Uveitis, an under-recognized adverse effect of pamidronate. Case report and literature review.

Ophthalmologic adverse effects of bisphosphonate therapy are infrequent and of unclear pathogenesis. The most common has been anterior uveitis, of which 18 cases have been reported. Onset was within 24 to 48 hours after infusion initiation, and both eyes were affected in most patients. The outcome was favorable within a few days after bisphosphonate discontinuation and topical glucocorticoid therapy, although rechallenge was frequently followed by a recurrence. Bisphosphonates are being used successfully in an increasingly broad range of disorders. We report a case of pamidronate-induced anterior uveitis and present a review of the relevant literature.

Fibrous dysplasia with intramuscular myxoma (Mazabraud's syndrome). Report of a case and review of the literature.

About 30 cases of fibrous dysplasia associated with one or more myxomas (Mazabraud's syndrome) have been reported since 1926. We report a new case in a woman with polyostotic fibrous dysplasia and a myxoma in the left femoral muscle. She also had a history of precocious sexual development and café au lait spots, two manifestations whose association with polyostotic fibrous dysplasia defines McCune-Albright syndrome.

Anteroposterior atlantoaxial subluxation in cervical spine osteoarthritis: case reports and review of the literature.

Nontraumatic anteroposterior atlantoaxial subluxation (AAS) has been described in several rheumatic or inherited disorders, especially rheumatoid arthritis and to a lesser extent the inflammatory spondyloarthropathies. We describe AAS secondary to osteoarthritis (OA) of the cervical spine in a 76-year-old man and a 73-year-old woman with severe cervical OA, symptomatic C1-C2 facet joints, and signs of generalized OA. Only 6 similar cases exist in the literature. OA should be added to the causes of AAS, and conversely AAS should be assessed in cases with severe OA of the upper cervical spine.

The intravertebral vacuum phenomenon ("vertebral osteonecrosis"). Migration of intradiscal gas in a fractured vertebral body?

STUDY DESIGN: Retrospective cohort. OBJECTIVES: To compare the prevalence of the association between contiguous intervertebral disc and vertebral collapses with or without an intravertebral vacuum phenomenon. SUMMARY OF BACKGROUND DATA: The mechanism of occasional gas accumulation within some vertebral collapses is poorly known. The current hypothesis is that this phenomenon is indicative of bone ischemia. In fact, avascular necrosis as the main pathologic event remains speculative, and should not explain per se the presence of gas within a vertebral body. METHODS: Comparison of the prevalence of intervertebral disc vacuum phenomenon adjacent to the affected vertebral body in 23 cases of intravertebral vacuum phenomenon in 19 patients (intravertebral vacuum phenomenon group) and in 708 osteoporotic collapses without intravertebral vacuum phenomenon in 199 patients (control group). RESULTS: There were no differences in sex and age between the two groups, and all the patients in the intravertebral vacuum phenomenon group had signs of underlying osteoporosis. A vacuum phenomenon in at least one intervertebral disc adjacent to the collapses on radiographs, conventional tomography, computed tomography, or magnetic resonance imaging was found in 19 cases (83%) in the intravertebral vacuum phenomenon group, compared with 13% in the control group (P < 0.0001). Considering plain radiographs only, this association was found in 50% of the intravertebral vacuum phenomenon group and in 9.7% of the control group (P < 0.0001). The intervertebral and intravertebral gaseous collections were connected through a fractured endplate in six cases. CONCLUSIONS: The high prevalence of the association of contiguous intervertebral and intravertebral vacuum phenomenon could have implications in the pathogenesis of the intravertebral vacuum phenomenon. We hypothesize that the intravertebral vacuum phenomenon could simply be the result of migration of an intradiscal-gaseous collection through the fractured endplate of some osteoporotic collapses.

Bone mineral density in patients given oral vitamin K antagonists.

BACKGROUND: Divergent results have been obtained in studies of bone mineral density in patients under oral vitamin K antagonists. OBJECTIVE: To gather prospective data on bone mineral density and bone metabolism in 70 aortic valve replacement patients. STUDY DESIGN: 49 patients who had been under oral vitamin K antagonists for at least one year after implantation of a mechanical aortic valve were compared with 21 recipients of a tissue aortic valve that did not require anticoagulant therapy. The following investigations were done in all patients: (1) dual-energy X-ray absorptiometry measurement of bone mineral density at the lumbar spine and femoral neck; (2) roentgenograms of the spine and pelvis; (3) serum assays of calcium, phosphate, creatinine, alkaline phosphatase, osteocalcin, 25-OH-vitamin D3, and parathyroid hormone. RESULTS: The two groups were comparable regarding age and sex ratio. No differences were found in lumbar or femoral neck bone mineral density even after adjustment for age and sex. A trend toward an increase in bone mineral density at both sites with increasing duration of vitamin K antagonist therapy was demonstrated. The only bone turnover marker difference between the two groups was a significantly lower serum osteocalcin level in the group under vitamin K antagonist therapy (P < 0.0001). CONCLUSIONS: Long-term vitamin K antagonist therapy does not affect bone mineral density at the lumbar spine or femoral neck and also fails to modify bone turnover markers, with the exception of osteocalcin.

Bone insufficiency fractures as an inaugural manifestation of primary hyperparathyroidism.

Primary hyperparathyroidism causes excessive bone resorption with a decrease in bone mineral density. Fractures of the vertebras and appendicular bones, however, seem uncommon, even in the long term. We report three patients who presented with bone insufficiency fractures as the inaugural symptom of primary hyperparathyroidism. The three patients were women, aged 62, 65 and 86 years, respectively, who presented with fractures of the medial tibial plateau, femoral neck of femoral neck and tarsus. Laboratory tests showed hypercalcemia, hypophosphatemia and elevated parathyroid hormone levels. Apart from confusion in the 86-year-old patient, there were no clinical manifestations. A bone biopsy obtained in one patient showed increased resorption parameters with no loss of bone trabecular volume; the two other patients underwent absorptiometry, which disclosed a marked decrease in bone mineral density at the spine and femoral neck. There were no risk factors for osteopenia apart from advanced age and female gender. A parathyroid adenoma was removed surgically in all three cases. Vitamin D deficiency was a concomitant abnormality that probably exacerbated the adverse effects of hyperparathyroidism on the skeleton.

Insufficiency fractures of the medial femoral condyle.

We report six cases of insufficiency fractures of the medial femoral condyle responsible for severe mechanical pain in the medial knee compartment in the absence of any identifiable precipitating factor. Suggestive changes were seen on initial roentgenograms in only one case, whereas increased radionuclide uptake was a consistent finding on the bone scan. The diagnosis was established only by magnetic resonance imaging in five cases. All six patients were women and four were older than 75 years. Two patients had a history of osteoporotic fractures, one had laboratory test evidence of osteomalacia and one had recurrent insufficiency fractures mistakenly ascribed for several years to migratory transient osteoporosis. Bone density was subnormal in five of the six patients. Rest and standard analgesics consistently ensured a favorable outcome within three to four weeks. To our knowledge there have been no previous reports of stress fractures of the medial femoral condyle. These lesions may be underdiagnosed since they are easily mistaken for primary osteonecrosis in the absence of magnetic resonance imaging. Primary osteonecrosis of the femoral condyle shares several features with insufficiency fractures, including predominance in elderly women with factors responsible for mechanical stress (varum, obesity, trivial trauma), mechanical pain, and increased radionuclide uptake. Because some cases of primary osteonecrosis may be secondary to undiagnosed stress-related microfractures, early diagnosis and elimination of weight bearing are essential.

[Post-traumatic psoriatic arthritis. 2 cases]. / Rhumatisme psoriasique post-traumatique. Deux observations.

Physical trauma is generally accepted as a possible factor in the pathogenesis of rheumatoid arthritis. In the last ten years, there have been a few rare case reports of physical trauma precipitating psoriasic arthritis. We observed two such cases following an occupational accident discovered one and a half year and two and a half year after onset of the first clinical manifestations. In the first case, a 43-year-old man had a fracture of the right calcaneus in March 1991. He was treated with nailing and also required emergency surgery of the posterior tibial artery. The tibiotarsal joint was normal radiologically. Pain persisted after treatment and in 1993 he presented with psoriasis of the scalp and several other localizations together with Hallopeau's acrodermatitis continua of the ankle, pathognomonic for psoriasic arthritis. Salazosufapyridin was given. The second case was a 50-year-old man who had major pain in both wrists immediately after falling on the palm of his hands in 1992. Bilateral carpal tunnel syndrome developed which did not respond well to surgery. In 1993, he developed inflammatory synovitis and also had psoriasis mainly located at the elbows. Immunological tests were negative. Cortisone and salazosulfapyridin were not particularly effective and the patient later developed arthritis of the hip and ankle joints. Physicians should be aware of physical trauma as a causative factor in psoriasic arthritis due to the potential legal implications. Criteria for imputability are: single major physical trauma, absence of clinical signs prior to the trauma, continuous clinical course, first signs occurring then predominating at the joint exposed to trauma. The pathophysiology of this type of arthritis is not well understood. Deep Koebner's phenomena could be involved. Activation of substance P has also been hypothesized.

Lack of correlation between pharmacokinetics and efficacy of low dose methotrexate in patients with rheumatoid arthritis.

OBJECTIVE: To determine if the variability in the efficacy of methotrexate (MTX) in rheumatoid arthritis (RA) is correlated with its pharmacokinetics. METHODS: MTX pharmacokinetics was evaluated in 46 patients with RA starting a weekly intramuscular low dose MTX treatment. The patients were divided into 32 responders and 14 nonresponders to MTX according to the clinical response in the 6 months after the pharmacokinetic study. MTX plasma (at T0, 0.25, 0.5, 0.75, 1, 2, 4, 6, 8, 12 and 24 h) and urine concentrations were measured with the TDx Abbott fluorescence polarization immunoassay. RESULTS: The MTX dosage, age, sex, RA duration, hepatic and renal functions of responders and nonresponders were not different. No difference was found either in peak concentration, residual 24th hour concentration, area under the curve, total body clearance, renal clearance, and terminal T1/2 life of MTX in responders and nonresponders. Surprisingly, patients with adverse reactions had higher total body and renal MTX clearances than those without side effects during the study. CONCLUSION: These data suggest that plasma MTX measurements are not helpful in defining an optimal treatment regimen.

Erosive polyarthritis and Crohn's disease. A case with HLA DRB1 determination.

The authors report the case of a 49-year-old woman with an asymmetrical, seronegative and peripheral polyarthritis with erosions, who subsequently developed Crohn's disease. She was diagnosed as having an erosive Crohn polyarthritis as no evidence of rheumatoid arthritis was found. However, the patient was homozygote for DR4 and the HLA DRB1 oligotyping was 0401/0404. This corresponds to two susceptibility alleles for rheumatoid arthritis responsible for the severity of this disease. Erosive polyarthritis is rarely encountered in inflammatory bowel diseases. The underlying mechanism of the erosions in these conditions is unknown but granulomatous synovitis with contiguous erosive bone changes has been reported. HLA DR has not been previously reported in erosive Crohn polyarthritis. The homozygosity for DR4 with DRB1*0401/0404 subtypes suggests that DR4 could contribute to the erosive course in this patient. This question is of interest, and HLA DR must be further studied in inflammatory bowel diseases with erosive arthritic manifestations.

Intervertebral disk vacuum phenomenon secondary to vertebral collapse: prevalence and significance.

PURPOSE: To evaluate the prevalence of the intervertebral vacuum phenomenon in disks adjacent to vertebral collapses of various causes. MATERIALS AND METHODS: The authors retrospectively studied 310 consecutive patients who had at least one vertebral collapse. The vacuum phenomena were divided into collapse-related (ie, adjacent to a collapse) and degenerative (ie, not associated with a vertebral fracture) ones. Plain radiography was performed in all patients, and, in addition, conventional tomography, computed tomography, and/or magnetic resonance imaging was performed in 175 patients. RESULTS: Collapse-related vacuum phenomena were seen in 15% of patients with plain radiography and in 21% when all of the imaging modalities were considered. They occurred in association with all of the causes of collapse (ie, osteoporosis, multiple myeloma, metastasis, acute trauma, and vertebral osteomyelitis), although the prevalence was lower in association with metastasis or trauma (P = .008). The occurrence of collapse-related vacuum phenomenon was correlated with age (P = .001) and with the coexistence of degenerative disk-related vacuum phenomenon in the same patient (P = .0002). CONCLUSION: Occurrence of a vacuum cleft in disks adjacent to a vertebral collapse is common and appears as the second cause of vacuum phenomenon after degenerative disk disease.

An unexpected, benign cause of increased muscular uptake at bone scintigraphy.

Bone scintigraphy was performed in a healthy 30-year-old man because of post-traumatic heel pain. The scintigraphy displayed a bilateral increased uptake of 99mTc-MDP in the pectoral muscles and triceps brachii. Further extensive investigations including muscle biopsy were normal and excluded diagnoses such as polymyositis, myositis ossificans or amyloidosis. The abnormalities were in fact explained by physical exercise performed a few hours prior to the radionuclide scan. This was confirmed by the absence of these changes on a second bone scan performed 3 months later. This case shows that muscle exercise should be kept in mind as a cause of skeletal muscle uptake of bone-seeking agents, with the view to avoid unnecessary investigations.

[Severe laryngeal involvement in rheumatoid arthritis requiring permanent tracheostomy]. / Atteinte laryngée sévère au cours d'une polyarthrite rhumatoïde nécessitant une trachéotomie définitive.

A 72-year-old woman with severe rheumatoid arthritis developed atlantoaxial subluxation requiring surgery. Tracheal intubation was performed during the procedure. Gradually worsening dysphonia and dyspnea developed postoperatively. Laryngoscopy showed palsy of both vocal cords and coalescence of the arytenoid cartilages. Despite surgical treatment, permanent tracheostomy was required. There is evidence that laryngeal involvement is common but usually subclinical or mild in rheumatoid arthritis patients. Tracheal intubation can cause the laryngeal lesions to flare. Clinical manifestations are nonspecific and sometimes misleading (e.g., cough or pharyngeal and laryngeal pain). Direct laryngoscopy and computed tomography of the larynx allow evaluation of the lesions of the different components of the larynx. The case reported herein demonstrates that rheumatoid arthritis patients should be screened for laryngeal involvement by history and ENT examination and that any laryngeal abnormalities should be reported to the anesthesiologist if tracheal intubation is planned.

Coexisting kappa light chain multiple myeloma and primary hyperparathyroidism.

The authors report the case of an 82 year old woman hospitalized for hypercalcemia associated with low serum phosphate. Multiple myeloma was first diagnosed. However, despite chemotherapy, hypercalcemia persisted and she was subsequently diagnosed as primary hyperparathyroidism; eucalcemic state was then obtained after parathyroidectomy. Fifteen similar cases are reported in the literature and the mechanisms and implications of such an association are discussed.