Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the Literature
Fibrodysplasia ossificans progressiva (FOP) is an autosomal dominant disease, characterized by the formation of heterotopic ossification (HO) in muscles, ligaments, and tendons. Flare-ups, an inflammatory process that often precedes the formation of HO, can occur spontaneously, but trauma is also a...
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| Format: | Article |
| Language: | English |
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Frontiers Media S.A.
2020-02-01
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| Series: | Frontiers in Endocrinology |
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| Online Access: | https://www.frontiersin.org/article/10.3389/fendo.2020.00006/full |
| _version_ | 1819169420308643840 |
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| author | Esmée Botman Jan Coen Netelenbos Thomas Rustemeyer Linda J. Schoonmade Jakko A. Nieuwenhuijzen Bernd P. Teunissen Marieke Visser Pieter Raijmakers Adriaan A. Lammertsma Max Dahele Marelise Eekhoff |
| author_facet | Esmée Botman Jan Coen Netelenbos Thomas Rustemeyer Linda J. Schoonmade Jakko A. Nieuwenhuijzen Bernd P. Teunissen Marieke Visser Pieter Raijmakers Adriaan A. Lammertsma Max Dahele Marelise Eekhoff |
| author_sort | Esmée Botman |
| collection | DOAJ |
| description | Fibrodysplasia ossificans progressiva (FOP) is an autosomal dominant disease, characterized by the formation of heterotopic ossification (HO) in muscles, ligaments, and tendons. Flare-ups, an inflammatory process that often precedes the formation of HO, can occur spontaneously, but trauma is also a common trigger. It is not known whether radiotherapy, especially in higher doses, might cause sufficient trauma or inflammation to trigger a flare-up and subsequent HO in FOP patients. We report the case of a patient undergoing radiotherapy for the treatment of a 1-cm-wide basal cell carcinoma (BCC) of the lower lip. In addition, we present a systematic review of the available literature. Our patient received 54 Gy in 18 fractions with orthovoltage therapy, resulting in a clinical complete response of the tumor. Six months after treatment, there were no signs of HO either clinically or on [18F]NaF PET/CT. The systematic review identified 11 publications describing either radiation treatment in FOP or radiation therapy as a cause of HO in non-FOP patients. Six case reports described the use of radiation in FOP patients for various reasons, including one with a high-dose treatment of a lip BCC using superficial X-ray therapy. The remaining five studies described the use of low-dose radiotherapy to prevent or treat either an FOP flare-up or HO formation. None of these cases showed worsening of disease that could be attributed to the use of radiation therapy. Radiation induced HO in non-FOP patients was rare and occurred in five studies. The largest of these studies suggested that HO was induced after treatment with high doses, resulting in more widespread evidence of tissue damage, potentially being the end result of this damage. In conclusion, available reports suggest no contraindication to radiotherapy in FOP patients; although the number of cases was small, systematic toxicity reports often were not available, and none of the reports described high-dose, high-energy radiation treatment at locations such as muscle and joint regions. |
| first_indexed | 2024-12-22T19:19:13Z |
| format | Article |
| id | doaj.art-2f8974ccef06415a9bc5e61a6b3fc958 |
| institution | Directory Open Access Journal |
| issn | 1664-2392 |
| language | English |
| last_indexed | 2024-12-22T19:19:13Z |
| publishDate | 2020-02-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Endocrinology |
| spelling | doaj.art-2f8974ccef06415a9bc5e61a6b3fc9582022-12-21T18:15:26ZengFrontiers Media S.A.Frontiers in Endocrinology1664-23922020-02-011110.3389/fendo.2020.00006511381Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the LiteratureEsmée Botman0Jan Coen Netelenbos1Thomas Rustemeyer2Linda J. Schoonmade3Jakko A. Nieuwenhuijzen4Bernd P. Teunissen5Marieke Visser6Pieter Raijmakers7Adriaan A. Lammertsma8Max Dahele9Marelise Eekhoff10Department of Internal Medicine Section Endocrinology, Amsterdam Movement Sciences, Amsterdam Bone Centre, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Internal Medicine Section Endocrinology, Amsterdam Movement Sciences, Amsterdam Bone Centre, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Dermatology, Amsterdam UMC, University of Amsterdam, Amsterdam, NetherlandsMedical Library, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Urology, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Radiology and Nuclear Medicine, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Neurology, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Radiology and Nuclear Medicine, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Radiology and Nuclear Medicine, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Radiation Oncology, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsDepartment of Internal Medicine Section Endocrinology, Amsterdam Movement Sciences, Amsterdam Bone Centre, Amsterdam UMC, Vrije Universiteit Amsterdam, Amsterdam, NetherlandsFibrodysplasia ossificans progressiva (FOP) is an autosomal dominant disease, characterized by the formation of heterotopic ossification (HO) in muscles, ligaments, and tendons. Flare-ups, an inflammatory process that often precedes the formation of HO, can occur spontaneously, but trauma is also a common trigger. It is not known whether radiotherapy, especially in higher doses, might cause sufficient trauma or inflammation to trigger a flare-up and subsequent HO in FOP patients. We report the case of a patient undergoing radiotherapy for the treatment of a 1-cm-wide basal cell carcinoma (BCC) of the lower lip. In addition, we present a systematic review of the available literature. Our patient received 54 Gy in 18 fractions with orthovoltage therapy, resulting in a clinical complete response of the tumor. Six months after treatment, there were no signs of HO either clinically or on [18F]NaF PET/CT. The systematic review identified 11 publications describing either radiation treatment in FOP or radiation therapy as a cause of HO in non-FOP patients. Six case reports described the use of radiation in FOP patients for various reasons, including one with a high-dose treatment of a lip BCC using superficial X-ray therapy. The remaining five studies described the use of low-dose radiotherapy to prevent or treat either an FOP flare-up or HO formation. None of these cases showed worsening of disease that could be attributed to the use of radiation therapy. Radiation induced HO in non-FOP patients was rare and occurred in five studies. The largest of these studies suggested that HO was induced after treatment with high doses, resulting in more widespread evidence of tissue damage, potentially being the end result of this damage. In conclusion, available reports suggest no contraindication to radiotherapy in FOP patients; although the number of cases was small, systematic toxicity reports often were not available, and none of the reports described high-dose, high-energy radiation treatment at locations such as muscle and joint regions.https://www.frontiersin.org/article/10.3389/fendo.2020.00006/fullfibrodysplasia ossificans progressiva (FOP)radiotherapyheterotopic ossification (HO)[18F]NaF PET/CTACVR1 gene mutation |
| spellingShingle | Esmée Botman Jan Coen Netelenbos Thomas Rustemeyer Linda J. Schoonmade Jakko A. Nieuwenhuijzen Bernd P. Teunissen Marieke Visser Pieter Raijmakers Adriaan A. Lammertsma Max Dahele Marelise Eekhoff Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the Literature Frontiers in Endocrinology fibrodysplasia ossificans progressiva (FOP) radiotherapy heterotopic ossification (HO) [18F]NaF PET/CT ACVR1 gene mutation |
| title | Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the Literature |
| title_full | Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the Literature |
| title_fullStr | Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the Literature |
| title_full_unstemmed | Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the Literature |
| title_short | Radiotherapy in Fibrodysplasia Ossificans Progressiva: A Case Report and Systematic Review of the Literature |
| title_sort | radiotherapy in fibrodysplasia ossificans progressiva a case report and systematic review of the literature |
| topic | fibrodysplasia ossificans progressiva (FOP) radiotherapy heterotopic ossification (HO) [18F]NaF PET/CT ACVR1 gene mutation |
| url | https://www.frontiersin.org/article/10.3389/fendo.2020.00006/full |
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