A rare case of intervertebral disc calcification combined with ossification of the posterior longitudinal ligament in a child: a case report and literature review.

BACKGROUND: Intervertebral disc calcification (IDC) combined with calcification in children has been sporadically reported, while ossification of the posterior longitudinal ligament (OPLL) in the cervical spine in pediatric patients is exceedingly rare. The aim of this study is to investigate the potential prognosis and outcomes associated with this condition. CASE PRESENTATION: We present an unusual case involving a 10-year-old Chinese child diagnosed with calcified cervical disc herniation and ossification of the posterior longitudinal ligament. Conservative treatment measures were implemented, and at the 1-month and 6-month follow-up, the patient's pain exhibited significant improvement. Subsequent cervical MRI and CT scans revealed the complete disappearance of OPLL and substantial absorption of the calcified disc. During the three-month follow-up, CT demonstrated slight residual disc calcification, however, the patient remained asymptomatic with no discernible limitation in cervical motion. CONCLUSIONS: We conducted a comprehensive review of several cases presenting with the same diagnosis. It is noteworthy that IDC combined with OPLL in children constitutes a rare clinical entity. Despite imaging indications of potential spinal canal occupation, the majority of such cases demonstrate complete absorption following conservative treatment, with OPLL exhibiting a faster absorption rate than calcified discs.

Ossification of the Posterior Longitudinal Ligament: Pathophysiology, Diagnosis, and Management.

Ossification of the posterior longitudinal ligament (OPLL) occurs as heterotopic bone forms in the posterior longitudinal ligament, resulting in neural compression, myelopathy, and radiculopathy. OPLL is most commonly observed in East Asian populations, with prevalence rates of 1.9% to 4.3% reported in Japan. OPLL rates are lower in North American and European patients, with reported prevalence of 0.1% to 1.7%. Patients typically develop symptoms due to OPLL in their cervical spines. The etiology of OPLL is multifactorial, including genetic, metabolic, and anatomic factors. Asymptomatic or symptomatic patients with OPLL can be managed nonsurgically, whereas patients with neurologic symptoms may require surgical decompression from an anterior, posterior, or combined approach. Surgical treatment can provide notable improvement in neurologic function. Surgical decision making accounts for multiple factors, including patient comorbidities, neurologic status, disease morphology, radiographic findings, and procedure complication profiles. In this study, we review OPLL epidemiology and pathophysiology, clinical features, radiographic evaluation, management, and complications.

Seesaw-Like Cervical Ossification of Posterior Longitudinal Ligament.

Ossification of posterior longitudinal ligament (OPLL) is a pathological process in which lamellar bone is deposited at the posterior longitudinal ligament and can lead to a limited range of cervical motion and spinal cord compression. A 64-year-old man presented with a 10-month history of worsening clumsiness in the hands and impaired gait, and he occasionally had a feeling of an electric shock in the limbs when the neck was flexed. Physical examination revealed atrophy of the intrinsic hand muscles, rapid reflexes in the lower extremities, and positive Hoffman sign and Babinski sign results. Seesaw-like OPLL was observed on hyperextension and hyperflexion x-rays, which also showed that the OPLL involved the spinal canal; laminoplasty and laminectomy were not recommended for this specific type of OPLL, even though the K-line was positive on both x-rays.

Consensus statement on diagnosis and treatment of cervical ossification of posterior longitudinal ligament from Asia Pacific Spine Society (APSS) 2020.

PURPOSE: The study aimed to develop an evidence-based expert consensus statement on diagnosis and treatment of cervical ossification posterior longitudinal ligament (OPLL). METHOD: Delphi method was used to perform such survey, and the panel members from Asia Pacific Spine Society (APSS) 2020 were invited to answer the open-ended questions in rounds 1 and 2. Then the results were summarized and developed into a Likert-style questionnaire for voting in round 3, and the level of agreement was defined as 80%. In the whole process, we conducted a systematic literature search on evidence for each statement. RESULTS: Cervical OPLL can cause various degrees of neurological symptoms, an it's thought to be more common in Asia population. CT reconstruction is an important imaging examination to assist diagnosis and guide surgical choice. Segmental, continuous, mixed, and focal type is the most widely used classification system. The non-surgical treatment is recommended for patients with no or mild clinical symptoms, or irreversible neurological damage, or failed surgical decompression, or condition cannot tolerant surgery, or refusing surgery. As OPLL may continue to develop gradually, surgical treatment would be considered in their course inevitably. The surgical choice should depend on various conditions, such as involved levels, thickness, and type of OPLL, skill-experiences of surgeons, which are listed and discussed in the article. CONCLUSION: In this statement, we describe the clinical features, classifications, and diagnostic criteria of cervical OPLL, and review various surgical methods (such as their indications, complications), and provide a guideline on their choice strategy.

The Effect of an Educational and Interactive Informed Consent Process on Patients With Cervical Spondylotic Myelopathy Caused by Ossification of the Posterior Longitudinal Ligament.

STUDY DESIGN: Prospective observational cohort study. OBJECTIVE: In this study, an educational and interactive informed consent (EIC) program was proposed for patients with cervical spondylotic myelopathy caused by ossification of the posterior longitudinal ligament (OPLL-CSM) to improve their comprehension level during the informed consent process. SUMMARY OF BACKGROUND DATA: OPLL-CSM is a slow progressive disease, and it is difficult for patients to understand the disease. Few studies have evaluated very specific programs to improve the informed consent process for these patients. METHODS: This prospective study evaluated patients with OPLL-CSM who either underwent the proposed EIC process (n = 63) or the standard consent process (n = 124). The standard consent process only included a physician-patient interview. During the EIC process, information was provided regarding OPLL-CSM through information booklets, a video, verbal information, and initial and second physician-patient interviews. After the second physician-patient interview, the patient was requested to answer 14 medical questions to assess their knowledge about OPLL-CSM. The proposed EIC process took approximately 90 minutes. They were asked to report the most useful educational method and the most effective method of reinforcing verbal communication. RESULTS: The mean questionnaire scores were higher in the EIC group than in the control group (P < 0.001). Video was selected by 50/63 patients (79.4%) as the most useful EIC process method, and the most effective method of reinforcing verbal communication was video (n = 61; 96.8%). Patients in the EIC group reported having higher satisfaction with surgery (P = 0.024) than did those in the control group. CONCLUSION: The proposed EIC process was shown to result in good patient comprehension and recall regarding OPLL-CSM. Using a video was the most informative and effective reinforcement of verbal communication. The enhanced educational group had better knowledge and improved satisfaction following surgery. The EIC process might help physicians educate and counsel patients regarding OPLL-CSM and its treatment. LEVEL OF EVIDENCE: 2.

How Does Ossification of Posterior Longitudinal Ligament Progress in Conservatively Managed Patients?

STUDY DESIGN: Retrospective cohort study. OBJECTIVE: The aim of this study was to elucidate the progression of ossification of the posterior longitudinal ligament (OPLL) in conservatively managed patients and determine its risk factors SUMMARY OF BACKGROUND DATA.: Although several studies have demonstrated how OPLL progresses after laminoplasty or fusion, its progression in conservatively managed patients remains unclear. METHODS: The vertical length of the ossified mass and its thickness at each segment were evaluated on sagittal computed tomography images. Patients with vertical growth >2 mm were included in the vertical progression group. Segments with a thickness progression >1 mm were classified as thickness progressed segments, and patients who had at least one progressed segment were included in the thickness progression group. Based on the characteristics at each disc level, the ossified mass at each segment was classified into four types: type 1, no disc space involvement; type 2, involving the disc space, but not crossing; type 3, crossing the disc space, but not fused; and type 4, completely fused. RESULTS: The progression of ossified mass was observed in younger patients (P < 0.01) and in C2-C3 involvement (P < 0.01) cases. Moreover, progression in both directions was observed more frequently in the mixed-type OPLL (P < 0.01). Progression occurred most often in type 3 segments (72.0%, P < 0.01). In type 3 segments, thickness progression was found more frequently in segments with segmental range of motion (ROM) ≥5° (55.6% vs. 27.8%, P = 0.04). The proportion of segments whose initial thickness was >5 mm was significantly higher among progressed segments (60.0% vs. 35.2%, P = 0.03). CONCLUSION: Young age, C2-C3 involvement, and mixed-type OPLL are risk factors for OPLL progression. Segments with morphology of crossing the segment, but without fusion (type 3), segmental ROM ≥5°, and initial thickness >5 mm need special attention. LEVEL OF EVIDENCE: 3.

Calcification of the intervertebral disc and ossification of posterior longitudinal ligament in children.

BACKGROUND: IDC in children, first reported by Baron in 1924, is very rare. OPLL of the cervical spine mainly affect people ages 50-70 years. The coexistence of IDC and OPLL in children is very rare, only six cases with 3 to 24 months' follow-up were reported to date. CASE PRESENTATION: A 6-year-old boy presented with complains of neck pain at July 2007. The boy was treated by conservative treatment and observed up for 9 years. Neck pain greatly improved after a one-month conservative treatment and never recur. Laboratory tests revealed elevated ESR and CRP at admission and found nothing abnormal at 19-month and 9-year follow-up. Computed tomography and magnetic resonance imaging revealed IDC at C2/3, C3/4 and OPLL at C3/4 at admission and found minor calcification at C2/3 remained but calcification at C3/4 and OPLL at C3/4 completely disappeared at 19-month and 9-year follow-up. Nineteen months after initial diagnosis, restoration of T2-weighted signal intensity of C2/3 and C3/4 discs was observed through MRI. Loss of T2-weighted signal intensity of C2/3 disc and decrease of T2-weighted signal intensity of C3/4 disc was observed at 9-year follow-up. CONCLUSIONS: IDC with OPLL in children is very rare. Conservative treatments are recommended with affirmative short-term and long-term clinical effects. More intensive observation with long-term follow-ups may be needed to warrant the clinical effects.

Clinical image of anterior longitudinal ligament syndrome - an interdisciplinary problem.

INTRODUCTION: Approximately 1.5-2.5% of all patients treated due to discopathy have anterior longitudinal ligament lesions. An intervertebral disc moving under the anterior longitudinal ligament causes ligament displacement and irritation of the autonomic nervous system structures, resulting in a disturbed function of the organs controlled by this system. Clinical image: Increased sympathetic system activity in the thoracic section may cause symptoms that mimic coronary heart disease while irritation of the autonomic structures in the lumbosacral section of the spine results in a clinical presentation similar to that of gastrointestinal or gynecological disorders. The clinical image of the most common disorders is presented. The diagnosis may be formulated with the use of magnetic resonance imaging. TREATMENT: pharmacotherapy is ineffective. Physiotherapy and rehabilitation constitute a method of choice in the treatment of this syndrome.

[Ossification of the posterior longitudinal ligament (OPLL) and the ligamentum flavum (OLF)].

The adult population is frequently sustained with ossification of the posterior longitudinal ligament (OPLL) and/or the ligamentum flavum (OLF) of the spine and the diseases may subsequently result is serious spinal cord insult with profound paralysis of the extremities. These pathologies are genetically denoted metaplasia of the elastic fibers of the ligament with consequent ectopic ossification process. The spinal cord is chronically compressed and will result in profound motor paralysis with sensory deficit. Both diseases are well imaged on CT and MRI, showing a various magnitude of spinal cord compression.

[Ossification of posterior longitudinal ligament].

Ossification of posterior longitudinal ligament (OPLL) is a hyperostotic disease of the spine associated with myelopathy which is occurred by an anterior compression to the spinal cord. OPLL was first reported by Key GA in 1838, and was previously considered specific to east Asian people, especially Japanese. However, now OPLL is recognized as a subtype of diffuse idiopathic skeletal hyperosteosis, which is detected in Europe and the United States. We discuss the etiology and natural history of OPLL in this review.

Laminoplasty versus conservative treatment for acute cervical spinal cord injury caused by ossification of the posterior longitudinal ligament after minor trauma.

BACKGROUND CONTEXT: No reports to date have accurately evaluated the management for acute spinal cord injury (SCI) caused by ossification of the posterior longitudinal ligament (OPLL) after minor trauma. PURPOSE: To assess whether outcomes of laminoplasty is better than conservative treatment. STUDY DESIGN/SETTING: A retrospective study. PATIENT SAMPLE: Thirty-one patients underwent surgery (L group) and 29 patients underwent conservative treatment (C group). OUTCOME MEASURES: Disability, muscle strength, sensation, and general health status. METHODS: Patients were managed according to routine clinical practice and the results between groups were compared. Clinical and radiographic outcomes were assessed at admission, discharge, 6 months and at the final visit. Causes for trauma, duration of hospital stay, and complication were also evaluated. RESULTS: Causes for trauma included falling, traffic accidents and sports. Mixed and segmental types were the most frequent cause of OPLL resulting into SCI. Duration of hospital stay and complications were less in the L group. Motor and sensory scores increased in the L group at discharge (p<.05) and at 6 months (p<.05), and maintained thereafter (p>.05); scores improved significantly in the C group at 6 months (p<.05), with a slight deterioration with time (p>.05); scores in the L group were higher than in the C group at each time point after surgery (p<.05). Bodily pain and mental health in SF-36 improved at discharge in the L group (p<.05); all scores improved at 6 months in both the groups (p<.05), with better improvements in the L group (p<.05). The canal diameter increased and occupation ratio decreased in the L group (p<.05), and maintained thereafter (p<.05); a slight increase of occupation ratio was observed in the C group (p>.05). Lordotic angle and range of motion were maintained in both the groups, with no significance between groups (p>.05). High-signal intensity decreased at 6 months (p<.05) in the L group; no significant change was found in the C group during the follow-up (p>.05); Significant difference was detected between the groups at 6 months and at the final visit (p<.05). CONCLUSIONS: Most of the OPLL patients displayed as incomplete SCI after minor trauma. Although spontaneous improvement of SCI without surgery is often observed, laminoplasty has more satisfactory outcomes, prevents late compression of cord, and reduces perioperative complications, although with no significant benefit in cervical alignment and range of motion.

Proline/arginine-rich end leucine-rich repeat protein converts stem cells to ligament tissue and Zn(II) influences its nuclear expression.

Our objective was to facilitate ligament tissue reconstruction by characterizing the mechanism of expression of ligament tissue. To accomplish this, we searched for proteins specific to the tissue and introduced them into mesenchymal stem cells. In the two-dimensional phosphorescent gel electrophoresis, the spots in common with the normal human ligament tissue were selected after removing the spots of the normal bone tissue from those of the ossified tissue in the spinal ligament. Proline/arginine-rich end leucine-rich repeat protein (PRELP) was identified in ligament-specific locations by liquid chromatography-tandem mass spectrometry. Transfection of PRELP into mouse mesenchymal stem cells yielded ligament-like connective tissue comprised of parallel fibers. Thus, expression of the PRELP protein could reconstruct the ligament tissue. Since zinc-related proteins were found with high incidence as a result of an array analysis of PRELP's ProtoArray, it was considered that there is a relationship to the zinc metabolism. Tissue induction was mediated by the tumor necrosis factor (TNF)-α via the zinc pathway. PRELP may be a useful gene in syndesmoplasty, provided zinc is present for tissue reconstruction. Chromosome division becomes active with the addition of zinc, and rapid tissue induction takes place in the presence of zinc and TNF-α. Currently, the reconstruction of a ruptured ligament tissue is difficult, but we expect that the PRELP protein expression may facilitate this process. This study describes the discovery of the gene responsible for the differentiation of stem cells into ligament tissue. This important finding may lead to treatments for gonarthrosis, cruciate ligament, and periodontal ligament ruptures, and ossification of the posterior longitudinal ligament.

Conservatively treated ossification of the posterior longitudinal ligament increases the risk of spinal cord injury: a nationwide cohort study.

The optimal treatment strategy for ossification of the posterior longitudinal ligament (OPLL) depends on symptoms and is uncertain. Whether the risk of spinal cord injury (SCI) is increased in patients with cervical spinal stenosis or myelopathy caused by OPLL remains unclear. This study aimed to evaluate the risk of SCI in patients with OPLL of the cervical spine when managed with conservative treatment (no surgery). Study subjects were identified from a nationwide cohort of 26,544,883 people from 1998 to 2005 and were divided into the OPLL group (n=265), who were hospitalized for OPLL but had conservative treatment (no surgery), and the comparison group (n=5339), composed of age- and sex-matched people. Until the end of 2008, a total of 5604 subjects were followed-up for 34,723.5 person-years. The propensity score method was used to adjust for covariates. Kaplan-Meier and Cox regression analyses were performed. The incidence rate of cervical SCI in the OPLL group was found to be significantly higher than in the comparison group (4.81 versus 0.18 per 1000 person-years; p<0.001). Cervical SCI was more likely to happen in the OPLL group than in the comparison group (crude hazard ratio [HR] 25.64; p<0.001). After adjustments, the OPLL group had a 32.16-fold (p<0.001) higher risk for cervical SCI. Disability caused by SCI had an even higher risk (HR=110.72, adjusted HR=104.78; p<0.001) for the OPLL group. Therefore, cervical SCI and related disabilities are more likely to happen in OPLL patients, who should be cautioned for subsequent SCI if treated conservatively.

Ossification of the posterior longitudinal ligament: pathogenesis, management, and current surgical approaches. A review.

Ossification of the posterior longitudinal ligament (OPLL) is an important cause of cervical myelopathy that results from bony ossification of the cervical or thoracic posterior longitudinal ligament (PLL). It has been estimated that nearly 25% of patients with cervical myelopathy will have features of OPLL. Patients commonly present in their mid-40s or 50s with clinical evidence of myelopathy. On MR and CT imaging, this can be seen as areas of ossification that commonly coalesce behind the cervical vertebral bodies, leading to direct ventral compression of the cord. While MR imaging will commonly demonstrate associated changes in the soft tissue, CT scanning will better define areas of ossification. This can also provide the clinician with evidence of possible dural ossification. The surgical management of OPLL remains a challenge to spine surgeons. Surgical alternatives include anterior, posterior, or circumferential decompression and/or stabilization. Anterior cervical stabilization options include cervical corpectomy or multilevel anterior cervical corpectomy and fusion, while posterior stabilization approaches include instrumented or noninstrumented fusion or laminoplasty. Each of these approaches has distinct advantages and disadvantages. While anterior approaches may provide more direct decompression and best improve myelopathy scores, there is soft-tissue morbidity associated with the anterior approach. Posterior approaches, including laminectomy and fusion and laminoplasty, may be well tolerated in older patients. However, there often is associated axial neck pain and less improvement in myelopathy scores. In this review, the authors discuss the epidemiology, imaging findings, and clinical presentation of OPLL. The authors additionally discuss the merits of the different surgical techniques in the management of this challenging disease.

Diffusion tensor imaging in the assessment of ossification of the posterior longitudinal ligament: a report on preliminary results in 3 cases and review of the literature.

Cervical spondylotic myelopathy due to ossification of the posterior longitudinal ligament (OPLL) is a common neurosurgical disease that carries high morbidity. OPLL and other degenerative processes cause narrowing of the central canal, with subsequent spinal cord injury. Repeated minor trauma and vascular aberrations have been purported to underlie cervical spondylotic myelopathy, although the exact pathophysiological mechanism is unclear. Regardless, detection of early axonal damage may allow more timely surgical intervention and prediction of functional outcome. Diffusion tensor (DT) imaging of the cervical spine is a novel technique with improved sensitivity compared with conventional anatomical MR imaging that is currently available on most clinical scanners. This review describes the theoretical basis, application, and analysis of DT imaging as it pertains to neurosurgery. Particular emphasis is placed on OPLL.

Management of cerebrospinal fluid leaks after anterior decompression for ossification of the posterior longitudinal ligament: a review of the literature.

OBJECT: Anterior decompression is an effective way to treat cervical myelopathy associated with ossification of the posterior longitudinal ligament (OPLL); however, this approach is associated with an increased risk of a dural tear and resultant CSF leak because fusion of the dura with the ossified PLL is common in these cases. The authors review the literature and present an algorithm for treatment of CSF leaks in these patients. METHODS: A MEDLINE review was performed to identify papers related to CSF leak after anterior decompression for OPLL, and data were summarized to identify treatment options for various situations. A treatment algorithm was identified based on these findings and the experience of the authors. RESULTS: Eleven studies were identified that presented data on intra- and postoperative management of a CSF leak during ventral surgery for OPLL. The incidence of cervical dural tears and CSF leaks after anterior decompression procedures for OPLL ranged from 4.3% to 32%. Techniques including preventative measures, intraoperative dural repair with various materials, and postoperative drainage or shunt placement have all been used. CONCLUSIONS: Although direct dural repair is the preferred treatment for CSF leak, this technique is not always technically possible. In these cases, intraoperative adjuncts in combination with postoperative measures can be used to decrease the pressure gradient across the dural tear.