Multimodal Imaging in Neurofibromatosis Type 1-associated Nerve Sheath Tumors

Salamon, Johannes; Mautner, Victor; Adam, Gerhard; Derlin, Thorsten

doi:10.1055/s-0035-1553505

Cited by 37 publications

(36 citation statements)

References 46 publications

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“…Despite the wide variation and lack of consensus regarding the cutoff SUVmax for suggesting malignant transformation, 18F-FDG PET/CT is a highly sensitive and specific modality for detection of MPNSTs, with reported sensitivity up to 100% and specificity of 77%–95%. [ 38 ]…”

Section: Discussionmentioning

confidence: 99%

Comparison of hybrid 18F.fluorodeoxyglucose positron emission tomography/magnetic resonance imaging and positron emission tomography/computed tomography for evaluation of peripheral nerve sheath tumors in patients with neurofibromatosis type 1

Raad¹,

Lala²,

Allen³

et al. 2018

World J Nucl Med

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Rapidly enlarging, painful plexiform neurofibromas (PN) in neurofibromatosis type 1 (NF1) patients are at higher risk for harboring a malignant peripheral nerve sheath tumor (MPNST). Fludeoxyglucose (FDG) positron emission tomography/computed tomography (PET/CT) has been used to support more invasive diagnostic and therapeutic interventions. However, PET/CT imparts an untoward radiation hazard to this population with tumor suppressor gene impairment. The use of FDG PET coupled with magnetic resonance imaging (MRI) rather than CT is a safer alternative but its relative diagnostic sensitivity requires verification. Ten patients (6 females, 4 males, mean age 27 years, range 8–54) with NF1 and progressive PN were accrued from our institutional NF Clinic. Indications for PET scanning included increasing pain and/or progressive disability associated with an enlarging PN on serial MRIs. Following a clinically indicated whole-body FDG PET/CT, a contemporaneous PET/MRI was obtained using residual FDG activity with an average time interval of 3–4 h FDG-avid lesions were assessed for both maximum standardized uptake value (SUVmax) from PET/CT and SUVmax from PET/MR and correlation was made between the two parameters. 26 FDG avid lesions were detected on both PET/CT and PET/MR with an accuracy of 100%. SUVmax values ranged from 1.4–10.8 for PET/CT and from 0.2-5.9 for PET/MRI. SUVmax values from both modalities demonstrated positive correlation (r = 0.45, P < 0.001). PET/MRI radiation dose was significantly lower (53.35% ± 14.37% [P = 0.006]). In conclusion, PET/MRI is a feasible alternative to PET/CT in patients with NF1 when screening for the potential occurrence of MPNST. Reduction in radiation exposure approaches 50% compared to PET/CT.

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Section: Discussionmentioning

confidence: 99%

Comparison of hybrid 18F.fluorodeoxyglucose positron emission tomography/magnetic resonance imaging and positron emission tomography/computed tomography for evaluation of peripheral nerve sheath tumors in patients with neurofibromatosis type 1

Raad¹,

Lala²,

Allen³

et al. 2018

World J Nucl Med

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“…Novel parameters such as metabolic tumor volume (MTV) and total lesion glycolysis (TLG) have been used which have shown promise but lack enough evidence to justify routine use. [ 1 ] Further research is needed to study for precise diagnosis.…”

Section: Discussionmentioning

confidence: 99%

“…And whole-body MRI is more suited to serial follow-up in patients with PNFs, owing to its no radiation exposure. [ 1 ] 18 F-FDG PET/CT usually provides a high sensitivity and specificity for monitoring malignant lesions, particularly for staging and restaging in patient with tumor, in order to change therapy planning in time and improve prognosis. Our aim is to discuss the role of 18 F-FDG PET/CT in NF1 patients.…”

Section: Introductionmentioning

confidence: 99%

The value of 18F-FDG PET/CT in patient with neurofibromatosis type 1

et al. 2018

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Rationale:Neurofibromatosis type one (NF1) is characterized by cutaneous and nervous lesions, and the tendency to form plexiform neurofibromas (PNFs). PNFs may undergo malignant transformation into a malignant peripheral nerve sheath tumors (MPNSTs). MPNSTs often carry an significant morbidity and mortality.Patient concerns:A 17-year-old man with gradually increased multiple subcutaneous soft lesions. He also presented with numerous lentigines and multiple café-au-lait macules on his body.Diagnoses:These were collagen neurofibroma, which were definitively diagnosed by pathology. NF1 was eventually diagnosed.Interventions:These lesions were abnormal uptake of radiotracer, when he underwent positron emission tomography (PET) with fluorine-18-fluorodeoxyglucose (FDG) scanning. Standard uptake value (SUV) and other parameters can help to distinguish benign and malignant lesions in patient with NF1. He was underwent serials 18F-FDG PET/CT examinations to followed up, in order to monitor these lesions malignant transformation.Outcomes:So far, these subcutaneous soft lesions were not malignant transformation.Lessons:18F-FDG PET/CT is being increasingly used as an imaging modality to discover the systemic lesions and to discriminate between benign and malignant plexiform neurofibromas.

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“…Clinically, 89 Zr-Tf could be applied in an analogous fashion to screen for benign masses that are transitioning to malignancy due to mTOR hyperactivation. This is a major unmet need for mTOR driven inherited disorders like tuberous sclerosis complex or neurofibromatosis that result in multiple benign masses which progress unpredictably to incurable malignancies during childhood or adolescence(15, 16). …”

Section: Discussionmentioning

confidence: 99%

Noninvasive Measurement of mTORC1 Signaling with 89Zr-Transferrin

Truillet

Cunningham

Parker

et al. 2017

Clinical Cancer Research

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Purpose mTOR regulates many normal physiological processes and when hyperactive, can drive numerous cancers and human diseases. However, it is very challenging to detect and quantify mTOR signaling non-invasively in clinically relevant animal models of disease or man. We hypothesized that a nuclear imaging tool measuring intracellular mTOR activity could address this unmet need. Experimental Design Although the biochemical activity of mTOR is not directly amenable to nuclear imaging probe development, we show that the transferrin receptor can be used to indirectly measure intracellular changes in mTOR activity. Results After verifying that the uptake of radiolabeled transferrin (the soluble ligand of the transferrin receptor) is stimulated by active mTORC1 in vitro, we showed that 89Zr-labeled transferrin can measure mTORC1 signaling dynamics in normal and cancerous mouse tissues with positron emission tomography (PET). Lastly, we show that 89Zr-Tf can detect the upregulation of mTORC1 by tumor cells to escape the antitumor effects of a standard of care antiandrogen, which is to our knowledge the first example of applying PET to interrogate the biology of treatment resistant cancer. Conclusions In summary, we have developed the first quantitative assay to provide a comprehensive measurement of mTOR signaling dynamics in vivo, in specific normal tissues and during tumor development in genetically engineered animal models using a nuclear imaging tool that is readily translatable to man.

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Multimodal Imaging in Neurofibromatosis Type 1-associated Nerve Sheath Tumors

Cited by 37 publications

References 46 publications

Comparison of hybrid 18F.fluorodeoxyglucose positron emission tomography/magnetic resonance imaging and positron emission tomography/computed tomography for evaluation of peripheral nerve sheath tumors in patients with neurofibromatosis type 1

Comparison of hybrid 18F.fluorodeoxyglucose positron emission tomography/magnetic resonance imaging and positron emission tomography/computed tomography for evaluation of peripheral nerve sheath tumors in patients with neurofibromatosis type 1

The value of 18F-FDG PET/CT in patient with neurofibromatosis type 1

Noninvasive Measurement of mTORC1 Signaling with 89Zr-Transferrin

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