Tumor induced osteomalacia in head and neck region: single center experience and systematic review
Tumor-induced osteomalacia in the head and neck region remains a challenging diagnosis to manage. Literature pertaining to management and outcome details remains sparse. We describe two cohorts: cohort 1 included seven patients from a single center in Western India with tumors located in paranasal s...
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| Format: | Article |
| Language: | English |
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Bioscientifica
2019-10-01
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| Series: | Endocrine Connections |
| Subjects: | |
| Online Access: | https://ec.bioscientifica.com/view/journals/ec/8/10/EC-19-0341.xml |
| _version_ | 1819044901562613760 |
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| author | Ravikumar Shah Anurag R Lila Swati Ramteke-Jadhav Virendra Patil Abhishek Mahajan Sushil Sonawane Puja Thadani Anil Dcruz Prathamesh Pai Munita Bal Subhada Kane Nalini Shah Tushar Bandgar |
| author_facet | Ravikumar Shah Anurag R Lila Swati Ramteke-Jadhav Virendra Patil Abhishek Mahajan Sushil Sonawane Puja Thadani Anil Dcruz Prathamesh Pai Munita Bal Subhada Kane Nalini Shah Tushar Bandgar |
| author_sort | Ravikumar Shah |
| collection | DOAJ |
| description | Tumor-induced osteomalacia in the head and neck region remains a challenging diagnosis to manage. Literature pertaining to management and outcome details remains sparse. We describe two cohorts: cohort 1 included seven patients from a single center in Western India with tumors located in paranasal sinuses (n = 3), intracranial (n = 2) and maxilla (n = 2). The unique features from our series is the management of persistent disease with radiation therapy (n = 2) and peptide receptor radionuclide therapy (PRRT) (n = 1). Cohort two has 163 patients identified from 109 publications for system atic review. Paranasal sinuses, mandible, intracranial disease, maxilla and oral cavit y, in descending order, are reportedly common tumor sites. Within this cohort, mean age was 46 ± 14 years at presentation with 44.1% having local symptoms. Duration of symptoms varied from 1 to 240 months. Pre-surgery mean serum phosphorus was 1.4 ± 0. 4 mg/dL and median FGF-23 levels were 3.6 (IQR:1.8–6.8) times of normal upper limi t of normal. Majority (97.5%) were managed primarily with surgical excision; however, primary radiotherapy (n = 2) and surgery combined with radiotherapy (n = 2) were also reported. Twenty patients had persistent disease while nine patients had recurrence, more commonly noted with intracranial and oral cavity tumors. Surgery was the most common second mode of treatment employed succeeded by radiotherapy. Four patients had metastatic disease. The most common histopathological diagnosis reported is PMT mix ed connective tissue, while the newer terminology ‘PMT mixed epithelial and connectiv e tissue type’ has been described in 15 patients. |
| first_indexed | 2024-12-21T10:20:02Z |
| format | Article |
| id | doaj.art-24e2419e2f074f6f8f1e13f85baed1d7 |
| institution | Directory Open Access Journal |
| issn | 2049-3614 2049-3614 |
| language | English |
| last_indexed | 2024-12-21T10:20:02Z |
| publishDate | 2019-10-01 |
| publisher | Bioscientifica |
| record_format | Article |
| series | Endocrine Connections |
| spelling | doaj.art-24e2419e2f074f6f8f1e13f85baed1d72022-12-21T19:07:27ZengBioscientificaEndocrine Connections2049-36142049-36142019-10-0181013301353https://doi.org/10.1530/EC-19-0341Tumor induced osteomalacia in head and neck region: single center experience and systematic reviewRavikumar Shah0Anurag R Lila1Swati Ramteke-Jadhav2Virendra Patil3Abhishek Mahajan4Sushil Sonawane5Puja Thadani6Anil Dcruz7Prathamesh Pai8Munita Bal9Subhada Kane10Nalini Shah11Tushar Bandgar12Department of Endocrinology, Seth GS Medical College & KEM Hospital, Parel, Mumbai, IndiaDepartment of Endocrinology, Seth GS Medical College & KEM Hospital, Parel, Mumbai, IndiaDepartment of Endocrinology, Seth GS Medical College & KEM Hospital, Parel, Mumbai, IndiaDepartment of Endocrinology, Seth GS Medical College & KEM Hospital, Parel, Mumbai, IndiaDepartment of Radiodiagnosis and Imaging, Tata Memorial Hospital, Mumbai, Maharashtra, IndiaDepartment of Endocrinology, Seth GS Medical College & KEM Hospital, Parel, Mumbai, IndiaDepartment of Endocrinology, Seth GS Medical College & KEM Hospital, Parel, Mumbai, IndiaDepartment of Head Neck Surgery, Tata Memorial Hospital, Mumbai, Maharashtra, IndiaDepartment of Head Neck Surgery, Tata Memorial Hospital, Mumbai, Maharashtra, IndiaDepartment of Pathology, Tata Memorial Hospital, Mumbai, Maharashtra, IndiaDepartment of Pathology, Tata Memorial Hospital, Mumbai, Maharashtra, IndiaDepartment of Endocrinology, Seth GS Medical College & KEM Hospital, Parel, Mumbai, IndiaDepartment of Endocrinology, Seth GS Medical College & KEM Hospital, Parel, Mumbai, IndiaTumor-induced osteomalacia in the head and neck region remains a challenging diagnosis to manage. Literature pertaining to management and outcome details remains sparse. We describe two cohorts: cohort 1 included seven patients from a single center in Western India with tumors located in paranasal sinuses (n = 3), intracranial (n = 2) and maxilla (n = 2). The unique features from our series is the management of persistent disease with radiation therapy (n = 2) and peptide receptor radionuclide therapy (PRRT) (n = 1). Cohort two has 163 patients identified from 109 publications for system atic review. Paranasal sinuses, mandible, intracranial disease, maxilla and oral cavit y, in descending order, are reportedly common tumor sites. Within this cohort, mean age was 46 ± 14 years at presentation with 44.1% having local symptoms. Duration of symptoms varied from 1 to 240 months. Pre-surgery mean serum phosphorus was 1.4 ± 0. 4 mg/dL and median FGF-23 levels were 3.6 (IQR:1.8–6.8) times of normal upper limi t of normal. Majority (97.5%) were managed primarily with surgical excision; however, primary radiotherapy (n = 2) and surgery combined with radiotherapy (n = 2) were also reported. Twenty patients had persistent disease while nine patients had recurrence, more commonly noted with intracranial and oral cavity tumors. Surgery was the most common second mode of treatment employed succeeded by radiotherapy. Four patients had metastatic disease. The most common histopathological diagnosis reported is PMT mix ed connective tissue, while the newer terminology ‘PMT mixed epithelial and connectiv e tissue type’ has been described in 15 patients.https://ec.bioscientifica.com/view/journals/ec/8/10/EC-19-0341.xmltumor-induced osteomalacia (TIO)oncogenic osteomalaciahead and necksystematic review |
| spellingShingle | Ravikumar Shah Anurag R Lila Swati Ramteke-Jadhav Virendra Patil Abhishek Mahajan Sushil Sonawane Puja Thadani Anil Dcruz Prathamesh Pai Munita Bal Subhada Kane Nalini Shah Tushar Bandgar Tumor induced osteomalacia in head and neck region: single center experience and systematic review Endocrine Connections tumor-induced osteomalacia (TIO) oncogenic osteomalacia head and neck systematic review |
| title | Tumor induced osteomalacia in head and neck region: single center experience and systematic review |
| title_full | Tumor induced osteomalacia in head and neck region: single center experience and systematic review |
| title_fullStr | Tumor induced osteomalacia in head and neck region: single center experience and systematic review |
| title_full_unstemmed | Tumor induced osteomalacia in head and neck region: single center experience and systematic review |
| title_short | Tumor induced osteomalacia in head and neck region: single center experience and systematic review |
| title_sort | tumor induced osteomalacia in head and neck region single center experience and systematic review |
| topic | tumor-induced osteomalacia (TIO) oncogenic osteomalacia head and neck systematic review |
| url | https://ec.bioscientifica.com/view/journals/ec/8/10/EC-19-0341.xml |
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