Does the Presence of Dystrophic Features in Patients With Type 1 Neurofibromatosis and Spinal Deformities Increase the Risk of Surgery?

Lykissas, Marios G.; Schorry, Elizabeth K.; Crawford, Alvin H.; Gaines, Sean; Rieley, Margaret B.; Jain, Viral V.

doi:10.1097/brs.0b013e31829a7779

Cited by 46 publications

(51 citation statements)

References 12 publications

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“…This fact could explain the slow progression of the signs that we had described. The deformation in scoliosis although discrete in our case corresponds to vertebral dysplasia also reported in the literature to varying degrees in 10% to 30% of cases [10].…”

Section: Discussionsupporting

confidence: 85%

“…In neurofibromatosis type 1, CT is less effective than MRI [6] in the diagnosis of spinal compression. It perfectly shows the bone lesions such as the scalloping which is very evocative [2] [4] [10] [11] that we have described. The vertebral dysplasia, causing scoliosis observed in our case, is the most common bone complications [2] [10].…”

Section: Discussionmentioning

confidence: 60%

“…It perfectly shows the bone lesions such as the scalloping which is very evocative [2] [4] [10] [11] that we have described. The vertebral dysplasia, causing scoliosis observed in our case, is the most common bone complications [2] [10]. It may require surgery in 37% of cases [2] [10].…”

Section: Discussionmentioning

confidence: 60%

“…The extensive mela-World Journal of Neuroscience no-dermal areas described in our case, have the same meaning as "Café-au-lait" spots [4] [11]. Although much less common, they can also contribute to the diagnosis of neurofibromatosis type 1 which remains clinical with well-defined criteria [4] [5] [10]. In case of neurologic manifestations as observed in our case, the imagery becomes an indispensable tool for the diagnosis in order to discuss the possibilities of surgical resection of the responsible lesion [6].…”

Section: Discussionmentioning

confidence: 88%

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Spinal Cord Compression, a Rare Neurofibromatosis Complication

Ouiminga¹,

Zabsonré²,

Kélani³

et al. 2019

WJNS

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Objective: The objective of this study is to report a case of spinal cord compression, which is a rare complication of neurofibromatosis type 1. Observation: We report the case of a 45-year-old man, which presented a syndrome of thoracic spinal cord compression at the stage of spastic paraparesis. Its installation was gradually over 6 months associated with the inaugural back pain. He had a clinical history of neurofibromatosis type 1 with "Café-au-lait" spots. There were multiple painless nodules under the skin of different size on the chest, forearms and legs. A large isolated nodule, purplish was observed on the chest. The neuro-imaging showed a para-spinal anterior mass expansion inside the spinal canal causing spinal compression at the level of the second and third thoracic vertebra. It extends into the intervertebral foramen of the third and fourth thoracic vertebra leading to a scalloping. A second large heterogeneous left intra-abdominal mass containing cyst areas and calcifications was discovered in imaging. After a spinal decompression with laminectomy of the second and third thoracic vertebra, the reduction of pain and motor recovery was gradual. The large nodule excision was performed. Histology found a plexiform neurofibroma. Excision of the left intra-abdominal mass could not be performed because the patient's consent had not been obtained. Conclusion: The spinal cord compression is a rare complication of neurofibromatosis type 1. However, it is essential to think about it in front of any spinal cord symptoms or any atypical long term spinal pain.

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Section: Discussionsupporting

confidence: 85%

Section: Discussionmentioning

confidence: 60%

Section: Discussionmentioning

confidence: 60%

Section: Discussionmentioning

confidence: 88%

See 2 more Smart Citations

Spinal Cord Compression, a Rare Neurofibromatosis Complication

Ouiminga¹,

Zabsonré²,

Kélani³

et al. 2019

WJNS

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“…About 10% of children with NF-1 develop scoliosis that predominantly involves the cervical and thoracic spine [13]. Dystrophic scoliosis with NF-1 has a high risk of rapid progression [5].…”

Section: Discussionmentioning

confidence: 99%

Posterior only instrumented fusion provides incomplete curve control for early-onset scoliosis in type 1 neurofibromatosis

Cai

Qiu

et al. 2020

BMC Pediatr

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Background: The mid-long term outcomes of posterior spinal fusion in pediatric neurofibromatosis type 1 (NF-1) patients are rarely reported, so does the effectiveness of itsorthopeidc maintenance function. This study aims to evaluate the mid-long term surgical outcomes of posterior only instrumented spinal fusion for early-onset scoliosis (EOS) in NF-1 patients. Methods: A retrospective review was performed on a cohort of 10 NF-1 patients having EOS from 2008 to 2014 in our hospital, the age averaged at 7.8 years old when they underwent posterior only instrumented spinal fusion for their EOS. Both general clinical data and surgical specific data of the patients were collected and reviewed, and the dystrophic progression of EOS was evaluated during the follow-up. Results: The average duration of follow-up was 54 months (24 to 88 months). All patients underwent posterior only instrumented spinal fusion at 1 stage. The primary curves of EOS were thoracic in 9 cases and 1 patient had lumbar scoliosis. Preoperative major curve was significantly corrected (from 66.1 to 31.1 degrees). However, the major curve deteriorated significantly to 40.1 degrees on average at the end of the follow-up. The T1-S1 distance increased 2.8 cm on average and kept increasing at a rate of 0.6 cm/year during the follow-up. Conclusions: Posterior only fusion surgery was not a good option to treat the EOS in NF-1 patients despite the relatively short segments involvement in the disease. The maintenance of orthopedic effect after treatment was not satisfactory.

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Classification of neurofibromatosis‐related dystrophic or nondystrophic scoliosis based on image features using Bilateral CNN

et al. 2021

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We developed a system that can automatically classify cases of scoliosis secondary to neurofibromatosis type 1 (NF1-S) using deep learning algorithms (DLAs) and improve the accuracy and effectiveness of classification, thereby assisting surgeons with the auxiliary diagnosis. Methods: Comprehensive experiments in NF1 classification were performed based on a dataset consisting 211 NF1-S (131 dystrophic and 80 nondystrophic NF1-S) patients. Additionally, 100 congenital scoliosis (CS), 100 adolescent idiopathic scoliosis (AIS) patients, and 114 normal controls were used for experiments in primary classification. For identification of NF1-S with nondystrophic or dystrophic curves, we devised a novel network (i.e., Bilateral convolutional neural network [CNN]) utilizing a bilinear-like operation to discover the similar interest features between whole spine AP and lateral x-ray images. The performance of Bilateral CNN was compared with spine surgeons, conventional DLAs (i.e., and Bilinear CNN [BCNN]), recently proposed DLAs (i.e., ShuffleNet, MobileNet, and EfficientNet), and Two-path BCNN which was the extension of BCNN using AP and lateral x-ray images as inputs. Results: In NF1 classification, our proposed Bilateral CNN with 80.36% accuracy outperformed the other seven DLAs ranging from 61.90% to 76.19% with fivefold cross-validation. It also outperformed the spine surgeons (with an average accuracy of 77.5% for the senior surgeons and 65.0% for the junior surgeons). Our method is highly generalizable due to the proposed methodology and data augmentation. Furthermore, the heatmaps extracted by Bilateral CNN showed curve pattern and morphology of ribs and vertebrae contributing most to the classification results. In primary classification, our proposed method with an accuracy of 87.92% also outperformed all the other methods with varied accuracies between 52.58% and 83.35% with fivefold cross-validation. Conclusions: The proposed Bilateral CNN can automatically capture representative features for classifying NF1-S utilizing AP and lateral x-ray images, leading to a relatively good performance. Moreover, the proposed method can identify other spine deformities for auxiliary diagnosis.

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Does the Presence of Dystrophic Features in Patients With Type 1 Neurofibromatosis and Spinal Deformities Increase the Risk of Surgery?

Abstract: 3.

Cited by 46 publications

References 12 publications

Spinal Cord Compression, a Rare Neurofibromatosis Complication

Spinal Cord Compression, a Rare Neurofibromatosis Complication

Posterior only instrumented fusion provides incomplete curve control for early-onset scoliosis in type 1 neurofibromatosis

Classification of neurofibromatosis‐related dystrophic or nondystrophic scoliosis based on image features using Bilateral CNN

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