{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,13]],"date-time":"2026-04-13T16:08:28Z","timestamp":1776096508981,"version":"3.50.1"},"reference-count":30,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"15","license":[{"start":{"date-parts":[[2026,4,13]],"date-time":"2026-04-13T00:00:00Z","timestamp":1776038400000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Neurosurg Case Lessons"],"published-print":{"date-parts":[[2026,4,13]]},"abstract":"<jats:sec>\n                    <jats:title>BACKGROUND<\/jats:title>\n                    <jats:p>Neurofibromatosis type 1 (NF1) is a multisystem neurocutaneous disorder associated with skeletal dysplasia, dural ectasia, and, less commonly, spinal meningoceles. Although these lesions are often asymptomatic, they may progressively enlarge over time, resulting in spinal deformity and compression of adjacent neurovascular and aerodigestive structures. Cervical and cervicothoracic meningoceles are particularly rare and pose unique diagnostic and management challenges.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>OBSERVATIONS<\/jats:title>\n                    <jats:p>A 43-year-old woman with NF1 presented with progressive gait imbalance and left upper limb dysesthesia and weakness more than 1 decade after initial diagnosis of a right-sided cervical meningocele that had been managed conservatively. Serial imaging over 11 years demonstrated gradual enlargement of a cervicothoracic lateral meningocele, ultimately measuring more than 10 cm, with associated severe kyphoscoliosis, vertebral scalloping, foraminal widening, spinal cord deviation, and displacement of the vertebral and carotid arteries, as well as anterior compression of the esophagus and airway. Neurological deficits correlated with foraminal compression at the C5\u2013T1 levels. Brain MRI additionally revealed bilateral optic pathway gliomas, suggesting a more extensive NF1 phenotype. Given progressive neurological decline and neurovascular compromise, surgical repair of the anterior cervical meningocele was performed, resulting in marked postoperative radiological improvement and relief of mass effect.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>LESSONS<\/jats:title>\n                    <jats:p>Spinal meningoceles associated with NF1 may follow an indolent but progressive course over many years, emphasizing the necessity of long-term clinical and radiological surveillance even in initially asymptomatic patients. Cervical involvement, although uncommon, can lead to significant spinal deformity and neurovascular compression. Early recognition of clinical deterioration should prompt timely surgical reassessment. Multidisciplinary management is essential in patients with complex NF1 manifestations, particularly when spinal pathology coexists with other CNS tumors.<\/jats:p>\n                    <jats:p>\n                      <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"https:\/\/thejns.org\/doi\/10.3171\/CASE26112\">https:\/\/thejns.org\/doi\/10.3171\/CASE26112<\/jats:ext-link>\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/case26112","type":"journal-article","created":{"date-parts":[[2026,4,13]],"date-time":"2026-04-13T15:27:35Z","timestamp":1776094055000},"source":"Crossref","is-referenced-by-count":0,"title":["Progressive cervicothoracic meningocele with neurovascular compression and spinal deformity in neurofibromatosis type 1: a decade-long radiological and clinical evolution with operative repair. Illustrative case"],"prefix":"10.3171","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7064-8281","authenticated-orcid":true,"given":"Ahmed Hafez","family":"Mousa","sequence":"first","affiliation":[{"name":"Neurosciences, Dubai Health, Dubai"},{"name":"Department of Neurosurgery, Graduate Medical Education, Mohammed Bin Rashid University of Medicine and Health Sciences, Dubai Health, Dubai, United Arab Emirates"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Badr","family":"Hafiz","sequence":"additional","affiliation":[{"name":"Neurosurgery Section, Department of Neurosciences, King Faisal Specialist Hospital and Research Centre, Jeddah"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alaa","family":"Turkistani","sequence":"additional","affiliation":[{"name":"Neurosurgery Section, Department of Neurosciences, King Faisal Specialist Hospital and Research Centre, Jeddah"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4042-509X","authenticated-orcid":true,"given":"Tala","family":"Alsindi","sequence":"additional","affiliation":[{"name":"Neurosurgery Section, Department of Surgery, King Abdulaziz University Hospital, Jeddah"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0818-5746","authenticated-orcid":true,"given":"Mohammed","family":"Aref","sequence":"additional","affiliation":[{"name":"Neurosurgery Section, Department of Neurosciences, King Faisal Specialist Hospital and Research Centre, Jeddah"},{"name":"College of Medicine, Alfaisal University, Riyadh, Saudi Arabia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1754","reference":[{"key":"bib1","article-title":"Neurofibromatosis type 1","author":"Adil","unstructured":"Adil AKoritala TMunakomi SSingh AK.Neurofibromatosis type 1. In: StatPearls. StatPearls Publishing; 2025."},{"issue":"9","key":"bib2","series-title":"Oncogene","first-page":"1235","article-title":"Neurofibromin and suppression of tumorigenesis: beyond the GAP","volume":"41","author":"Mo","year":"2022","unstructured":"Mo JMoye SLMcKay RMLe LQ.Neurofibromin and suppression of tumorigenesis: beyond the GAP. Oncogene. 2022;41(9):1235-1251. doi:10.1038\/s41388-021-02156-y"},{"issue":"5","key":"bib3","series-title":"Neurosciences (Riyadh)","first-page":"375","article-title":"Clinical spectrum of neurofibromatosis type 1 among children in a tertiary care center","volume":"25","author":"Bashiri","year":"2020","unstructured":"Bashiri FAAlZamil LRAldhuwayhi RA.Clinical spectrum of neurofibromatosis type 1 among children in a tertiary care center. Neurosciences (Riyadh). 2020;25(5):375-379. doi:10.17712\/nsj.2020.5.20200081"},{"issue":"3","key":"bib4","series-title":"Cancers (Basel)","first-page":"463","article-title":"Cutaneous findings in neurofibromatosis type 1","volume":"13","author":"Ozarslan","year":"2021","unstructured":"Ozarslan BRusso TArgenziano GSantoro CPiccolo V.Cutaneous findings in neurofibromatosis type 1. Cancers (Basel). 2021;13(3):463. doi:10.3390\/cancers13030463"},{"issue":"1","key":"bib5","series-title":"Sci Rep","first-page":"16889","article-title":"Phenotyping spinal abnormalities in patients with neurofibromatosis type 1 using whole-body MRI","volume":"11","author":"Well","year":"2021","unstructured":"Well LCareddu AStark MPhenotyping spinal abnormalities in patients with neurofibromatosis type 1 using whole-body MRI. Sci Rep. 2021;11(1):16889. doi:10.1038\/s41598-021-96310-x"},{"issue":"10","key":"bib6","series-title":"Int J Clin Exp Med","first-page":"17289","article-title":"Coexistence of multiple rare spinal abnormalities in type 1 neurofibromatosis: a case report and literature review","volume":"8","author":"Zhao","year":"2015","unstructured":"Zhao CMZhang WJHuang ABCoexistence of multiple rare spinal abnormalities in type 1 neurofibromatosis: a case report and literature review. Int J Clin Exp Med. 2015;8(10):17289-17294."},{"key":"bib7","series-title":"Pan Afr Med J","first-page":"226","article-title":"Dural ectasia: a manifestation of type 1 neurofibromatosis","volume":"31","author":"Derdabi","year":"2018","unstructured":"Derdabi IJouadi HEEdderai M.Dural ectasia: a manifestation of type 1 neurofibromatosis. Pan Afr Med J. 2018;31:226. doi:10.11604\/pamj.2018.31.226.9797"},{"issue":"3","key":"bib8","series-title":"Childs Nerv Syst","first-page":"165","article-title":"Cervical meningocele and associated spinal anomalies","volume":"3","author":"Delashaw","year":"1987","unstructured":"Delashaw JBPark TSCail WMVollmer DG.Cervical meningocele and associated spinal anomalies. Childs Nerv Syst. 1987;3(3):165-169. doi:10.1007\/BF00717894"},{"issue":"7","key":"bib9","series-title":"Neuroradiology","first-page":"1235","article-title":"Unruptured giant lateral thoracic meningocele: extremely rare cause of cerebrospinal fluid hypotension in neurofibromatosis type 1","volume":"66","author":"Krishnan","year":"2024","unstructured":"Krishnan VRana AVattoth S.Unruptured giant lateral thoracic meningocele: extremely rare cause of cerebrospinal fluid hypotension in neurofibromatosis type 1. Neuroradiology. 2024;66(7):1235-1238. doi:10.1007\/s00234-024-03381-4"},{"issue":"1","key":"bib10","series-title":"J Cardiothorac Surg","first-page":"303","article-title":"Neurofibromatosis type 1 with huge intrathoracic meningoceles misdiagnosed as pleural effusion: a case report and literature review","volume":"19","author":"Chen","year":"2024","unstructured":"Chen NLi WMin LHuang QBian J.Neurofibromatosis type 1 with huge intrathoracic meningoceles misdiagnosed as pleural effusion: a case report and literature review. J Cardiothorac Surg. 2024;19(1):303. doi:10.1186\/s13019-024-02819-3"},{"key":"bib11","series-title":"Cancer Manag Res","first-page":"667","article-title":"Neurofibromatosis type 1-associated optic pathway gliomas: current challenges and future prospects","volume":"15","author":"Tang","year":"2023","unstructured":"Tang YGutmann DH.Neurofibromatosis type 1-associated optic pathway gliomas: current challenges and future prospects. Cancer Manag Res. 2023;15:667-681. doi:10.2147\/CMAR.S362678"},{"issue":"21","key":"bib12","series-title":"Neurology","first-page":"S15","article-title":"Functional outcome measures for NF1-associated optic pathway glioma clinical trials","volume":"81","author":"Fisher","year":"2013","unstructured":"Fisher MJAvery RAAllen JCFunctional outcome measures for NF1-associated optic pathway glioma clinical trials. Neurology. 2013;81(21)(suppl 1):S15-S24. doi:10.1212\/01.wnl.0000435745.95155.b8"},{"issue":"5","key":"bib13","series-title":"J Neurosurg","first-page":"778","article-title":"Carboplatin and vincristine chemotherapy for children with newly diagnosed progressive optic pathway gliomas","volume":"79","author":"Packer","year":"1993","unstructured":"Packer RJLange BAter JCarboplatin and vincristine chemotherapy for children with newly diagnosed progressive optic pathway gliomas. J Neurosurg. 1993;79(5):778-782. doi:10.3171\/jns.1993.79.5.0778"},{"issue":"15","key":"bib14","series-title":"N Engl J Med","first-page":"1430","article-title":"Selumetinib in children with inoperable plexiform neurofibromas","volume":"382","author":"Gross","year":"2020","unstructured":"Gross AMWolters PLDombi ESelumetinib in children with inoperable plexiform neurofibromas. N Engl J Med. 2020;382(15):1430-1442. doi:10.1056\/NEJMoa1912735"},{"issue":"7","key":"bib15","series-title":"Lancet Oncol","first-page":"1011","article-title":"Selumetinib in paediatric patients with BRAF-aberrant or neurofibromatosis type 1-associated recurrent, refractory, or progressive low-grade glioma: a multicentre, phase 2 trial","volume":"20","author":"Fangusaro","year":"2019","unstructured":"Fangusaro JOnar-Thomas AYoung Poussaint TSelumetinib in paediatric patients with BRAF-aberrant or neurofibromatosis type 1-associated recurrent, refractory, or progressive low-grade glioma: a multicentre, phase 2 trial. Lancet Oncol. 2019;20(7):1011-1022. doi:10.1016\/S1470-2045(19)30277-3"},{"key":"bib1","article-title":"Neurofibromatosis type 1","author":"Adil","unstructured":"Adil AKoritala TMunakomi SSingh AK.Neurofibromatosis type 1. In: StatPearls. StatPearls Publishing; 2025."},{"issue":"9","key":"bib2","series-title":"Oncogene","first-page":"1235","article-title":"Neurofibromin and suppression of tumorigenesis: beyond the GAP","volume":"41","author":"Mo","year":"2022","unstructured":"Mo JMoye SLMcKay RMLe LQ.Neurofibromin and suppression of tumorigenesis: beyond the GAP. Oncogene. 2022;41(9):1235-1251. doi:10.1038\/s41388-021-02156-y"},{"issue":"5","key":"bib3","series-title":"Neurosciences (Riyadh)","first-page":"375","article-title":"Clinical spectrum of neurofibromatosis type 1 among children in a tertiary care center","volume":"25","author":"Bashiri","year":"2020","unstructured":"Bashiri FAAlZamil LRAldhuwayhi RA.Clinical spectrum of neurofibromatosis type 1 among children in a tertiary care center. Neurosciences (Riyadh). 2020;25(5):375-379. doi:10.17712\/nsj.2020.5.20200081"},{"issue":"3","key":"bib4","series-title":"Cancers (Basel)","first-page":"463","article-title":"Cutaneous findings in neurofibromatosis type 1","volume":"13","author":"Ozarslan","year":"2021","unstructured":"Ozarslan BRusso TArgenziano GSantoro CPiccolo V.Cutaneous findings in neurofibromatosis type 1. Cancers (Basel). 2021;13(3):463. doi:10.3390\/cancers13030463"},{"issue":"1","key":"bib5","series-title":"Sci Rep","first-page":"16889","article-title":"Phenotyping spinal abnormalities in patients with neurofibromatosis type 1 using whole-body MRI","volume":"11","author":"Well","year":"2021","unstructured":"Well LCareddu AStark MPhenotyping spinal abnormalities in patients with neurofibromatosis type 1 using whole-body MRI. Sci Rep. 2021;11(1):16889. doi:10.1038\/s41598-021-96310-x"},{"issue":"10","key":"bib6","series-title":"Int J Clin Exp Med","first-page":"17289","article-title":"Coexistence of multiple rare spinal abnormalities in type 1 neurofibromatosis: a case report and literature review","volume":"8","author":"Zhao","year":"2015","unstructured":"Zhao CMZhang WJHuang ABCoexistence of multiple rare spinal abnormalities in type 1 neurofibromatosis: a case report and literature review. Int J Clin Exp Med. 2015;8(10):17289-17294."},{"key":"bib7","series-title":"Pan Afr Med J","first-page":"226","article-title":"Dural ectasia: a manifestation of type 1 neurofibromatosis","volume":"31","author":"Derdabi","year":"2018","unstructured":"Derdabi IJouadi HEEdderai M.Dural ectasia: a manifestation of type 1 neurofibromatosis. Pan Afr Med J. 2018;31:226. doi:10.11604\/pamj.2018.31.226.9797"},{"issue":"3","key":"bib8","series-title":"Childs Nerv Syst","first-page":"165","article-title":"Cervical meningocele and associated spinal anomalies","volume":"3","author":"Delashaw","year":"1987","unstructured":"Delashaw JBPark TSCail WMVollmer DG.Cervical meningocele and associated spinal anomalies. Childs Nerv Syst. 1987;3(3):165-169. doi:10.1007\/BF00717894"},{"issue":"7","key":"bib9","series-title":"Neuroradiology","first-page":"1235","article-title":"Unruptured giant lateral thoracic meningocele: extremely rare cause of cerebrospinal fluid hypotension in neurofibromatosis type 1","volume":"66","author":"Krishnan","year":"2024","unstructured":"Krishnan VRana AVattoth S.Unruptured giant lateral thoracic meningocele: extremely rare cause of cerebrospinal fluid hypotension in neurofibromatosis type 1. Neuroradiology. 2024;66(7):1235-1238. doi:10.1007\/s00234-024-03381-4"},{"issue":"1","key":"bib10","series-title":"J Cardiothorac Surg","first-page":"303","article-title":"Neurofibromatosis type 1 with huge intrathoracic meningoceles misdiagnosed as pleural effusion: a case report and literature review","volume":"19","author":"Chen","year":"2024","unstructured":"Chen NLi WMin LHuang QBian J.Neurofibromatosis type 1 with huge intrathoracic meningoceles misdiagnosed as pleural effusion: a case report and literature review. J Cardiothorac Surg. 2024;19(1):303. doi:10.1186\/s13019-024-02819-3"},{"key":"bib11","series-title":"Cancer Manag Res","first-page":"667","article-title":"Neurofibromatosis type 1-associated optic pathway gliomas: current challenges and future prospects","volume":"15","author":"Tang","year":"2023","unstructured":"Tang YGutmann DH.Neurofibromatosis type 1-associated optic pathway gliomas: current challenges and future prospects. Cancer Manag Res. 2023;15:667-681. doi:10.2147\/CMAR.S362678"},{"issue":"21","key":"bib12","series-title":"Neurology","first-page":"S15","article-title":"Functional outcome measures for NF1-associated optic pathway glioma clinical trials","volume":"81","author":"Fisher","year":"2013","unstructured":"Fisher MJAvery RAAllen JCFunctional outcome measures for NF1-associated optic pathway glioma clinical trials. Neurology. 2013;81(21)(suppl 1):S15-S24. doi:10.1212\/01.wnl.0000435745.95155.b8"},{"issue":"5","key":"bib13","series-title":"J Neurosurg","first-page":"778","article-title":"Carboplatin and vincristine chemotherapy for children with newly diagnosed progressive optic pathway gliomas","volume":"79","author":"Packer","year":"1993","unstructured":"Packer RJLange BAter JCarboplatin and vincristine chemotherapy for children with newly diagnosed progressive optic pathway gliomas. J Neurosurg. 1993;79(5):778-782. doi:10.3171\/jns.1993.79.5.0778"},{"issue":"15","key":"bib14","series-title":"N Engl J Med","first-page":"1430","article-title":"Selumetinib in children with inoperable plexiform neurofibromas","volume":"382","author":"Gross","year":"2020","unstructured":"Gross AMWolters PLDombi ESelumetinib in children with inoperable plexiform neurofibromas. N Engl J Med. 2020;382(15):1430-1442. doi:10.1056\/NEJMoa1912735"},{"issue":"7","key":"bib15","series-title":"Lancet Oncol","first-page":"1011","article-title":"Selumetinib in paediatric patients with BRAF-aberrant or neurofibromatosis type 1-associated recurrent, refractory, or progressive low-grade glioma: a multicentre, phase 2 trial","volume":"20","author":"Fangusaro","year":"2019","unstructured":"Fangusaro JOnar-Thomas AYoung Poussaint TSelumetinib in paediatric patients with BRAF-aberrant or neurofibromatosis type 1-associated recurrent, refractory, or progressive low-grade glioma: a multicentre, phase 2 trial. 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