Diagnostic value of magnetic resonance imaging in fibrodysplasia ossificans progressiva

E. Botman; B.P. Teunissen; P. Raijmakers; P. de Graaf; M. Yaqub; S. Treurniet; T. Schoenmaker; N. Bravenboer; D. Micha; G. Pals; A. Bökenkamp; J.C. Netelenbos; A.A. Lammertsma; E.M.W. Eekhoff

doi:10.1002/jbm4.10363

Diagnostic value of magnetic resonance imaging in fibrodysplasia ossificans progressiva

E. Botman, B.P. Teunissen, P. Raijmakers, P. de Graaf, M. Yaqub, S. Treurniet, T. Schoenmaker, N. Bravenboer, D. Micha, G. Pals, A. Bökenkamp, J.C. Netelenbos, A.A. Lammertsma, E.M.W. Eekhoff

Periodontology

Research output: Contribution to Journal › Article › Academic › peer-review

Abstract

Using [18F] Sodium Fuoride (NaF) Positron Emission Tomography (PET) it is not only possible to identify the ossifying potency of a flare-up, but also to identify an asymptomatic chronic stage of fibrodysplasia ossificans progressiva (FOP). The purpose of this study was to investigate the diagnostic role of a more widely available imaging modality, Magnetic Resonance Imaging (MRI), which is of special interest for studies in pediatric FOP patients. MRI and [18F]NaF PET/CT images at time of inclusion and subsequent follow-up CT scans of 4 patients were analyzed retrospectively. Presence, location, and intensity of edema identified by MRI were compared with activity on [18F]NaF PET. Occurrence or progression of heterotopic ossification (HO) was examined on the follow-up CT images. Thirteen different lesions in various muscle groups were identified: five lesions with only edema, five lesions with both edema and increased [18F]NaF uptake, one lesion with only increased [18F]NaF uptake, and two lesions with neither edema nor uptake of [18F]NaF. Mild edema, found in three lesions, was present at asymptomatic sites, which did not show increased [18F] NaF uptake or progression of HO on consecutive CT images. Moderate edema was found in three symptomatic lesions, with increased [18F]NaF on PET and progression of HO on CT. Severe edema was identified in four lesions. Interestingly, two of these lesions did not develop HO during follow-up; one of these two even gave obvious symptoms of a flare-up. MRI can identify whether symptoms are the result of an acute flare-up by the presence of moderate to severe edema. The occurrence of severe edema on MRI was not always related to an ossifying lesion. The additional diagnostic value of MRI requires further investigation, but MRI does not seem to fully replace the diagnostic characteristics of [18F]NaF PET/CT in FOP. © 2020 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

Original language	English
Article number	e10363
Pages (from-to)	e10363
Number of pages	6
Journal	JBMR Plus
Volume	4
Issue number	6
DOIs	https://doi.org/10.1002/jbm4.10363
Publication status	Published - Jun 2020

Bibliographical note

Funding

We thank the FOP patients who shared their data with us to improve our understanding of their disease. Authors' roles: Study design: EB, EMWE, JCN, BPT, PG, and AAL. Study conduct: EB, EMWE, JCN, BPT, PG, and AB. Data collection: EB, EMWE, BPT, PG, PR, and MY. Data analysis: EB, EMWE, BT, PG, PR, and MY. Data interpretation: EB, EMWE, BPT, PG, PR, and MY. Drafting manuscript: EB, EMWE, JCN, BT, and PG. Revising manuscript content: AB, ST, TS, NB, DM, GP, and AAL. Approving final version of manuscript: EB, EMWE, AAL, BPT, PG. EB, EMWE, and AAL take responsibility for the integrity of the data analysis.

Funders	Funder number
BPT
Active and Assisted Living programme

UN SDGs

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Botman, E., Teunissen, B. P., Raijmakers, P., de Graaf, P., Yaqub, M., Treurniet, S., Schoenmaker, T., Bravenboer, N., Micha, D., Pals, G., Bökenkamp, A., Netelenbos, J. C., Lammertsma, A. A., & Eekhoff, E. M. W. (2020). Diagnostic value of magnetic resonance imaging in fibrodysplasia ossificans progressiva. JBMR Plus, 4(6), e10363. Article e10363. https://doi.org/10.1002/jbm4.10363

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title = "Diagnostic value of magnetic resonance imaging in fibrodysplasia ossificans progressiva",

abstract = "Using [18F] Sodium Fuoride (NaF) Positron Emission Tomography (PET) it is not only possible to identify the ossifying potency of a flare-up, but also to identify an asymptomatic chronic stage of fibrodysplasia ossificans progressiva (FOP). The purpose of this study was to investigate the diagnostic role of a more widely available imaging modality, Magnetic Resonance Imaging (MRI), which is of special interest for studies in pediatric FOP patients. MRI and [18F]NaF PET/CT images at time of inclusion and subsequent follow-up CT scans of 4 patients were analyzed retrospectively. Presence, location, and intensity of edema identified by MRI were compared with activity on [18F]NaF PET. Occurrence or progression of heterotopic ossification (HO) was examined on the follow-up CT images. Thirteen different lesions in various muscle groups were identified: five lesions with only edema, five lesions with both edema and increased [18F]NaF uptake, one lesion with only increased [18F]NaF uptake, and two lesions with neither edema nor uptake of [18F]NaF. Mild edema, found in three lesions, was present at asymptomatic sites, which did not show increased [18F] NaF uptake or progression of HO on consecutive CT images. Moderate edema was found in three symptomatic lesions, with increased [18F]NaF on PET and progression of HO on CT. Severe edema was identified in four lesions. Interestingly, two of these lesions did not develop HO during follow-up; one of these two even gave obvious symptoms of a flare-up. MRI can identify whether symptoms are the result of an acute flare-up by the presence of moderate to severe edema. The occurrence of severe edema on MRI was not always related to an ossifying lesion. The additional diagnostic value of MRI requires further investigation, but MRI does not seem to fully replace the diagnostic characteristics of [18F]NaF PET/CT in FOP. {\textcopyright} 2020 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.",

author = "E. Botman and B.P. Teunissen and P. Raijmakers and {de Graaf}, P. and M. Yaqub and S. Treurniet and T. Schoenmaker and N. Bravenboer and D. Micha and G. Pals and A. B{\"o}kenkamp and J.C. Netelenbos and A.A. Lammertsma and E.M.W. Eekhoff",

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T1 - Diagnostic value of magnetic resonance imaging in fibrodysplasia ossificans progressiva

AU - Botman, E.

AU - Teunissen, B.P.

AU - Raijmakers, P.

AU - de Graaf, P.

AU - Yaqub, M.

AU - Treurniet, S.

AU - Schoenmaker, T.

AU - Bravenboer, N.

AU - Micha, D.

AU - Pals, G.

AU - Bökenkamp, A.

AU - Netelenbos, J.C.

AU - Lammertsma, A.A.

AU - Eekhoff, E.M.W.

PY - 2020/6

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N2 - Using [18F] Sodium Fuoride (NaF) Positron Emission Tomography (PET) it is not only possible to identify the ossifying potency of a flare-up, but also to identify an asymptomatic chronic stage of fibrodysplasia ossificans progressiva (FOP). The purpose of this study was to investigate the diagnostic role of a more widely available imaging modality, Magnetic Resonance Imaging (MRI), which is of special interest for studies in pediatric FOP patients. MRI and [18F]NaF PET/CT images at time of inclusion and subsequent follow-up CT scans of 4 patients were analyzed retrospectively. Presence, location, and intensity of edema identified by MRI were compared with activity on [18F]NaF PET. Occurrence or progression of heterotopic ossification (HO) was examined on the follow-up CT images. Thirteen different lesions in various muscle groups were identified: five lesions with only edema, five lesions with both edema and increased [18F]NaF uptake, one lesion with only increased [18F]NaF uptake, and two lesions with neither edema nor uptake of [18F]NaF. Mild edema, found in three lesions, was present at asymptomatic sites, which did not show increased [18F] NaF uptake or progression of HO on consecutive CT images. Moderate edema was found in three symptomatic lesions, with increased [18F]NaF on PET and progression of HO on CT. Severe edema was identified in four lesions. Interestingly, two of these lesions did not develop HO during follow-up; one of these two even gave obvious symptoms of a flare-up. MRI can identify whether symptoms are the result of an acute flare-up by the presence of moderate to severe edema. The occurrence of severe edema on MRI was not always related to an ossifying lesion. The additional diagnostic value of MRI requires further investigation, but MRI does not seem to fully replace the diagnostic characteristics of [18F]NaF PET/CT in FOP. © 2020 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

AB - Using [18F] Sodium Fuoride (NaF) Positron Emission Tomography (PET) it is not only possible to identify the ossifying potency of a flare-up, but also to identify an asymptomatic chronic stage of fibrodysplasia ossificans progressiva (FOP). The purpose of this study was to investigate the diagnostic role of a more widely available imaging modality, Magnetic Resonance Imaging (MRI), which is of special interest for studies in pediatric FOP patients. MRI and [18F]NaF PET/CT images at time of inclusion and subsequent follow-up CT scans of 4 patients were analyzed retrospectively. Presence, location, and intensity of edema identified by MRI were compared with activity on [18F]NaF PET. Occurrence or progression of heterotopic ossification (HO) was examined on the follow-up CT images. Thirteen different lesions in various muscle groups were identified: five lesions with only edema, five lesions with both edema and increased [18F]NaF uptake, one lesion with only increased [18F]NaF uptake, and two lesions with neither edema nor uptake of [18F]NaF. Mild edema, found in three lesions, was present at asymptomatic sites, which did not show increased [18F] NaF uptake or progression of HO on consecutive CT images. Moderate edema was found in three symptomatic lesions, with increased [18F]NaF on PET and progression of HO on CT. Severe edema was identified in four lesions. Interestingly, two of these lesions did not develop HO during follow-up; one of these two even gave obvious symptoms of a flare-up. MRI can identify whether symptoms are the result of an acute flare-up by the presence of moderate to severe edema. The occurrence of severe edema on MRI was not always related to an ossifying lesion. The additional diagnostic value of MRI requires further investigation, but MRI does not seem to fully replace the diagnostic characteristics of [18F]NaF PET/CT in FOP. © 2020 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

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DO - 10.1002/jbm4.10363

M3 - Article

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JF - JBMR Plus

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M1 - e10363

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Diagnostic value of magnetic resonance imaging in fibrodysplasia ossificans progressiva

Abstract

Bibliographical note

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