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      Thoracic ossification of the ligamentum flavum causing acute myelopathy in a patient with cervical ossification of the posterior longitudinal ligament: illustrative case

      case-report

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          Abstract

          BACKGROUND

          Ossification of the ligamentum flavum (OLF) has been well characterized as a distinct entity but also in tandem with ossification of the posterior longitudinal ligament (OPLL) in noncontiguous spinal regions. The majority of OLF cases are reported from East Asian countries where prevalent, but such cases are rarely reported in the North American population.

          OBSERVATIONS

          The authors present a case of a Thai-Cambodian American who presented with symptomatic thoracic OLF in tandem with asymptomatic cervical OPLL. A “floating” thoracic laminectomy, resection of OLF, and partial dural ossification (DO) resection with circumferential release of ossified dura were performed. Radiographic dural reexpansion and spinal cord decompression occurred despite the immediate intraoperative appearance of persistent thecal sac compression from retained DO.

          LESSONS

          Entire spinal axis imaging should be considered for patients with spinal ligamentous ossification disease, particularly in those of East Asian backgrounds. A floating laminectomy is one of several surgical approaches for OLF, but no consensus approach has been clearly established. High surgical complication rates are associated with thoracic OLF, most commonly dural tears/cerebrospinal fluid (CSF) leaks. DO commonly coexists with OLF, is recognizable on computed tomographic scans, and increases the risk of CSF leaks.

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          Most cited references42

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          Prevalence, distribution, and morphology of ossification of the ligamentum flavum: a population study of one thousand seven hundred thirty-six magnetic resonance imaging scans.

          Large scale, cross-sectional imaging study of a general population. To evaluate the prevalence, morphology, and distribution of ossification of the ligamentum flavum (OLF) in a population, and synthesize the scientific literature on the prevalence of OLF and some factors associated with its occurrence. OLF is a rare disease in which the pathogenesis has not been conclusively established. Little is known about its epidemiology. To date, there is no study that comprehensively assessed the distribution and prevalence of OLF in the whole spine using magnetic resonance imaging (MRI). A total of 1736 southern Chinese volunteers (1068 women; 668 men) between 8 and 88 years of age (mean, 38 years) were recruited by open invitation. MRI was administered to all the participants. T2-weighted, 5-mm spin-echo MRI sequences of the whole spine were obtained. Presence of OLF was identified as an area of low signal intensity in the T2 sagittal sequence located in the posterior part of the spinal canal, and subsequently confirmed by computed tomography scans showing areas of ossification within the ligamentum flavum. The distribution of OLF was classified into 3 types: the isolated type, continuous type, and noncontinuous type. While the morphology of the lesion was classified into triangular, round, and beak shapes based on the pattern of ossification on T2-weighted sagittal MRIs. OLF was identified in a total of 66 subjects or 3.8% of the population (52 women and 14 men). In 45(68.2%) cases, OLF was present at a single-level (isolated type), whereas in 21 (31.8%) cases OLF was present at multiple levels. The isolated type was found in 45 (68.2%) cases, continuous type in 11 (16.7%), and noncontinuous type in 10 (15.2%). The most common site of involvement is the lower thoracic spine, but they can also occur in the upper thoracic spine. The majority of the segments had a round morphology (n = 75: 81.5%), while 17 (18.5%) segments were triangular in shape. A literature review of the past 26 years showed only 4 reports on the prevalence of OLF, all were in special patient groups. Case reports have described postoperative paraplegia from failure to identify and decompress all stenotic segments of OLF. This study demonstrated that OLF is not uncommon, and that some 15% of the lesions are noncontinuous, and therefore could be missed. The authors recommend that for patients undergoing surgical decompression for 1 level of OLF, the whole spine should be routinely screened for other stenotic segments. Failure to do so could result in paraplegia from the nondecompressed levels.
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            Thoracic myelopathy caused by ossification of the ligamentum flavum: clinical features and surgical results in the Japanese population.

            Data obtained in patients with thoracic myelopathy caused by ossification of the ligamentum flavum (OLF) were retrospectively reviewed to clarify clinical features and surgical outcomes in the Japanese population. Seventy-two patients who underwent surgery for OLF-induced myelopathy in the Miyagi Prefecture, Japan, between 1988 and 2002 were observed for at least 2 years. Clinical data were collected from medical and operative records. The patients were evaluated pre- and postoperatively using the modified Japanese Orthopaedic Association (JOA) scale (maximum score 11). The relationships among various factors (age, sex, and preoperative duration of symptoms) affecting the preoperative severity of myelopathy and postoperative improvement were also examined. In this series the surgical outcome was relatively good and depended on the severity of myelopathy; thus early and correct diagnosis is required to avoid poorer results. The male/female ratio was 3:2 and the mean patient age at surgery was 61 years for men and 68 for women. The patients commonly noticed numbness or pain in their lower legs or gait disturbances. In a total of 104 decompressed intervertebral disc levels, more than 80% of the ossified ligaments were at the T9-10 level or lower. The mean preoperative JOA score of 5.1 improved to 7.9 after an average of 46 months. The postoperative results statistically depended on the preoperative severity of myelopathy. Among studies of patients with OLF-related myelopathy, the present study had the largest sample size, which should help clarify the clinical features of OLF myelopathy.
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              Epidemiological survey of ossification of the ligamentum flavum in thoracic spine: CT imaging observation of 993 cases.

              To investigate the characteristics of epidemiological distribution of the ossification of the ligamentum flavum (OLF) in the thoracic spine including the incidence, segmental distribution, and shape.
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                Author and article information

                Journal
                J Neurosurg Case Lessons
                J Neurosurg Case Lessons
                J Neurosurg Case Lessons
                Journal of Neurosurgery: Case Lessons
                American Association of Neurological Surgeons
                2694-1902
                6 September 2021
                6 September 2021
                : 2
                : 10
                : CASE2178
                Affiliations
                [1 ]Department of Molecular, Cellular, and Developmental Biology, University of California, Los Angeles, Los Angeles, California; and
                [2 ]Department of Neurosurgery, Scripps Clinic Medical Group, La Jolla, California
                Author notes
                Correspondence Christopher M. Uchiyama: Scripps Clinic Medical Group, La Jolla, CA. uchiyama.christopher@ 123456scrippshealth.org .

                INCLUDE WHEN CITING Published September 6, 2021; DOI: 10.3171/CASE2178.

                Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

                Article
                CASE2178
                10.3171/CASE2178
                9265228
                35855184
                29e0f6bd-b3a1-4290-bea4-9d216c08b4cb
                © 2021 The authors

                CC BY-NC-ND 4.0 ( http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                : 4 February 2021
                : 4 August 2021
                Page count
                Figures: 4, Tables: 0, References: 43, Pages: 5
                Categories
                Spine, Spine
                Thoracic, Thoracic
                Case Lesson

                ossification of ligamentum flavum,olf,ossification of posterior longitudinal ligament,opll,floating laminectomy,dural ossification,bmp = bone morphogenetic protein,csf = cerebrospinal fluid,ct = computed tomography,do = dural ossification,joa = japanese orthopedic association,mir = microrna,mri = magnetic resonance imaging,olf = ossification of the ligamentum flavum,opll = ossification of the posterior longitudinal ligament

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