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:
- Citation:
- Journal of medical case reports. 2014 Mar 04;8(1):84
- Journal:
- 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:
- Local subject classification:
- BONE DISORDERS
Full metadata record
DC Field | Value | Language |
---|---|---|
dc.contributor.author | Crowley, Rachel K | en_GB |
dc.contributor.author | Kilbane, Mark | en_GB |
dc.contributor.author | King, Thomas FJ | en_GB |
dc.contributor.author | Morrin, Michelle | en_GB |
dc.contributor.author | O’Keane, Myra | en_GB |
dc.contributor.author | McKenna, Malachi J | en_GB |
dc.date.accessioned | 2014-04-07T10:52:03Z | - |
dc.date.available | 2014-04-07T10:52:03Z | - |
dc.date.issued | 2014-03-04 | - |
dc.identifier.citation | Journal of medical case reports. 2014 Mar 04;8(1):84 | en_GB |
dc.identifier.uri | http://dx.doi.org/10.1186/1752-1947-8-84 | - |
dc.identifier.uri | http://hdl.handle.net/10147/315458 | - |
dc.description.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. | - |
dc.language.iso | en | en |
dc.subject | MUSCULOSKELETAL DISORDERS | en_GB |
dc.subject.other | BONE DISORDERS | en_GB |
dc.title | Hungry bone syndrome and normalisation of renal phosphorus threshold after total parathyroidectomy for tertiary hyperparathyroidism in X-linked hypophosphataemia: a case report | en_GB |
dc.type | Article | en |
dc.identifier.journal | Journal of medical case reports | en_GB |
dc.language.rfc3066 | en | - |
dc.rights.holder | Rachel K Crowley et al.; licensee BioMed Central Ltd. | - |
dc.description.status | Peer Reviewed | - |
dc.date.updated | 2014-04-02T10:37:05Z | - |
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