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PubMed2002

Large-scale screening for candidate genes of ossification of the posterior longitudinal ligament of the spine.

Ossification of the posterior longitudinal ligament of the spine (OPLL) is the predominant myelopathy among Japanese, and is usually diagnosed by ectopic bone formation in the paravertebral ligament in Japanese and other Asians. To detect genetic determinants associated with OPLL, we performed an extensive nonparametric linkage study with 126 affected sib-pairs using markers for various candidate genes by distinct analyses, SIBPAL and GENEHUNTER. Eighty-eight candidate genes were selected by comparing the genes identified by complementary DNA (cDNA) microarray analysis of systematic gene expression profiles during osteoblastic differentiation of human mesenchymal stem cells with the genes known to be involved in bone metabolism. Of the 24 genes regulated during osteoblastic differentiation, only one, the alpha B crystalline gene, showed evidence of linkage (p = 0.016, nonparametric linkage [NPL] score = 1.83). Of 64 genes known to be associated with bone metabolism, 7 showed weak evidence of linkage by SIBPAL analysis (p < 0.05): cadherin 13 (CDH13), bone morphogenetic protein 4 (BMP4), proteoglycan 1 (PRG1), transforming growth factor beta 3 (TGFb3), osteopontin (OPN), parathyroid hormone receptor 1 (PTHR1), and insulin-like growth factor 1 (IGF1). Among these genes, BMP4 (NPL = 2.23), CDH13 (NPL = 2.00), TGFb3 (NPL = 1.30), OPN (NPL = 1.15), and PTHR1 (NPL = 1.00) showed evidence of linkage by GENEHUNTER. Only BMP4 reached criteria of suggestive evidence of linkage. Because this gene is a well-known factor in osteogenetic function, BMP4 should be screened in further study for the polymorphism responsible.

Animals↗
PubMed2005

Desmoid tumor with ossification in chest wall: possible involvement of BAMBI promoter hypermethylation in metaplastic bone formation.

UNLABELLED: A rare case of desmoid-type fibromatosis with focal metaplastic bone in the chest wall suggested that enhanced responsiveness to BMP signaling by decreasing BAMBI expression through promoter hypermethylation plays a crucial role in the formation of metaplastic bone. INTRODUCTION: Desmoid-type fibromatosis, originating from mesenchymal cells with myofibroblastic features, is a locally aggressive and frequently recurring infiltrative lesion. One such sporadic case with metaplastic ossification in the chest wall is presented. MATERIALS AND METHODS: A 43-year-old man was referred to the hospital with a gradually enlarging hard mass in the left anterolateral chest wall. A thoracotomy was carried out, and histopathological specimens were used for immunohistochemical, genetic, and methylation studies. RESULTS: Accumulation of altered beta-catenin associated with a somatic heterozygous activating mutation in codon 41 was detected in the typical desmoid-type fibromatosis and at the ossifying focus. Among factors related to bone formation and the classical wnt-beta-catenin signaling pathway, BMP and activin membrane-bound inhibitor (BAMBI) expression was specifically downregulated at the ossifying focus. Hypermethylation of the BAMBI promoter was observed in microdissected tissue from the ossifying focus but not in that from the typical desmoid-type fibromatosis. CONCLUSIONS: Because both BMP and classical Wnt/beta-catenin/LEF1 signaling cooperatively and mutually induce differentiation of mesenchymal cells into osteoblastic cells and promote bone formation, the epigenetic event leading to the enhanced responsiveness to BMP signaling may play a crucial role in the formation of metaplastic bone.

Adult↗
PubMed1992

Extensive diffuse pulmonary ossification.

Diffuse lung injury can either heal normally or progress to fibrosis. Calcification in the setting of fibrosis is common. The appearance of mature woven bone is not. We report a patient with extensive diffuse pulmonary ossification and discuss some of the theories relating to the development of this phenomenon.

Adult↗
PubMed1993

Heterotopic ossification complicating critical illness.

Heterotopic ossification (HO), deposition of para-articular ectopic bone, is associated with musculoskeletal trauma and certain congenital and metabolic disorders. Additionally, HO may follow paralysis from diverse traumatic and nontraumatic neurologic insults. We describe three cases of HO associated with catastrophic nontraumatic respiratory illness requiring prolonged chemical paralysis and cardiorespiratory support.

Adult↗
PubMed1998

[Ossification of the posterior longitudinal ligament in the cervical spine: case report].

Ossification of the posterior longitudinal ligament (OPLL) is an uncommon cause of compressive myelopathy in the Caucasian population. A case of spastic paraparesis in a Caucasian man whose radiological investigation showed OPLL is presented. The radiographs of the cervical spine showed a strip of bony density posterior to the vertebral bodies, extending from C2 to T1. Computerized tomography (CT) and CT myelography showed OPLL at the same level. Magnetic resonance showed an area of increased signal on T2-weighted sequences at C7-T1 level suggestive of myelomalacia. The patient underwent an open-door laminoplasty (C2 to C7) with improvement of the paraparesis. OPLL should be included in the differential diagnosis of cervical myelopathy. It can be easily detected by plain radiographs and CT of the cervical spine. A review of the clinical and radiological features and the treatment of OPLL is presented.

Humans↗
PubMed1998

Results of surgical treatment for ossification of the posterior longitudinal ligament of the thoracic spine.

Conservative treatment is ineffective for ossification of the posterior longitudinal ligament (OPLL) in the thoracic spine, and surgical treatment is indicated for most cases, while such cases are not often experienced. In the present study, the results of surgical management involving mainly posterior decompression for this disease were evaluated clinically. The study included 9 patients (1 man and 8 women) who underwent surgical treatment for OPLL of the thoracic spine between 1984 and 1993. Laminectomy was performed in 5 patients, and laminectomy plus anterior decompression of the OPLL via the posterior approach based on Otsuka's method was performed in 2 patients. In 1 patient, laminoplasty for OPLL of the cervical spine was combined with laminectomy of the symptomatic lesion in the thoracic spine. One patient underwent anterior decompression and fusion. The results were evaluated using the Japanese Orthopaedic Association score (JOA score) and recovery rate. The postoperative follow-up period ranged from 1 year to 10 years and 3 months (mean, 4 years and 6 months). The mean JOA score was 4.8 before surgery and improved to 7.6 at the final examination. This was a mean recovery rate of 50.1%. Symptoms caused by OPLL in the thoracic spine can be alleviated by posterior decompression where OPLL extends from the upper to the middle thoracic spine or extends from the middle to the lower thoracic spine. It seems, however, that OPLL localized to the middle thoracic spine requires anterior decompression.

Adult↗
PubMed1991

[Ossification of the thoracic ligamentum flavum presenting with intercostal neuralgia. Case report].

A 46-year-old female was admitted complaining of progressive, severe girdle pain consistent with the left Th3 dermatome. Neurological examination on admission revealed dysesthesia and radiating pain in the left Th3 territory. Plain X-rays and tomograms of the thoracic spine revealed a beak-like bony excrescence arising from the lamina and projecting moderately to the Th3/4 intervertebral foramen, suggesting ossification of the thoracic ligamentum flavum (OYL). Myelography showed the dural sac compressed from laterally just below the left Th3 pedicle, which suggested that the Th3 nerve root was compressed by the OYL. After Th3 nerve root decompression through hemilaminectomy and foraminotomy, the girdle pain disappeared. OYL is known to cause thoracic radiculomyelopathy, but presentation with intercostal neuralgia only is very rare. The authors review the literature and stress the importance of myelography for diagnosis.

Female↗
PubMed2005

Cervical cord compression secondary to ossification of the posterior longitudinal ligament.

STUDY DESIGN: Resident's case problem. BACKGROUND: A 52-year-old Chinese male with a 10-year history of gradually worsening right hip stiffness, weakness, and pain was referred to physical therapy by his orthopedist, who made a diagnosis of developmental dysplasia of the right hip, with possible Legg-Calve-Perthes disease. The patient reported multiple falls over the last several years and a gradual onset of low back pain with an onset of "electricity" down both legs. The patient also reported mild numbness in both forearms and the right hand over the previous several months. This resident's case problem illustrates how a physical therapist recognized the presence of an atypical musculoskeletal pathology through the use of hypothesis-driven clinical reasoning and detailed physical examination. DIAGNOSIS: Examination of the patient's lumbar and cervical spine and hips revealed joint dysfunctions. Neurological testing revealed hyperreflexia. Special testing revealed lower extremity clonus with a positive Babinski sign with gait disturbances. The patient was referred back to his primary physician and then to a neurologist and neurosurgeon. An MRI revealed cervical myelopathy due to ossification of the posterior longitudinal ligament from C3/C4 to C5/C6. The patient then underwent a C3 through C7 laminectomy. DISCUSSION: It is always imperative that sound clinical reasoning be used when performing physical therapy evaluations, regardless of the referral status of the patient. Patients with nonmusculoskeletal pathology may seek physical therapy services and it is the physical therapist's responsibility to complete a thorough examination and refer to specialists when appropriate.

China↗
PubMed1993

Ossification of the ligamentum flavum in a Caucasian: case report.

Ossification of the ligamenta flava (OLF) and posterior longitudinal ligament (OPLL) of the spine causing spinal cord compression is described mainly in Japanese patients and is often termed as 'Japanese disease'. We are reporting a case of OLF of the thoracic spine seen in a male Caucasian who presented with an intractable intercostal pain. This is the first case report of OLF in a Caucasian.

Adult↗
PubMed1994

Ossification of the posterior longitudinal ligament mimicking cord compression from a dorsal disc protrusion: a cautionary tale.

A patient with focal anterior compression of the dorsal cord at the level of the D6/7 disc was mistakenly diagnosed as a dorsal disc protrusion on the basis of CT myelography. The CT scanning had been confined to axial views at the level of the cord compression. A transthoracic exploration failed to reveal a disc protrusion and the correct diagnosis, ossification of the posterior longitudinal ligament, was only revealed by a subsequent sagittal CT scan. This case emphasizes the dangers of relying on limited axial imaging in diagnosing a spinal lesion.

Diagnostic Errors↗
PubMedDate not supplied

The relationship of heterotopic ossification to passive movements in paraplegic patients.

Heterotopic ossification (HO) occurs in 4-49% of patients with spinal cord injury, but the cause of the complication has not been established. The aim of this study was to investigate clinical factors related to the occurrence of HO. The incidence of HO was determined in 91 consecutive patients with traumatic lesions of the spinal cord who had been admitted to the National Spinal Injuries Centre for management and rehabilitation. Clinical data were analysed. Clinically apparent HO occurred only in 10 of 56 patients in whom the start of passive movements to their paralysed limbs was delayed until 7 days or more from time of injury. The findings of this study are consistent with the view that HO occurs as a result of trauma induced by passive movements carried out on joints where contractures have started to develop.

Adolescent↗
PubMed1993

Noncollagenous proteins in heterotopic ossification. Immunohistochemical analysis in 15 paraplegies.

We used immunohistochemical techniques to investigate the distribution pattern of osteonectin, osteocalcin, bone sialoprotein II and the small proteoglycans decorin and PG 100 during different stages of heterotopic ossification (HO) in pressure sores of paraplegic patients. All these noncollagenous proteins (NCPs) accumulated in fibroblasts and preosteoblasts, predominantly in the activity centers of early osteogenetic areas. Mature types of HO showed a more discrete expression pattern for this protein group, with weaker reactions in the narrow osteoblastic rims. Decorin was detected predominantly in the stroma of HO. Our results indicate that the NCPs are important components during the pathogenesis of HO and that fibroblasts may serve as osteoprogenitor cells.

Adult↗
PubMed1998

Indomethacin for prevention of heterotopic ossification after hip arthroplasty. A randomized comparison between 4 and 8 days of treatment.

In a randomized, parallel group study, we evaluated the efficacy of a 4-day versus an 8-day course of indomethacin (50 mg, 3 times per day), given as prophylaxis against heterotopic ossification after cementless total hip arthroplasty in 209 patients with arthrosis. Patients receiving the prophylaxis for 8 days had less (p = 0.03) severe heterotopic bone formation.

Anti-Inflammatory Agents, Non-Steroidal↗
PubMed1998

Preoperative irradiation for prophylaxis of ectopic ossification after hip arthroplasty. A randomized study in 62 hips.

62 hips in 57 patients who were all considered to be at risk for ectopic bone formation were randomly divided into two groups: one control group and one group who received single dose radiotherapy of 5 Gy at the operation field preoperatively. With a mean follow-up of 2.5 years, the incidence of heterotopic ossification in the control group was 16 of the 19 hips and in the irradiated group 6 of the 43 hips (p = 0.001).

Adult↗
PubMed2001

Outcome analysis of anterior decompressive surgery and fusion for cervical ossification of the posterior longitudinal ligament: report of 107 cases and review of the literature.

OBJECT: This study was designed to determine the surgical technique and surgery-related outcome, fusion rate, and complication of anterior decompression and fusion (with various graft materials) performed in patients with ossification of the posterior longitudinal ligament (OPLL) of the cervical spine. METHODS: Between 1980 and 1998, 107 patients with radiologically proven OPLL underwent surgery via the anterior approach for direct removal of the ossified mass. Graft materials included iliac crest in 45 cases, vertebral body (VB) in 37 cases; and interbody fusion cages in 25 cases. In four patients with three-level VB grafts and one with a two-level VB graft, anterior plates were placed. Surgery-related outcome was excellent or good in 89% and fair in 11%. This clinical improvement correlated well with the severity of preoperative myelopathy. Only one patient with severe myelopathy due to extensive mixed-type OPLL developed a segmental weakness of the bilateral upper extremities. The overall fusion rate was 97%. Three patients with obvious spinal instability due to pseudarthrosis required reoperation. Of the graft materials used in this series, VB grafts were the most fragile. CONCLUSIONS: The anterior approach is an effective route for decompressing the cervical cord with OPLL. Slight asymptomatic kyphotic deformity may be encountered. Of the graft materials used in our series, VB graft was considered most fragile, and thus least optimal.

Adult↗
PubMed2002

Anterior decompression for cervical spondylosis associated with an early form of cervical ossification of the posterior longitudinal ligament.

OBJECT: The goal of this study was to determine the appropriate surgical strategy for cervical spondylosis associated with an early form of ossification of the posterior longitudinal ligament (EOPLL) of the cervical spine. METHODS: Patients with EOPLL-associated cervical spondyosis were selected for treatment. Medical records and radiographs were retrospectively reviewed. Specimens taken at the time of operation were histologically examined. There were 24 men and six women ranging in age from 39 to 74 years (mean 57.6 years). Symptoms consisted of myelopathy in 28 cases and radiculopathy in two cases. Anterior decompressive surgery was performed. The EOPLL, hypertrophy of the posterior longitudinal ligament (HPLL), and the disc-PLL complex were directly resected. The mean preoperative Japan Orthopaedic Association score was 12.6, and the mean postoperative score was 14.4. Histologically, EOPLL was consistent with foci of compact lamellar bone in the degenerative thickening of the PLL. CONCLUSIONS: Appropriate corpectomy should follow direct removal of EOPLL associated with HPLL compressing the spinal cord to achieve good outcomes.

Adult↗
PubMed1984

Ultrastructure of early calcification in cervical ossification of the posterior longitudinal ligament.

A case of ossification of the cervical posterior longitudinal ligament was investigated with the electron microscope. The posterior longitudinal ligament was composed of bundles of collagen fibers intermingled with occasional fibroblasts and rare blood vessels. Some ligaments contained matrix vesicles in the vicinity of degenerated cells. Hydroxyapatite crystals were frequently precipitated within the matrix vesicles. These findings are similar to the fine structure of the early stage of calcification in normal and pathological calcifying tissues described previously. In this study, the calcification process of the posterior longitudinal ligament suggests that matrix vesicles originate from degenerated cells, and acquire hydroxyapatite crystal deposits. Some eventually coalesce to form a large calcifying mass. Substantial amounts of collagen fibers comprising the ligament may serve an important role in orienting apatite crystal precipitation.

Humans↗