Ken_GB
dc.contributor.authorKilbane, Marken_GB
dc.contributor.authorKing, Thomas FJen_GB
dc.contributor.authorMorrin, Michelleen_GB
dc.contributor.authorO’Keane, Myraen_GB
dc.contributor.authorMcKenna, Malachi Jen_GB
dc.date.accessioned2014-04-07T10:52:03Z-
dc.date.available2014-04-07T10:52:03Z-
dc.date.issued2014-03-04-
dc.identifier.citationJournal of medical case reports. 2014 Mar 04;8(1):84en_GB
dc.identifier.urihttp://dx.doi.org/10.1186/1752-1947-8-84-
dc.identifier.urihttp://hdl.handle.net/10147/315458-
dc.description.abstractAbstract Introduction This is the first report of which the authors are aware to describe this c.2166delinsGG mutation in X-linked hypophosphataemia and to describe normalisation of renal threshold for phosphate excretion after parathyroidectomy for tertiary hyperparathyroidism in X-linked hypophosphataemia. Case presentation We present the case of a 34-year-old Caucasian woman with X-linked hypophosphataemia. She developed tertiary hyperparathyroidism with markedly high bone turnover requiring total parathyroidectomy and had prolonged requirement for intravenous calcium infusion after surgery. She had a novel mutation in her phosphate-regulating gene with homologies to endopeptidases on the X-chromosome and had an unusual degree of dependence on phosphate supplementation. Prior to operative intervention she had a trial of cinacalcet that improved bone turnover markers when used in isolation but which led to a paradoxical rise in parathyroid hormone levels when given with phosphate supplementation. After correction of hungry bone syndrome, the renal phosphorus threshold normalised as a manifestation of hypoparathyroid state despite marked elevation in level of fibroblast growth factor 23. Conclusions This case illustrates the risk of tertiary hyperparathyroidism as a complication of treatment for hypophosphataemia; it highlights the morbidity associated with hungry bone syndrome and provides novel insight into renal handling of phosphorus.-
dc.language.isoenen
dc.subjectMUSCULOSKELETAL DISORDERSen_GB
dc.subject.otherBONE DISORDERSen_GB
dc.titleHungry bone syndrome and normalisation of renal phosphorus threshold after total parathyroidectomy for tertiary hyperparathyroidism in X-linked hypophosphataemia: a case reporten_GB
dc.typeArticleen
dc.identifier.journalJournal of medical case reportsen_GB
dc.language.rfc3066en-
dc.rights.holderRachel K Crowley et al.; licensee BioMed Central Ltd.-
dc.description.statusPeer Reviewed-
dc.date.updated2014-04-02T10:37:05Z-
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