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Ossification of the ligamenta flava is rare and has been described almost exclusively in Japanese people. We present a case in which ossification of the thoracic ligamenta flava caused compressive myelopathy in a Caucasian.
The management of the profoundly deaf child with a cochlear implant poses a special challenge, particularly when total ossification of the cochlea is present. In this setting, insertion of an electrode array into a child's cochlea is often difficult. Our experience supports the feasibility of partial insertion of a multichannel implant into the basal turn of an ossified cochlea. Five children with ossified cochleae who had undergone partial implantation of a multichannel electrode were compared with the performance of matched controls who had full insertion of multichannel implants. No dramatic differences were detected during a 6- to 18-month follow-up period on selected test measures. These preliminary results suggest that active electrode number may exert a limited effect on performance with a cochlear implant. Drilling out the basal turn of an ossified cochlea in conjunction with partial insertion of a multichannel implant appears to be an acceptable surgical and rehabilitational alternative for placement of a cochlear implant prosthesis in children with complete cochlear ossification.
Estimations of serum alkaline phosphatase were carried out prospectively on a series of patients having a total hip replacement. The levels of serum alkaline phosphatase before operation indicated a group of patients who subsequently developed heterotopic ossification. Levels of this enzyme after operation did not indicate those patients who were developing heterotopic ossification.
In this series, 15 patients with ossification of the posterior longitudinal ligament underwent anterior decompression to relieve moderate or severe myelopathy, which in 11 included urinary disturbance. The operation consisted of partial resection of the vertebrae, release of the ossified plaque from the surrounding tissue and the insertion of an iliac bone graft. The extent of ossification was confirmed by computerised tomography before and after operation. The plaque was completely detached and moved forward in half of the patients, but only partially moved in the remainder. Symptoms improved considerably. Urinary disturbance disappeared in all patients, but sensory disturbance was left in most. Two patients had prolonged symptoms which were not relieved despite the complete release of the ossified defect.
Ossification of the ligamentum flavum and secondary spinal-cord compression were produced experimentally in mice by implanting bone morphogenetic protein (BMP) in the lumbar extradural space. The ligamentum flavum became hypertrophied and ossified, and protruded into the spinal canal. The thickness of the ossified ligament increased gradually with time, leading to compression and deformation of the spinal cord which showed various degrees of degeneration. Demyelination occurred in the posterior and lateral white columns and neuronal loss or chromatolysis in the grey matter. The pathological findings in the experimental animals closely resemble those found in the human disease and suggest that BMP may be a causative factor of ossification of the ligamentum flavum in man. This experimental model may be useful for the study of myelopathy caused by gradual spinal-cord compression.
We have carried out a prospective, randomised study of prophylaxis for heterotopic ossification (HO) comparing indomethacin for 7 and 14 days, acetylsalicylic acid, and fractional (4 x 3 Gy) or single exposure of 5 or 7 Gy irradiation after operation. We initially had 723 patients (733 hip replacements), but after withdrawals there were 685 hips of which 18.4% developed HO; 14% were grade I, 2.9% grade II and 1.5% grade III of the Brooker classification. We compared the results between these groups with those of a matched control series and found that indomethacin, 2 x 50 mg for 7 and 14 days, and postoperative irradiation of 4 x 3 Gy or 1 x 7 Gy, significantly reduced the development of HO compared with the control group. Patients in the acetylsalicylic acid group and those with a single irradiation of 5 Gy after operation developed significantly more ossification than those in the indomethacin and other irradiation groups. We suggest the use of 2 x 50 mg of indomethacin with mucoprotection for seven days as prophylaxis against HO after total hip replacement for all patients. A single irradiation of 7 Gy is recommended for patients who have developed HO after previous operations or to whom administration of indomethacin is contraindicated.
We present nine patients (five men and four women) who underwent surgical excision of clinically significant heterotopic ossification at the elbow. They also received perioperative radiation therapy using total doses between 600 and 1000 cGy. Five received fractionated radiotherapy, with two fractions of 500 cGy applied on the first two postoperative days, and the remaining four were irradiated with single doses of 600 and 700 cGy. After a mean period of observation of 7.7 months (6 to 13) none had radiological recurrence of heterotopic ossification and eight showed clinical improvement. Assessment of the functional outcome showed a mean improvement in the Morrey score from 33.3 to 84.5 points indicating a high therapeutic efficacy of prophylactic irradiation.
Progressive osseous heteroplasia (POH) is a rare disorder characterized by dermal ossification beginning in infancy followed by increasing and extensive bone formation in deep muscle and fascia. We describe two unrelated girls with typical clinical, radiographic, and histological features of POH who also have findings of another uncommon heritable disorder, Albright hereditary osteodystrophy (AHO). One patient has mild brachydactyly but no endocrinopathy, whereas the other manifests brachydactyly, obesity, and target tissue resistance to thyrotropin and parathyroid hormone (PTH). Levels of the alpha-subunit of the G protein (Gsalpha) were reduced in erythrocyte membranes from both girls and a nonsense mutation (Q12X) in exon 1 of the GNAS1 gene was identified in genomic DNA from the mildly affected patient. Features of POH and AHO in two individuals suggest that these conditions share a similar molecular basis and pathogenesis and that isolated severe extraskeletal ossification may be another manifestation of Gsalpha deficiency.
Ossification of the posterior longitudinal ligament (OPLL) of the spine is the leading cause of myelopathy in Japan. In earlier studies, we provided genetic linkage and allelic association evidence of distinct differences in the human collagen alpha2(XI) gene (COL11A2) that might constitute inherited predisposition to OPLL. In the present study, a strong allelic association with non-OPLL (p = 0.0003) was observed with an intron 6 polymorphism [intron 6 (-4A)], in which the intron 6 (-4A) allele is more frequently observed in non-OPLL subjects than in OPLL patients. In addition, a newly identified polymorphism in exon 6 [exon 6 (+28A)] was in linkage disequilibrium with the intron 6 (-4A). The functional impact of the polymorphisms was analyzed by comparing the differences in messenger RNA (mRNA) splicing by reverse-transcription polymerase chain reaction (RT-PCR) analysis in cultured cells from the interspinous ligament and an in vitro exon trapping study. The intron 6 (-4A) allele resulted in skipping exon 6 and retaining exon 7, while the exon 6 (+28A) allele was not associated with alteration in mRNA splicing. Similar mRNA species were observed in undifferentiated osteoblast (Ob) cells and in cells from posterior longitudinal ligament of non-OPLL subjects. The region containing exons 6-8 is an acidic subdomain presumably exposed to the surface that could interact with molecules of the extracellular matrix. Accordingly, retaining exon 7 together with removal of exon 6 observed in intron 6 (-4A) could play a protective role in the ectopic ossification process because the same pattern was observed in undifferentiated Ob cells and nonossified posterior longitudinal ligament cells.
BACKGROUND: Although uncommon, complete ankylosis of the elbow secondary to heterotopic ossification results in severe disability. The results of surgical management remain unclear. METHODS: A single surgeon used a consistent operative technique to treat complete osseous ankylosis of the elbow in eleven limbs in seven patients after severe burns and in nine elbows in eight patients after trauma. The elbows in the burn cohort were more often ankylosed in extension (average, 47 degrees of flexion) compared with those in the trauma cohort (66 degrees of flexion), and they had more skin problems (three elbows required a free microvascular muscle transfer for coverage) and associated problems of the shoulder, wrist, and hand. RESULTS: Four patients in the burn cohort and three patients in the trauma cohort failed to regain at least 80 degrees of ulnohumeral motion. After a repeat release in three burn patients and three trauma patients, and at an average follow-up of forty months, the average arc of ulnohumeral motion was 81 degrees in the burn cohort and 94 degrees in the trauma cohort. Six of the eleven limbs in the burn cohort and five of the nine in the trauma cohort had a good result. The average score according to the American Shoulder and Elbow Surgeons elbow assessment form was 72 points for the burn cohort and 76 points for the trauma cohort. CONCLUSIONS: Osseous ankylosis of the elbow is a severely disabling problem, and attempts to regain mobility are both worthwhile and safe. The results are comparable when the ankylosis is caused by burns or trauma despite the greater complexity of osseous ankylosis in the burned arm. Patients and surgeons should be aware of the small risk of recurrent heterotopic ossification and the moderate risk of pain or recurrent contracture after operative release.
A 61-year-old woman had been coughing up blood-tinged sputum since May 1998. Chest radiography and computed tomography (CT) scans revealed a solitary mass (3 cm in greatest dimension) in the right lower field, accompanied by a surrounding area of ground glass and reticular appearance. Surgical lung biopsy was performed to the surrounding area. The pathological diagnosis was pulmonary ossification of the dendriform type. Alveolar macrophages obtained from her lung differentiated into tartrate-resistant acid phosphatase (TRAP)-positive multinucleated giant cells (MGCs) in the presence of autologous T cells or of macrophage colony stimulating factor (M-CSF) and interleukin-4 (IL-4). This results suggest the possibility that monocytes/macrophages may have the ability to form osteoclasts in the presence of cytokines that may be involved in the development of pulmonary ossification.
It was shown that intersterno-costoclavicular ossification often appears together with pustulosis palmoplantaris by Sonozaki et al., 1980. We report here a patient of Graves' disease associated with intersterno-costoclavicular ossification and pustulosis palmoplantaris, and discuss the relationship with these conditions.
A thirty-three-year-old man with diffuse pulmonary ossification associated with chronic bronchitis is described. Chest roentgenograms of the patient showed diffuse reticulo-nodular shadows partially super-imposed by calcified reticular opacities. Marked declines in forced vital capacity and air-flow limitation were associated with decreased diffusing capacity of the lung for CO, hypoxemia, and hypercapnia. In the postmortem examination, diffuse dendriform ossification was found mainly in the alveolar space. Neither interstitial fibrosis nor capillary congestion was found histopathologically in the lung.
Diffuse idiopathic skeletal hyperostosis (DISH) is a common ossifying diathesis in middle-aged and elderly patients characterized by bone proliferation along the anterior aspect of the spine and at extraspinal sites of ligament and tendon attachment to bone. Four patients with DISH revealed extensive calcification and ossification of the posterior longitudinal ligament in the cervical spine. Review of cervical spine radiographs in 74 additional patients with DISH demonstrated bony hyperostosis of the posterior aspect of the vertebrae in 41%, posterior spinal osteophytosis in 34%, and posterior longitudinal ligament calcification and ossification in 50%. These ligamentous findings, which have previously been described almost exclusively in Japanese people, appear to be an additional skeletal manifestation of DISH.
In cases of paravertebral ossification in humans, the radiographic characteristics usually lead to a specific diagnosis. Similar manifestations are sometimes described in other species. We applied knowledge of the radiographic appearance of spinal alterations in modern humans to the evaluation of 48 fused thoracic and lumbar fossilized spine specimens from the prehistoric saber-toothed cat (Smilodon californicus), in which prominent paravertebral ossification is frequently found. Inspection, conventional radiography, and, in some cases, CT and fluoride analyses were performed. The spinal alterations in the saber-toothed cat, which previously were believed to be caused primarily by trauma, showed characteristics of three major pathologic processes: trauma, diffuse idiopathic skeletal hyperostosis, and inflammatory disease of a type similar to ankylosing spondylitis. The last two categories have rarely been diagnosed in species other than humans. The results suggest that diffuse idiopathic skeletal hyperostosis and inflammatory spondyloarthropathy are diseases that occurred in prehistoric times and are not confined to the human species.
BACKGROUND/AIM: In the prevention of periarticular heterotopic ossification (HO), a common complication after total hip arthroplasty (THA), nonsteroidal antiinflammatory drugs (NSAID) and irradiation are used. Some theories presume that local hypoxia of the soft tissue causes HO. The aim of this study was to investigate if the early use of pulsed electromagnetic fields (PEMF) could prevent this ossification since it accelerates the circulation and oxigenation of soft tissue. METHODS: The study included three groups of the patients with primary THA. The group C consisted of 66 patients/79 hips who had only kinesitherapy in postoperative rehabilitation. The group B consisted of 117 patients/131 hips who had PEMF and interferential current (IC) which, on average, started on the 14th day after the surgery combined with the standard kinesitherapy. The group A consisted of 117 patients/131 hips who had PEMF from the third postoperative day and IC from, on average, the 14th postoperative day with the standard kinesitherapy. The classification of HO was done on a standard AP roentgenograms of the hips, taken at least one year after the surgery. RESULTS: The overall HO was seen in 50.63% of the group C patients, in 43.51% of the B group and in 16.67% of the group A. Severe HO (III and IV class according to Brooker) was seen in 26.58% of the group C patients and in 6.10% of the group B, but none in the group A. CONCLUSION: According to the obtained results an early treatment with PEMF could prevent severe HO and reduce the overall HO.
Seventy patients, representing all the survivors from a group of 117 consecutive Moore arthroplasties, were examined 2 years after the operation. A high incidence of para-articular ossification was observed. The ossification was associated with impaired function of the hip joint. The separation of periost and fascia from the greater trochanter in McFarland's approach may be responsible for the high incidence of bone formation. This theory is compatible with experimental evidence.
A case of para-articular ossifications associated with melorheostosis gave differential diagnostic problems, as the only symptom referred to the ossifications in the left knee region.