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Differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates
CONTEXT: Tumoral calcinosis is a disorder of phosphate metabolism characterized by ectopic calcification around major joints. Surgery is the current treatment of choice, but a suboptimal choice in recurrent and multicentric lesions. AIMS: To evaluate the efficacy of bisphosphonates for the managemen...
Autores principales: | , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Medknow Publications & Media Pvt Ltd
2014
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4138909/ https://www.ncbi.nlm.nih.gov/pubmed/25143910 http://dx.doi.org/10.4103/2230-8210.137511 |
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author | Balachandran, Karthik Kamalanathan, Sadishkumar Sahoo, Jaya Prakash Das, Ashok Kumar Halanaik, Dhanapathi |
author_facet | Balachandran, Karthik Kamalanathan, Sadishkumar Sahoo, Jaya Prakash Das, Ashok Kumar Halanaik, Dhanapathi |
author_sort | Balachandran, Karthik |
collection | PubMed |
description | CONTEXT: Tumoral calcinosis is a disorder of phosphate metabolism characterized by ectopic calcification around major joints. Surgery is the current treatment of choice, but a suboptimal choice in recurrent and multicentric lesions. AIMS: To evaluate the efficacy of bisphosphonates for the management of tumoral calcinosis on optimized medical treatment. SETTINGS AND DESIGN: The study was done in the endocrine department of a tertiary care hospital in South India. We prospectively studied two patients with recurrent tumoral calcinosis who had failed therapy with phosphate lowering measures. MATERIALS AND METHODS: After informed consent, we treated both patients with standard age adjusted doses of bisphosphonates for 18 months. The response was assessed by X ray and whole body 99mTc-methylene diphosphonate bone scan at the beginning of therapy and at the end of 1 year. We also estimated serum phosphate levels and urinary phosphate to document serial changes. RESULTS: Two patients (aged 19 and 5 years) with recurrent idiopathic hyperphosphatemic tumoral calcinosis, following surgery were studied. Both patients had failed therapy with conventional medical management − low phosphate diet and phosphate binders. They had restriction of joint mobility. Both were given standard doses of oral alendronate and parenteral pamidronate respectively for more than a year, along with phosphate lowering measures. At the end of 1 year, one of the patients had more than 95% and 90% reduction in the size of the lesions in right and left shoulder joints respectively with total improvement in range of motion. In contrast, the other patient (5-year-old) had shown no improvement, despite continuing to maintain normophosphatemia following treatment. CONCLUSIONS: Bisphosphonate therapy in tumoral calcinosis is associated with lesion resolution and may be used as a viable alternative to surgery, especially in cases with multicentric recurrence or treatment failure to other drugs. |
format | Online Article Text |
id | pubmed-4138909 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2014 |
publisher | Medknow Publications & Media Pvt Ltd |
record_format | MEDLINE/PubMed |
spelling | pubmed-41389092014-08-20 Differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates Balachandran, Karthik Kamalanathan, Sadishkumar Sahoo, Jaya Prakash Das, Ashok Kumar Halanaik, Dhanapathi Indian J Endocrinol Metab Original Article CONTEXT: Tumoral calcinosis is a disorder of phosphate metabolism characterized by ectopic calcification around major joints. Surgery is the current treatment of choice, but a suboptimal choice in recurrent and multicentric lesions. AIMS: To evaluate the efficacy of bisphosphonates for the management of tumoral calcinosis on optimized medical treatment. SETTINGS AND DESIGN: The study was done in the endocrine department of a tertiary care hospital in South India. We prospectively studied two patients with recurrent tumoral calcinosis who had failed therapy with phosphate lowering measures. MATERIALS AND METHODS: After informed consent, we treated both patients with standard age adjusted doses of bisphosphonates for 18 months. The response was assessed by X ray and whole body 99mTc-methylene diphosphonate bone scan at the beginning of therapy and at the end of 1 year. We also estimated serum phosphate levels and urinary phosphate to document serial changes. RESULTS: Two patients (aged 19 and 5 years) with recurrent idiopathic hyperphosphatemic tumoral calcinosis, following surgery were studied. Both patients had failed therapy with conventional medical management − low phosphate diet and phosphate binders. They had restriction of joint mobility. Both were given standard doses of oral alendronate and parenteral pamidronate respectively for more than a year, along with phosphate lowering measures. At the end of 1 year, one of the patients had more than 95% and 90% reduction in the size of the lesions in right and left shoulder joints respectively with total improvement in range of motion. In contrast, the other patient (5-year-old) had shown no improvement, despite continuing to maintain normophosphatemia following treatment. CONCLUSIONS: Bisphosphonate therapy in tumoral calcinosis is associated with lesion resolution and may be used as a viable alternative to surgery, especially in cases with multicentric recurrence or treatment failure to other drugs. Medknow Publications & Media Pvt Ltd 2014 /pmc/articles/PMC4138909/ /pubmed/25143910 http://dx.doi.org/10.4103/2230-8210.137511 Text en Copyright: © Indian Journal of Endocrinology and Metabolism http://creativecommons.org/licenses/by-nc-sa/3.0 This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Original Article Balachandran, Karthik Kamalanathan, Sadishkumar Sahoo, Jaya Prakash Das, Ashok Kumar Halanaik, Dhanapathi Differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates |
title | Differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates |
title_full | Differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates |
title_fullStr | Differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates |
title_full_unstemmed | Differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates |
title_short | Differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates |
title_sort | differential response of idiopathic sporadic tumoral calcinosis to bisphosphonates |
topic | Original Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4138909/ https://www.ncbi.nlm.nih.gov/pubmed/25143910 http://dx.doi.org/10.4103/2230-8210.137511 |
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