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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...

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Autores principales: Balachandran, Karthik, Kamalanathan, Sadishkumar, Sahoo, Jaya Prakash, Das, Ashok Kumar, Halanaik, Dhanapathi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Medknow Publications & Media Pvt Ltd 2014
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.
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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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