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An unusual case of myositis ossificans traumatica lesion located in the paraspinal region is reported. Despite the contiguity of the lesion with the cervical vertebrae and ominous appearance of the biopsy material, the history of antecedent trauma and computed tomography findings allowed preoperative accurate diagnosis. To our knowledge, myositis ossificans traumatica located in the cervical paraspinal region is very rare.
Polymyositis, dermatomyositis, and inclusion body myositis are idiopathic inflammatory myopathies of unknown etiology with autoimmune pathogenesis. For choosing an individual and efficient therapy, diagnostic assignment is an important factor. Therapeutic options in dermatomyositis and polymyositis include corticosteroids and immunosuppressives. Intravenous immunoglobulins are only needed in special cases. In inclusion body myositis, corticosteroids and immunosuppressives are not successful. At the moment intravenous immunoglobulins are the only therapeutic possibility.
BACKGROUND: Cyclic strabismus is a rare disorder in which strabismus and orthotropia regularly alternate over a period of mostly 48 h. It may occur spontaneously, upon squint surgery, or in association with lesions of the central nervous system. In most cases the deviations are convergent. METHODS: Clinical case report. RESULTS: A 34-year-old woman with bilateral recurrent ocular myositis for 2 years had developed cyclic vertical deviation 6 months after clinical remission. A hypotropia of the left eye alternated with an orthotropia, following a 48-h rhythm. Three months after recession of the inferior rectus muscle the alternating squint had disappeared. DISCUSSION: The aetiology of cyclic eye deviations, most of them occurring in a constant rhythm, is not known. The association with lesions of the central nervous system indicates a primary central dysregulation of a "biological clock". Their well-known occurrence, however, after squint surgery and, as in the present case, after orbital myositis, suggests that alteration of peripheral structures may contribute to a central dysregulation. Squint surgery seems to be the treatment of choice, even in rare cases with vertical deviations.
UNLABELLED: Group A streptococcal (GAS) infection is the most common cause of bacterial pharyngitis and has an important role in the pathogenesis of post-infective phenomena including rheumatic fever and glomerulonephritis. Mortality from GAS is uncommon, particularly in the paediatric population. Toxic shock syndrome reflects the most severe form of GAS-related disease and is often associated with fasciitis or myositis. CONCLUSION: We present three cases of toxic shock syndrome secondary to (GAS) myositis demonstrating the importance of early recognition and provision of intensive care management.
A 69-year old farmer developed Aspergillus myositis in the right psoas and paravertebral muscles extending to the retroperitoneum and the fifth lumbar vertebra. The infection appeared after two local instillations of steroid for back pain. Although the patient was not immunocompromised, surgical drainage and antifungal therapy failed to cure him; he died of a bacterial pulmonary superinfection while cultures of the abscess drainage fluid grew Aspergillus. The likely portal of entry in this patient was direct inoculation during infiltration of the steroid; the steroid probably caused a local impairment in host defenses. Only six cases of Aspergillus myositis have been reported previously. All of them occurred in severely immunosuppressed patients and the outcome was fatal in all cases.
This report summarizes clinical features and diagnostic criteria, and the newest advances related to seeking the pathogenic mechanism(s) of sporadic inclusion-body myositis. On the basis of the authors' research, several processes seem to be important in relation to the still-speculative pathogenesis: increased transcription and accumulation of amyloid-b precursor protein and accumulation of its proteolytic fragment amyloid-b; abnormal accumulation of components related to lipid metabolism (eg, low-density lipoprotein receptors and cholesterol; accumulation of cholesterol is possibly caused by its abnormal trafficking); oxidative stress; accumulations of other Alzheimer-related proteins including phosphorylated tau; a milieu of muscle cellular aging in which these changes occur. The authors' basic hypothesis is that overexpression of amyloid-b precursor protein within the aging muscle fibers is an early upstream event causing the subsequent pathogenic cascade. The remarkable pathologic similarities between inclusion-body myositis muscle and Alzheimer's disease brain are discussed.
Myositis ossificans progressiva is a rare autosomal dominant disorder characterized by a progressive ectopic ossification of the connective tissue of the voluntary muscles and ligaments with associated debilitation and characteristic skeletal malformations. There have been less than 600 cases in the literature. This is the first reported case of a viable pregnancy in a patient with myositis ossificans progressiva.
Myositis is an autoimmune inflammatory muscle disease of unknown etiology. We demonstrate directly that the antigen to the myositis-specific anti-Jo-1 antibody is histidyl-tRNA synthetase. The anti-Jo-1 antibody inhibits human HeLa and rat liver histidyl-tRNA synthetase. Using conventional and immunoaffinity chromatography with immobilized anti-Jo-1 antibody, we have purified rat liver histidyl-tRNA synthetase which has a subunit Mr 64,000 and an estimated native Mr suggesting an alpha 2 structure. The evidence indicates that the Jo-1 antigen is histidyl-tRNA synthetase, and that some of the histidyl-tRNA synthetase structure are conserved across species.
Rimmed vacuoles are small areas of focal destruction of muscle fibres, found in inclusion body myositis, oculopharyngeal muscular dystrophy and other muscle disorders. They are known to contain amyloid proteins, probably of beta-amyloid type. We examined rimmed vacuoles immunohistochemically in 12 patients with inclusion body myositis and two patients with oculopharyngeal muscular dystrophy with antibodies to beta-amyloid precursor protein and cathepsin B and D. We found evidence for the presence of all these markers in rimmed vacuoles. These results confirm the presence of beta-amyloid in rimmed vacuoles, and provide additional support for the hypotheses that rimmed vacuoles are of lysosomal origin and that lysosomes are probably important in the metabolism of amyloid precursor protein.
A case of proliferative myositis arising in the tongue is described. Light and electron micrographs revealed the characteristic infiltrative growth pattern and cellular pleomorphism of this lesion. A review of the literature disclosed 50 reported cases of proliferative myositis, including 10 that originated in the head and neck. The rapid growth rate and unusual gross and histologic appearance of this infiltrative lesion have contributed to its relatively frequent misdiagnosis and inappropriate treatment. Consequently it is hoped that this report will help clarify its benign nature.
Inclusion body myositis (IBM) is a distinct type of muscle disease. The characteristic electron microscopic findings, intranuclear or intracytoplasmic inclusions composed of microtubular filaments, morphologically resemble paramyxovirus nucleocapsids. These findings and the reported immunoreactivity of the inclusions with mumps virus antibodies have suggested that inclusion body myositis is a chronic virus infection. We analyzed skeletal muscle specimens from three patients with characteristic light microscopic features and electron microscopically verified inclusions of IBM by immunocytochemistry using antibodies raised against members of the paramyxovirus group, and by in situ hybridization with a cRNA probe representing the mumps virus nucleocapsid gene. The specificity of the reactions was demonstrated with infected and uninfected cultured cells. No immunocytochemical staining or hybridization signal was observed in biopsy specimens from IBM patients. These findings speak against a paramyxovirus etiology of IBM.
Proliferative myositis (PM) is a head and neck mass of rare etiology. In a review of the literature, 57 cases of PM were found. Of these, only 12 occurred in the head and neck region. Proliferative myositis is most rare in children, with only three reported cases, none of which involved the head and neck region. A case of a 9-year-old girl with PM of the mylohyoid muscle, presenting as a neck mass, is reported.
Traumatic myositis ossificans in the left masseter muscle of a 25-year-old man is described. The lesion, which extended intramuscularly from the lateral surface of the zygomatic bone to the mandibular angle, was resected by an intraoral approach. The specimen was composed of mature cortical bone in the outer portion and spongy bone, including degenerated muscle fibers and marrow spaces, in the inner portion. At the lower end of the specimen was a sequence of bone formation involving fibrous connective tissue, cartilage, woven bone, and mature lamellar bone with many osteoblasts. Clinicopathologic features of 26 reported cases of traumatic myositis ossificans of the maxillofacial region are reviewed, and the pathogenesis of the disease is discussed.
Infantile myositis, observed in the neonatal period, is rare and may be confused with congenital muscular dystrophy. The patient presented here showed evidence of a myopathy with in utero onset with intrauterine growth retardation and decreased fetal movements. A muscle biopsy demonstrated characteristic perifascicular atrophy and inflammatory infiltrates. Initial magnetic resonance (MR) examination revealed high signal intensity in several proximal muscle groups. With subsequent corticosteroid treatment, the patient's symptoms, clinical course and MR findings have gradually improved. The diagnosis of infantile myositis should be established as early as possible, since proper treatment can lead to resolution of clinical symptomatology and MR findings.
Myositis ossificans circumscripta (or pseudomalignant osseous tumor of soft tissue) may be confused with malignant neoplasms if there is no clear history of trauma. Two cases are presented in which magnetic resonance imaging (MRI) clearly demonstrates disproportionally extensive abnormal signal intensity along the course of muscle fibers. Although the specificity of this finding is uncertain, MRI enhances the level of confidence in diagnosing myositis ossificans.
Myositis ossificans progressiva is a rare disease leading to complete ossification of the muscular system. Very little information about this rare disease and its anesthetic implications has appeared in anesthetic literature. This disease is felt to have an autosomal dominant pattern of inheritance and is usually associated with anomalies of the hands and feet. Afflicted patients are frequently misdiagnosed in childhood as having a rheumatologic disorder. Later in life (as true bone is formed in striated muscle, ligaments, and fascia), the correct diagnosis becomes obvious. Although muscles of the heart, diaphragm, larynx, and sphincters are spared, those of the chest wall are not, and pulmonary function progressively deteriorates. Death frequently occurs as a result of a pulmonary infection. Specific anesthetic considerations include positioning to avoid injury, potential need for fiberoptic intubation or tracheostomy, decreased thoracic compliance with the need for increased ventilating pressures, and the ultimate in disuse atrophy contraindicating the use of succinylcholine. Myositis ossificans progressiva can present the anesthesiologist with interesting challenges. Anesthetic management will need to be individualized according to the severity of the disease.
Streptococcus dysgalactiae subspecies equisimilis (SDSE) is an emerging human pathogen closely related to group A Streptococcus. However, its genetic requirements for survival and growth in different conditions and for causing invasive infections remain poorly understood. To address this gap, transposon-directed insertion-site sequencing was used to identify genes contributing to fitness in experimental necrotizing myositis in nonhuman primates. Using two SDSE stG62647 human clinical isolates, MGCS36044 and MGCS36089, highly saturated transposon mutant libraries were generated and analyzed following in vitro growth and in vivo infection in eight nonhuman primates. A total of 398 essential genes were identified to be shared by both strains during growth in vitro and in vivo, and 17 and 7 conditionally essential genes required only in vitro or only in vivo, respectively. Additionally, 117 and 110 genes in MGCS36044 and MGCS36089, respectively, were found to be associated with fitness during necrotizing myositis. Transposon insertions in 34 MGCS36044 genes conferred increased fitness, whereas mutation of 83 genes conferred decreased fitness. Similarly, in MGCS36089, mutations in 38 and 72 genes conferred increased or decreased fitness, respectively. Importantly, both strains shared 46 fitness-associated genes, including an enrichment of transporter genes, highlighting nutrient acquisition as a dominant requirement during infection. The results provide critical information for guiding future translational efforts to develop preventive and therapeutic strategies against human SDSE infections.
CASE REPORT: A 5-year-old girl was hospitalised for fever, abdominal and lumbar pain, associated with general impairment state and a whitlow. One of the blood cultures and CSF grew A beta haemolytic Streptococcus, muscular echography and MRI showed paravertebral myositis, which was complicated by an epidural abscess. The outcome was good with medical treatment alone. DISCUSSION: Streptococcal myositis is a rare and severe skeletal muscle infection caused by A beta haemolytic Streptococcus. It is characterized by a muscle necrosis, without abscess formation. It has to be distinguished from pyomyositis, usually caused by Staphylococcus aureus, where a muscle abscess occurs, which must be treated by surgical drainage and antibiotics. Prognosis of this infection is poorer than other muscle infections such as pyomyositis, with a high mortality rate. The diagnosis is difficult and often delayed. Practitioners should keep in mind this diagnosis, even if symptoms are non specific, in front of an undetermined infectious syndrome associated with pain, and make an echography or nuclear magnetic resonance imaging to confirm the diagnosis.