Ülkühan ÖZTOPRAK, Erhan AKSOY, Nesrin CEYLAN, Ayse Secil EKSİOGLU, Deniz YÜKSEL

Düzeltme: Nörofibromatozis Tip 1 Tanılı Çocuklarda Desen Görsel Uyarılmış Potansiyellerin Değerlendirilmesi ve Radyolojik Bulgularla Karşılaştırılması

Amaç: Nörofibramotozis Tip 1 (NF tip 1)’li çocukların desen görsel uyarılmış (pattern visual evoked potentials; P-VEP) potansiyellerini değerlendirmek ve P-VEP sonuçlarının, optik gliom ve NF Tip 1 ilişkili bilinmeyen parlak objeler (unidentified brigtht objects; UBO) ile korelasyonunu irdelemek amaçlanmıştır. Gereç ve Yöntem: 2017-2020 yılları arasında Dr.Sami Ulus Kadın Doğum ve Çocuk Sağlığı ve Hastalıkları Eğitim Araştırma Hastanesi Çocuk Nöroloji Polikliniği’nde NF Tip 1 tanısı ile takip edilen, yaş ortalaması 9,61±3,7 yaş olan, 16 (%57)’sı erkek, 28 çocuk hastanın P-VEP P 100 latans değerleri, yaş ve cinsiyeti benzer 28 sağlıklı çocuktan oluşan kontrol grubunun P100 latansları ile karşılaştırıldı. Sonuç: Hastaların %82’sinde beyin magnetik resonans görüntülemede UBO, %21’inde optik gliom saptandı. Hasta grubunda P100 latansları kontrol grubuna göre anlamlı olarak daha uzun idi (p=0.013, p=0.043). Optik gliomu olan hastaların P100 latanslarının, optik gliomun anotomik lokalizas yonu ile uyumlu şekilde, optik gliomu olmayan hastalara göre anlamlı olarak daha uzun olduğu görüldü(p=0.042,p=0.025). Sonuç: Bu çalışma ile P-VEP testinin NF Tip 1’li çocuklarda görme yollarının fonksiyonel değerlendir mesinde kullanılabilecek objektif bir elektrofizyolojik test olduğu gösterilmiştir. NF Tip 1’li çocukların klinik takibinde P-VEP testinin kullanılmasının özellikle optik gliomların erken dönem tespitinde yararlı olabileceğini düşünmekteyiz.

Anahtar Kelimeler:

Neurofibromatosis type 1, Pattern visual evoked potentials, Children

Düzeltme: Evaluation of Visual Evoked Potentials in Children with Neurofibromatosis Type 1 and Comparison With Radiological Findings

Purpose: It was aimed to examine the pattern visual evoked potentials (P-VEP) of children with neurofibromatosis type 1 (NF type 1) and to evaluate the correlation of P-VEP results with optical gliomas and unidentified bright objects (UBO) associated with NF Type 1. Materials and Methods: P-VEP and P100 latencies of 28 children including 16 (%57) boys, who were followed up with the diagnosis of NF Type 1 in the Pediatric Neurology Outpatient Clinic in the Dr. Sami Ulus Maternity and Children's Health and Diseases Research and Training Hospital between 2017 and 2020, with a mean age of 9.61 ± 3.7 years of age were compared with the P100 latencies of a control group consisting of 28 healthy children of similar age and gender. Results: Unidentified bright objects were found in the brain of 82% of the patients by using magnetic resonance imaging, and optic glioma was found in 21% of the patients. P100 latencies were significantly longer in the patient group than in the control group (p = 0.013, p = 0.043). Patients with optic glioma were found to have significantly longer P100 latencies than patients without optic glioma, consistent with the anatomical location of the optic glioma (p = 0.042, p = 0.025). Conclusion: With this study, it has been shown that the P-VEP test is an objective electrophysiological test that can be used in the functional assessment of visual pathways in children with NF Type 1. We think that the use of the P-VEP test in the clinical follow-up of children with NF Type 1 may be particularly useful in the early detection of optic gliomas.

Keywords:

Çocuk, Nörofibromatozis tip 1, Desen görsel uyarılmış potansiyeller,

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1. Dunning-Davies BM, Parker AP. Annual review of children with neurofibromatosis type 1. Arch Dis Child Educ Pract Ed 2016 ;101(2):102-11.
2. Ferner RE, Huson SM, Thomas N, et al.Guidelines for the diagnosis and management of individuals with neurofibromatosis 1. J Med Genet 2007;44(2):81-8.
3. Abramowicz A, Gos M. Neurofibromin in neurofibromatosis type 1-mutations in NF1gene as a cause of disease. Dev Period Med 2014;18(3):297-306.
4. Lewis RA, Gerson LP, Axelson KA, et al. Von Recklinghausen neurofibromatosis: II. Incidence of optic gliomata. Ophthalmology 1984;91(8):929-35.
5. Rasool N, Odel JG, Kazim M. Optic pathway glioma of childhood. Curr Opin Ophthalmol 2017;28(3):289-95.
6. DiPaolo DP, Zimmerman RA, Rorke LB, et al. Neurofibromatosis type 1: pathologic substrate of high-signal-intensity foci in the brain. Radiology 1995;195(3):721-4.
7. Szudek J, Friedman J. Unidentified bright objects associated with features of neurofibromatosis 1. Pediatr Neurol 2002;27(2):123-7.
8. Jacques C, Dietemann J. Imagerie de la neurofibromatose de type 1. J Neuroradiol 2005;32(3):180-97.
9. Sherman J. Visual evoked potential (VEP): Basic concepts and clinical applications. J Am Optom Assoc 1979; 50(1):19-30.
10. Jeon J, Oh S, Kyung S. Assessment of visual disability using visual evoked potentials. BMC Ophthalmol 2012;12(1):36.
11. Stark D. Clinical uses of the visually evoked potential. Aust J Ophthalmol 1980;8(3):211.
12. Wolsey DH, Larson SA, Creel D,et al. Can screening for optic nerve gliomas in patients with neurofibromatosis type I be performed with visual-evoked potential testing? J AAPOS 2006;10(4):307-11.
13. Davies M, Williams R, Haq N, et al. MRI of optic nerve and postchiasmal visual pathways and visual evoked potentials in secondary progressive multiple sclerosis. Neuroradiology 1998;40(12):765-70.
14. Al-Eajailat SM, Senior MVA-M. The role of Magnetic Resonance Imaging and Visual Evoked Potential in management of optic neuritis. Pan Afr Med J 2014;17(1):54.
15. Neurofibromatosis N. Conference statement. National Institutes of Health consensus development conference. Arch Neurol 1988; 45(5):575-8.
16. Yerdelen D, Koc F, Durdu M,et al. Electrophysiological findings in neurofibromatosis type 1. J Neurol Sci 2011;306(1-2):42-8.
17. Iannaccone A, McCluney RA, Brewer VR, et al.Visual evoked potentials in children with neurofibromatosis type 1. Doc Ophthalmol 2002;105(1):63-81.
18. Aoki S, Barkovich A, Nishimura K, et al. Neurofibromatosis types 1 and 2: cranial MR findings. Radiology 1989;172(2):527-34.
19. DeBella K, Poskitt K, Szudek J,et al. Use of “unidentified bright objects” on MRI for diagnosis of neurofibromatosis 1 in children. Neurology 2000;54(8):1646-51.
20. Duffner PK, Cohen ME, Seidel FG, et al.The significance of MRI abnormalities in children with neurofibromatosis. Neurology. 1989;39(3):373-8.
21. Ferraz Filho JRL, Munis MP, Souza AS, et al. Unidentified bright objects on brain MRI in children as a diagnostic criterion for neurofibromatosis type 1. Pediatr Radiol 2008;38(3):305-10.
22. Menor F, Marti-Bonmati L, Arana E, et al. Neurofibromatosis type 1 in children: MR imaging and follow-up studies of central nervous system findings. Eur J Radiol 1998;26(2):121-31.
23. Rosenbaum T, Kim HA, Boissy YL, at al. Neurofibromin, the neurofibromatosis type 1 Ras‐GAP, is required for appropriate P0 expression and myelination. Ann N Y Acad Sci 1999;883(1):203-14.
24. Van Mierlo C, Spileers W, Legius E, et al. Role of visual evoked potentials in the assessment and management of optic pathway gliomas in children. Doc Ophthalmol 2013;127(3):177-90.
25. North K, Cochineas C, Tang E, et al. Optic gliomas in neurofibromatosis type 1: role of visual evoked potentials. Pediatr Neurol 1994;10(2):117-23.
26. Avery RA, Ferner RE, Listernick R, et al. Visual acuity in children with low grade gliomas of the visual pathway: implications for patient care and clinical research. J Neurooncol 2012;110(1):1-7.
27. Listernick R, Louis DN, Packer RJ,et al. Optic pathway gliomas in children with neurofibromatosis 1: consensus statement from the NF1 Optic Pathway Glioma Task Force. Ann Neurol 1997;41(2):143-9.
28. Dunn DW, PURVIN V. Optic pathway gliomas in neurofibromatosis. Dev Med Child Neurol 1990;32(9):820-4.
29. Jabbari B, Maitland CG, Morris LM, et al. The value of visual evoked potential as a screening test in neurofibromatosis. Arch Neurol 1985;42(11):1072-4.
30. Rossi L, Pastorino G, Scotti G,et al. Early diagnosis of optic glioma in children with neurofibromatosis type 1. Childs Nerv Syst 1994;10(7):426-9.
31. Kelly JP, Leary S, Khanna P, et al. Longitudinal measures of visual function, tumor volume, and prediction of visual outcomes after treatment of optic pathway gliomas. Ophthalmology 2012;119(6):1231-7.
32. Ammendola A, Ciccone G, Ammendola E. Utility of multimodal evoked potentials study in neurofibromatosis type 1 of childhood. Pediatr Neurol 2006;34(4):276-80.
33. Vagge A, Camicione P, Pellegrini M, et al. Role of visual evoked potentials and optical coherence tomography in the screening for optic pathway gliomas in patients with neurofibromatosis type I. Eur J Ophthalmol 2020; DOI: 10.1177/1120672120906989
34. Listernick R, Ferner RE, Liu GT, et al.Optic pathway gliomas in neurofibromatosis‐1: controversies and recommendations. Ann Neurol 2007;61(3):189-98.