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PubMed2002

Abnormal collagen fibrils in tendons of biglycan/fibromodulin-deficient mice lead to gait impairment, ectopic ossification, and osteoarthritis.

Small leucine-rich proteoglycans (SLRPs) regulate extracellular matrix organization, a process essential in development, tissue repair, and metastasis. In vivo interactions of biglycan and fibromodulin, two SLRPs highly expressed in tendons and bones, were investigated by generating biglycan/fibromodulin double-deficient mice. Here we show that collagen fibrils in tendons from mice deficient in biglycan and/or fibromodulin are structurally and mechanically altered resulting in unstable joints. As a result, the mice develop successively and progressively 1) gait impairment, 2) ectopic tendon ossification, and 3) severe premature osteoarthritis. Forced use of the joints increases ectopic ossification and osteoarthritis in the double-deficient mice, further indicating that structurally weak tendons cause the phenotype. The study shows that mutations in SLRPs may predispose to osteoarthritis and offers a valuable and unique animal model for spontaneous osteoarthritis characterized by early onset and a rapid progression of the disease.

Animals↗
PubMed1999

Heterotopic mesenteric ossification ('intraabdominal myositis ossificans'): report of five cases.

Intraabdominal heterotopic ossification is a very uncommon disorder. We report five new cases, review the previous literature, and discuss the clinical and pathologic features of these lesions. The clinical features of the current cases and of those previously reported are remarkably similar. All patients were middle-aged to elderly men (range, 43-80 years; mean, 61 years) who had small bowel obstruction associated with heterotopic bone formation in the small bowel mesentery, often after one or more abdominal operations. In one case, an initial diagnosis of extraosseous osteosarcoma was considered. This unusual reactive process shares many of the clinical and pathologic features of myositis ossificans, as classically described in somatic soft tissues. We propose to designate this condition heterotopic mesenteric ossification.

Adult↗
PubMed2001

Continuous passive motion in the management of heterotopic ossification in a brain injured patient.

We report a man admitted to inpatient rehabilitation 6 wk after traumatic brain injury, who presented with bilateral knee heterotopic ossification. In addition to conventional physical therapy, we applied a continuous passive motion device during 4 wk increasing the range of motion of the knees. On the basis of the limited current literature and this case, we suggest that the use of continuous passive motion devices for heterotopic ossification may be effective and safe and should be the subject of further study.

Adult↗
PubMedDate not supplied

Elbow anchylosis due to postburn heterotopic ossification.

In a retrospective study performed on 2280 patients that had had major thermal injuries, six patients experienced elbow anchylosis due to heterotopic ossification (two of them bilaterally). In every patient the ossification was localized at the posterior-medial aspect of the elbow. The treatment of this complication consisted of resection of the bone mass and transposition of the ulnar nerve. Mobilization was begun early, and rehabilitation was kept up for at least 4 months. The results obtained have been evaluated following the criteria of Merle d'Aubigné and Cañadell, who consider a very good result a gain of more than 60% in the arch of flexion-extension of the elbow; good, between 40% and 60%; fair between 20% and 40%; and poor, a gain of less than 20%. In five of the patients (six elbows), the result was very good with an average gain of 73.6% (minimum, 61.5%, maximum, 84.6%). In one patient the result was poor with gain of less than 20%. The incidence of this complication, its treatment, and the results obtained are analyzed.

Adult↗
PubMedDate not supplied

Ossification of a rectal tumor: CT evaluation.

Adenocarcinoma of the colon is a common malignancy. Not infrequently, this tumor can calcify. An extremely rare occurrence in these tumors is ossification. This is the first reported case of rectal carcinoma in which ossification was demonstrated with CT.

Adenocarcinoma↗
PubMedDate not supplied

Intraarticular heterotopic ossification in the knee following intramedullary nailing of the fractured femur using a retrograde method.

The cases of a forty-five-year-old woman and a twenty-year-old man who developed severe intraarticular and periarticular heterotopic ossification around the knee following intramedullary nailing of a femur fracture using a retrograde technique. The association of musculoskeletal heterotopic ossification with closed head injuries seems well established and can occur in and around the knee following retrograde intramedullary nailing. This complication may occur more often than has been reported.

Accidents, Traffic↗
PubMed1988

Heterotopic ossification around the hip with intramedullary nailing of the femur.

Heterotopic ossification (HO) at the proximal end of a reamed intramedullary femoral nail is a complication of the procedure. Kuntscher stated that these "callus caps" were due to the prominence of the nail above the greater trochanter. A study of patients undergoing locked intramedullary nailing of the femur to assess subsequent development of heterotopic ossification at the proximal end of the implant was done. The purpose was to study the clinical significance of the ectopic bone and identify etiologic factors of bone formation. This group consisted of 59 patients with 60 locked intramedullary nails who were reviewed after prospective clinical and radiologic followup. Ectopic bone around the proximal end of the nail was classified with respect to quantity and clinical importance. A number of parameters were evaluated and a multifactorial analysis was performed. There were 52 males and seven females. Heterotopic bone was graded as: none--32%; minimal--20%, mild--28%; moderate--15%; and severe--5%. There was a positive correlation between bone formation and head injury, Injury Severity Score, and ventilator and ICU days. Prominence of the nail was not significant.

Adolescent↗
PubMed2002

Surgical approaches for nonneurogenic elbow heterotopic ossification with ulnar neuropathy.

BACKGROUND: Few reports are available concerning elbow heterotopic ossification (HO) and its optimal management in nonneurologic or nonburn patients after repetitive elbow manipulation. The unique anatomic relationship of the ulnar nerve at the elbow renders it rather vulnerable to injury when elbow HO occurs medially or posteromedially and extends into the vicinity of the cubital tunnel. METHODS: A total of 16 consecutive patients without neurologic injury were diagnosed with heterotopic ossification formation in the elbow and referred to the Upper Extremity Unit of Chang Gung Memorial Hospital. All 16 patients were diagnosed with ulnar palsy, and 14 were found to have sensory dysfunction and muscle wasting for an average of 5.2 months. RESULTS: Fourteen of 16 patients achieved functional range of motion or more after surgery. The final gain in range of motion averaged 80.3 degrees. All except one had complete neurologic recovery postoperatively. The patient who had ulnar palsy for 1 year presurgery did not achieve any neurologic recovery. CONCLUSION: Forceful and repetitive manipulation may add further damage to an already stiffened elbow and should be avoided in an elbow after immobilization or surgery. Early surgical resection of HO and ulnar nerve decompression followed by gentle and aggressive physical therapy terminate the vicious cycle and yield encouraging results.

Adolescent↗
PubMed1999

Osteoplastic anterolateral vertebrotomy without fusion for multilevel cervical ossification of the posterior longitudinal ligament.

OBJECTIVE: To introduce a new method for osteoplastic anterolateral vertebrotomy without fusion to resect multilevel cervical ossification of the posterior longitudinal ligament. METHODS: A multilevel vertebral column graft containing portions of the intervertebral discs is cut with a microsurgical saw from the anterolateral part of the vertebra via a thin lateral gutter placed in advance. A sufficiently wide oblique operative field is provided for resection of the ossified ligament with this method, and only a narrow bony defect remains after simple replacement of the vertebral graft. Postoperatively, patients are allowed to walk, usually within 2 to 3 days, wearing simple cervical collars. RESULTS: Twelve patients underwent resection of the ossified ligaments in two to five vertebral bodies. Clinical results were satisfactory except in one patient, who died as a result of a pulmonary embolism that occurred 10 days after surgery. In all of the other patients, the vertebral column grafts were fused within a few months, and there were no graft-related problems. Postoperative lateral radiographs revealed that cervical alignment was preserved, with intervertebral mobility at the operated segment in flexion-extension views. CONCLUSION: The osteoplastic anterolateral vertebrotomy method provided a sufficiently wide operative field for satisfactory resection of multilevel cervical ossification of the posterior longitudinal ligament, simplifying graft replacement and postoperative patient care. The cervical alignment was preserved.

Adult↗
PubMed2002

Heterotopic ossification of peripheral nerve ("neuritis ossificans"): report of two cases.

OBJECTIVE AND IMPORTANCE: Heterotopic ossification ("neuritis ossificans") is among the least frequently encountered reactive lesions in peripheral nerves. Only two cases have been described previously, one in the median nerve of a 34-year-old man, and the other in the ulnar nerve of an adult woman. The architecture of this lesion is distinctly zonal. Consisting of a central fibroblastic core, an intervening zone of osteoid production, and a peripheral layer of ossification, the pattern is remarkably similar to that of myositis ossificans. This similarity and the occurrence of the process in superficial nerves have led to speculation that trauma plays a role in its pathogenesis; this hypothesis remains unproved. We describe two additional cases of neuritis ossificans. CLINICAL PRESENTATION: One patient, a 41-year-old man, experienced pain and numbness in the left leg for several months but had no history of local trauma. A mass was detected in the saphenous nerve. The second patient, a 16-year-old boy, noted subacute onset of pain in the popliteal fossa and decreased sensation in the distribution of the lateral sural cutaneous nerve. A mass was found within the tibial nerve at the knee level. INTERVENTION: In each patient, resection of the mass required sacrifice of a segment of the nerve. CONCLUSION: In each patient, the mass was composed of fibrovascular tissue with osteoid and bone deposition arranged in a zonal pattern. The ossifying process was intraneural but encased rather than directly involving nerve fascicles. These exceptionally intact examples of neuritis ossificans underscore its resemblance to myositis ossificans. Nerve-sparing resection of such masses is not always possible.

Adolescent↗
PubMed1989

Genetic studies on ossification of the posterior longitudinal ligament of the spine.

To study the genetic background in ossification of the posterior longitudinal ligament of the spine (OPLL), 1,030 relatives of probands with cervical OPLL in 347 families were investigated by observing their spinal x-ray films, and the following results were obtained. Ossification of the posterior longitudinal ligament was found in 26.15% of the parents of the probands and 28.89% of the siblings of the probands. These prevalences were significantly higher than in the general population. The segregation rate in the siblings corresponded neither with the hypothesis of autosomal dominant inheritance nor with that of autosomal recessive inheritance. The possibility of polygene inheritance was also ruled out, considering the risk of recurrence in the siblings of the probands. However, owing to the segregation rate in the siblings exceeding 0.25 and the high prevalence of OPLL in the parents, OPLL was suggested to be a disorder possibly controlled by autosomal dominant inheritance.

Adult↗
PubMed1990

Circumspinal decompression for thoracic myelopathy due to combined ossification of the posterior longitudinal ligament and ligamentum flavum.

Ossification of the posterior longitudinal ligament (OPLL) combined with ossification of the ligamentum flavum (OLF) in the thoracic spine can result in serious myelopathy, leading to circumferential compression of the spinal cord in advanced stages of the disease. The authors performed circumspinal decompression (circumferential decompression of the spinal cord) on these patients. This operation consists of two steps: posterior and lateral decompression of the spinal cord by removal of the OLF (first step) and anterior removal of the OPLL for anterior decompression (second step), followed by interbody fusion. In the first step, two deep parallel gutters, covering the extent of the OPLL to be removed anteriorly, are drilled down from the rear into the vertebral body along both sides of the dura to easily and safely remove the OPLL anteriorly at the second step. In the second step, the surgical approach varies according to the affected level; costotransversectomy in the upper thoracic spine and standard thoracotomy in the middle or lower thoracic spine. According to the authors, circumspinal decompression is not an easy procedure, but from their results in 10 patients, they identify it as a radical and promising surgical procedure.

Adult↗
PubMed1994

Ossification of the posterior longitudinal ligament in evolution in 12 patients.

Ossification of the posterior longitudinal ligament (OPLL) in evolution (OEV), an early form of OPLL, was newly discovered in 12 of 43 (28%) patients who had OPLL and underwent operations from 1989-1992. Magnetic resonance imaging and CT studies of OEV patients who were younger, in their mid-forties, and had less severe spastic myeloradiculopathy, showed hypertrophied posterior longitudinal ligaments containing punctate ossification at disc spaces and contiguous endplates, with occasional extension behind vertebral bodies. However, both location and neuroradiologic appearance allowed OEV to be confused readily with disc or spondylotic disease. Only heightened awareness of the clinical and radiographic findings of OEV allowed the correct diagnosis. The appropriately extended anterior discectomies or corpectomies with fusions contributed to better outcomes.

Adult↗
PubMed1994

Evaluation of serum osteoblast mitogenic activity in spinal cord and head injury patients with acute heterotopic ossification.

STUDY DESIGN: This was a blind, prospective study of the effect of sera from patients with spinal cord and head injuries on osteoblast proliferation. OBJECTIVES: The authors studied whether a humoral factor that stimulates the formation of heterotopic bone is released into the circulation after a neural injury. BACKGROUND DATA: Other authors have shown that a humoral osteoinductive factor may be released after head and spinal cord injuries. METHODS: Serum was obtained at certain times throughout the first 12 weeks post-injury and from control subjects. It was incubated with osteoblasts harvested from fetal rats, as well as with fibroblast controls. RESULTS: There was a significant rise in serum mitogenic activity after injury in both groups. When patients that developed heterotopic ossification were compared to other patients and controls, no significant differences were seen. CONCLUSIONS: This in vitro study fails to support a humoral mechanism for heterotopic ossification after spinal cord or brain injuries.

Adult↗
PubMed1997

Paraplegia due to ossification of ligamenta flava in X-linked hypophosphatemia. A case report.

STUDY DESIGN: Spinal canal decompression at the most prominent of multiple posterior calcified thoracic lesions in a case of X-linked hypophosphatemia was undertaken for treatment and diagnosis purposes, as well as to assess possible nature of the pathophysiology underlying the presenting deficits. OBJECTIVES: To discuss the clinical assessment diagnostic and treatment aspects of this rare coincidence of ossification of ligamenta flava in the patient with the skeletal deformities of X-linked hypophosphatemia. SUMMARY OF BACKGROUND DATA: The patient with the stigmata and chemical findings of an X-linked hypophosphatemia presented with paraplegia and multiple calcified posterior spinal thoracic lesions. This was studied with magnetic resonance imaging and electrophysiologic studies of the spinal sensory pathways of the legs. These data constituted the preoperative information required to assess later results of surgical intervention. METHODS: Presurgical clinical, imaging and electrophysiologic studies and laboratory and pathologic investigations of the surgical specimens. RESULTS: Resolution of the paraplegia with walking and return to work in a physically demanding job for the last 4 or 5 years of postoperative follow-up after surgical decompression of the spinal cord only at the worst and highest of the effected spinal levels. CONCLUSION: The coincidence of X-linked hypophosphatemia and ossification of ligamenta flava has been reported only in two or three cases in the literature. Removal of the offending ossifying lesion is known to result in resolution of the clinical deficits but similar lesions at other spinal levels are suspected of producing recurrences. The return of function and of the corresponding electrophysiologic correlates indicate a neurono-apractic nature of the neurologic symptoms.

Bone and Bones↗
PubMed2005

The extent of ossification of posterior longitudinal ligament of the spine associated with nucleotide pyrophosphatase gene and leptin receptor gene polymorphisms.

STUDY DESIGN: A case-control study using radiograph findings and the PCR assay with regard to the susceptibility and the severity of ossification of posterior longitudinal ligament of the spine (OPLL). OBJECTIVE: To analyze whether polymorphisms of the nucleotide pyrophosphatase (NPPS) gene and the leptin receptor gene predispose to an increased frequency and severity of OPLL. SUMMARY OF BACKGROUND DATA: The NPPS gene is responsible for ectopic ossification in the ttw mouse, an animal model for OPLL. The Zucker fatty rat, another animal model for OPLL, has a missense mutation in the leptin receptor gene. METHODS: Analysis of 172 OPLL patients and 93 non-OPLL controls was performed. Radiographs of the cervical, thoracic and lumber spine were analyzed to determine whether OPLL was present and to what degree. Genomic DNA was extracted from all participants. Polymorphisms of the NPPS gene and the leptin receptor gene were analyzed using the PCR assay. The association of the polymorphisms with the development and extent of OPLL were statistically evaluated. RESULTS: No significant association was found between the polymorphisms and the existence of OPLL in both the NPPS and the leptin receptor genes. However, the IVS20-11delT variant in the NPPS gene and the A861G variant in the leptin receptor gene were more frequent in patients with OPLL in the thoracic spine compared with patients whose OPLL was restricted to the cervical spine. CONCLUSION: The present results suggest that the IVS20-11delT variant of the NPPS gene and the A861G variant of the leptin receptor gene are associated with more extensive OPLL, but not with the frequency with which it occurs.

Adenine↗
PubMed2005

COL6A1, the candidate gene for ossification of the posterior longitudinal ligament, is associated with diffuse idiopathic skeletal hyperostosis in Japanese.

STUDY DESIGN: Genetic screening of collagen 6A1 gene (COL6A1) in patients with diffuse idiopathic skeletal hyperostosis (DISH) recruited in Japan and the Czech Republic. OBJECTIVE: To investigate allelic associations between DISH and nucleotide variants of COL6A1. SUMMARY OF BACKGROUND DATA: DISH is a skeletal hyperostotic disease characterized by ligamentous ossification of the anterolateral side of the spine. Ossification of the posterior longitudinal ligament (OPLL) is a related disorder with DISH, and COL6A1 was identified as a susceptibility gene to OPLL. COL6A1 was examined for susceptibility in DISH patients from Japan and the Czech Republic. METHODS: Seven single nucleotide polymorphisms of COL6A1 were genotyped by direct sequencing. The allele frequencies were compared between 97 Japanese DISH patients and 298 Japanese controls, and between 96 Czech DISH patients and 96 Czech controls by chi2 test. RESULTS: The intron 32 (-29) single nucleotide polymorphisms of COL6A1 was significantly associated with the Japanese DISH patients (chi2 = 9.33; P = 0.0022), but not with the Czech DISH patients. CONCLUSIONS: Because COL6A1 could be a susceptibility to the occurrence of DISH and OPLL in the Japanese population, we consider that COL6A1 could be responsible for the hyperostotic state, leading to ectopic bone formation in the spinal ligament.

Aged↗
PubMed2004

Metaplastic ossification in a cutaneous pyogenic granuloma: a case report.

Cutaneous ossification may occur in association with a variety of cutaneous neoplasms and inflammatory conditions, such as pilomatricomas, basal cell carcinomas, nevi, chondroid syringomas, venous stasis, and scars. However, it has rarely been reported in pyogenic granuloma, a relatively common benign vascular tumor of the skin and mucous membranes. We herein presented a rare case of cutaneous pyogenic granuloma with ectopic ossification on the big toe of a 37-year-old man, with high recurrence despite repeated CO2 laser ablations. We propose the hypothesis that vascular endothelial growth factor (VEGF) and bone morphogenetic proteins (BMPs) play pathologic roles in the development of ectopic bone formation in pyogenic granuloma.

Adult↗