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PubMed1980

Juvenile myasthenia gravis.

We studied 32 children with myasthenia gravis over a period of 12 years. The mean age at onset was 7.7 years. Presentation was ocular in 63% of patients. Another major disease in addition to myasthenia occurred in 44% of patients; a seizure disorder was the most commonly associated disease. Serum IgG antibody to nicotinic acetylcholine receptor was present in 53% of patients and did not correlate with severity of disease or treatment. Medical management was effective in 63%; thymectomy was effective in only 28%. We conclude that myasthenia gravis appears commonly before age 10 and is associated with the risk of some disease other than hyperthyroidism. Serum IgG nicotinic acetylcholine receptor antibody is present less frequently than in normal adults, and vigorous medical management should be attempted before thymectomy.

Adolescent↗

PubMed1981

Myasthenia gravis in infancy.

Symptoms of myasthenia gravis in infancy may occur from passive transfer of maternal disease, acquired autoimmune disease, or nonautoimmune hereditary disease. Of nine patients with infantile-onset myasthenia who were not born to mothers with the disease, two had detectable antibody to acetylcholine receptor. Patients with or without antibodies were clinically indistinguishable, except for the occurrence of similar disease in siblings of patients without antibody. Differentiation of autoimmune and hereditary, myasthenia in infancy is necessary for appropriate therapeutic measures and genetic counseling. Antibody determinations provide a useful aid in this differentiation.

Antibodies↗

PubMed1986

Familial autoimmune myasthenia gravis and thymoma: occurrence in two brothers.

At ages 31 and 42 years, two brothers presented with clinical, pharmacologic, electrophysiologic, and immunologic characteristics of autoimmune myasthenia gravis. At thymectomy, both had histologic findings of epithelial thymoma. HLA analysis revealed A2, A3, B7, and B39 antigens in one patient and A3, A24, B7, and B40 antigens in the other. Familial myasthenia gravis with thymoma has not been described previously. Familial thymoma has been rarely reported, but never with myasthenia gravis.

Adult↗

PubMed1991

Repeat thymectomy in chronic refractory myasthenia gravis.

Encouraged by recent results with "extended" thymectomy in the treatment of myasthenia gravis, we carried out repeat thymectomy in six patients with chronic, refractory disease who did not initially have extended thymectomy. All were completely disabled with longstanding myasthenia. Initial thymectomy (four transsternal, one transcervical, and one substernal) was carried out at a mean of 8.9 years previously (range, 5 to 18). There was no residual thymus observed with CT, but at repeat thymectomy, residual thymic tissue was present in five of six patients. Five patients significantly improved and four returned to full-time work. Mean prednisone dose declined from 51 mg to 18 mg/d, and mean pyridostigmine dose fell from 1,290 mg to 415 mg/d. No patient to date has had a complete remission. These results suggest that repeat thymectomy may benefit some patients with chronic disabling myasthenia gravis, especially when it is uncertain from a review of the operative report whether all thymic tissue was removed at the initial thymectomy.

Adult↗

PubMed1992

Levels of CD5+ B lymphocytes do not differ between patients with myasthenia gravis and healthy individuals.

CD5+ B cells might be involved in autoimmunity mainly as autoantibody-producing cells. To investigate the possible role of these cells in myasthenia gravis, we studied the numbers of CD5+ B cells, CD5- B cells, and CD19+ B cells as well as CD5+ T cells in the peripheral blood from 31 patients with myasthenia gravis and 31 healthy individuals. Both absolute percentages (percent of peripheral blood mononuclear cells) and relative percentages (percent of total CD19+ B cells) of CD5+ B cells were the same in patients as in controls. The numbers of CD5- B cells and CD19+ B cells were the same in both groups, whereas CD5+ T cells were lower in the patients. There was no correlation between clinical stage, sex, thymectomy, or pathology of thymus and the levels of CD5+ B cells, CD5- B cells, or CD19+ B cells. Patients treated with azathioprine had lower levels of CD5+ B cells than untreated patients and controls. Our results show that patients with myasthenia gravis have the same levels of CD5+ B cells as healthy individuals.

Adult↗

PubMedDate not supplied

Anesthetic considerations for myasthenia gravis and pregnancy.

Three pregnant patients with myasthenia gravis are presented and anesthetic considerations discussed. The course of myasthenia gravis is highly variable and unpredictable during pregnancy. Anticholinesterase therapy should be maintained during labor, and the IM route of administration is preferred. Exacerbations very often occur in the immediate postpartum period. Regional anesthesia is preferred for vaginal delivery. In the case of cesarean section, general anesthesia may be preferable. Neonatal myasthenia gravis is a transient syndrome that appears in 20 to 30% of the newborns of myasthenic mothers.

Adult↗

PubMed2005

A novel immunomodulator, FTY720, prevents development of experimental autoimmune myasthenia gravis in C57BL/6 mice.

Prophylactic oral administration of a novel immunomodulator (immunosuppressant), FTY720 (1 mg/kg, three times a week), completely prevented the development of experimental autoimmune myasthenia gravis (EAMG) in C57BL/6 mice. EAMG has been used as an animal model for human myasthenia gravis, and was established by immunizing the mice with acetylcholine receptor (AChR) from Torpedo californica. FTY720 also suppressed the production of both anti-Torpedo californica AChR antibody and anti-mouse AChR autoantibody by the mice, which were observed in mice in which EAMG had become established. These results strongly suggest that FTY720 is a promising candidate for treatment of human myasthenia gravis.

Animals↗

PubMed2005

Video-assisted thoracic surgery thymectomy for nonthymomatous myasthenia gravis.

STUDY OBJECTIVES: Minimal-access thymectomy has become increasingly popular as surgical treatment for patients with nonthymomatous myasthenia gravis (NTMG) because of its comparable efficacy, safety, and lesser degree of tissue trauma compared with conventional open surgery. We reviewed and analyzed our data on video-assisted thoracic surgery (VATS) thymectomy and present the clinical outcomes according to the Myasthenia Gravis Foundation of America classification. DESIGN: A retrospective review of VATS thymectomy for NTMG in a university hospital over a 12-year period. Data were collected from the medical records and supplemented with telephone surveys. The impact of surgery and other variables potentially affecting complete stable remission (CSR) were calculated using Kaplan-Meier survival curves; comparisons between survival curves was performed using the log-rank test. RESULTS: A total of 38 consecutive patients underwent VATS thymectomy for NTMG. Median postoperative stay was 3 days. Pathologic examination revealed thymic hyperplasia in 61.1% of cases, normal thymus in 22.2%, and thymic atrophy in 16.6%. There was no perioperative mortality; complications occurred in four patients. After a median follow-up of 69 months, 91.6% of patients experienced improvement, with crude CSR achieved in 22.2%. Kaplan-Meier survival curve demonstrated a 75% CSR rate at 10-year follow-up. On univariate analysis, only disease duration < or = 12 months (p = 0.03) was associated with a statistically significant improvement in CSR. CONCLUSIONS: VATS thymectomy for NTMG results in symptomatic improvement in the vast majority of patients, with a high rate of CSR. The procedure is associated with low morbidity and no perioperative mortality. Future studies on thymectomy for myasthenia gravis should be reported in a standardized manner to allow accurate comparisons between results in the absence of randomized prospective trials.

Adult↗

PubMed1984

Postoperative respiratory care after transsternal thymectomy in myasthenia gravis. A 3-year experience in 53 patients.

During a three-year period, 53 patients with myasthenia gravis underwent transsternal thymectomy by a partial sternum-splitting technique at our institution. This procedure was carried out (1) because an x-ray film or a computed tomography scan had suggested the presence of a thymoma or (2) to manage symptoms of myasthenia gravis. In 41 patients the endotracheal tube was removed in the post-anesthesia recovery room. The remaining 12 patients were extubated in the Respiratory Care Unit--in five, after prolonged mechanical ventilation. From our experience, the only factor useful for predicting the probable need for prolonged postoperative mechanical ventilation is the degree of bulbar involvement. Patients in Osserman classification groups 3 and 4 have an extremely high incidence of postoperative respiratory failure. Consideration should be given to the use of preoperative plasmapheresis in myasthenia gravis patients who have significant bulbar symptoms.

Adolescent↗

PubMed1982

Development of the gonadotrophic resistant ovary syndrome in myasthenia gravis: suggestion of similar autoimmune mechanisms.

A woman with myasthenia gravis who developed hypergonadotrophic amenorrhoea was studied. This patient fulfilled all accepted criteria for the diagnosis of the gonadotrophin resistant ovary syndrome: high levels of serum LH and FSH by radioimmunoassay and urinary gonadotrophin excretion by bioassay, low serum oestradiol, lack of response to exogenous gonadotrophin and ovaries with multiple non-stimulated primordial follicles. The serum of this patient contained a substance which behaving like a gamma globulin, inhibited FSH specific binding to receptors in an in vitro system. This inhibitor was looked for in further patients with hypergonadotrophic amenorrhoea, other immunological diseases or myasthenia gravis with normal menses. Ovarian biopsy was not available in these patients. In all except one who also had the association of myasthenia gravis and hypergonadotrophic amenorrhoea, the inhibitor was not detected. It is suggested that development of antibodies directed against gonadotrophin receptors or a receptor related membrane domain may play a role in the mechanism of failure of gonadotrophin stimulation in some patients with the gonadotrophin resistant ovary syndrome.

Adolescent↗

PubMed1976

Familial myasthenia gravis. Report of four cases.

Two pairs of siblings with myasthenia gravis, belonging to two different families, are reported. This is the only record of familial myasthenia during the past twenty years, in a total of 145 patients seen at the Neurological Clinic of the São Paulo Medical School. In spite of the fact that myasthenia gravis does not show hereditary characteristics, the peculiar features of the four cases justify the present report. The two pairs of siblings were born from non myasthenic nor consanguineous parents. The disease started at birth showing bilateral partial eyelid ptosis in all patients. The course of the illness has been favorable. There was no thymoma.

Adult↗

PubMed1990

[An unusual association: polymyositis, myasthenia gravis and thymoma].

Based on the inexistence of references in Brazilian and International literature of the association polymyositis, myasthenia gravis and thymoma in a young male adult, the authors propose to describe one case presenting the associated syndrome. The disease began when the patient was 22 years old, being the climax of its clinical expression four years later. It is emphasized the difficulty on the differential diagnosis between polymyositis and myasthenia gravis, once these diseases frequently have their clinical manifestations well individualized. In this study we call attention to the existence of clinical aspects of myasthenia gravis in patients presenting polymyositis and, on the other hand, histopathological alterations typical of polymyositis in myasthenic muscles, what makes the clinical diagnosis uncertain many times.

Adult↗

PubMed1993

[Scleroderma, thyroiditis, and myasthenia gravis: study of a case].

A thirty-six years old woman with myasthenia gravis developed cold intolerance after two years, and Hashimoto's thyroiditis diagnosis was established. Four years later she exhibited skin thickening (limited scleroderma). Laboratory and clinical findings suggested the CREST type of systemic sclerosis. Difficulty in scleroderma diagnosis is discussed, as overlap of syndromes. Hashimoto's thyroiditis and myasthenia gravis are infrequently associated. Occurrence of scleroderma and myasthenia gravis in the same patient is rare. The coincidence of these three disorders was not found in literature.

Adult↗

PubMed1999

Myasthenia gravis and thymoma. Evaluation of 41 patients.

We evaluated the epidemiological, clinical, laboratory and therapeutical aspects of 41 patients with thymomatous myasthenia gravis. Thirty five patients (85.36%) were submitted to thymectomy. Follow-up ranged from two to 18 years. Diagnosis of thymoma was based upon clinical investigations and CT scan of the anterior mediastinum and in 11 patients supported by immunological tests of anti-striated muscle antibodies with a positive result in more than 80% of cases. Histopathologic examination of all thymomectomized patients confirmed the diagnosis of thymoma. There was a significant predominance of benign over malignant thymoma. Occurred higher prevalence of male patients and of patients over 40 years of age. The therapeutical strategy to control myasthenic clinical findings was the same as that for non-thymomatous myasthenia gravis. The corticosteroids associated to cytotoxic drugs were less often used. Radiotherapy of the anterior mediastinum was more often used in patients having invasive tumors submitted to surgery or not. With regard to survival and control of myasthenia gravis, especially in younger patients and in those submitted to early surgery, results of treatment were surprisingly favorable.

Adult↗

PubMed2005

The role of the thymus in the pathogenesis of myasthenia gravis.

Myasthenia gravis is an organ-specific autoimmune disease characterized by the production of anti-acetylcholine receptor antibodies. In this review, I describe the pathophysiological importance of the altered chemokine receptor-mediated signaling in the thymus and peripheral blood of myasthenia gravis patients. The epidemiological and clinical features of myasthenia gravis are also discussed.

Animals↗

PubMed1983

Myasthenia gravis. Pathogenesis and current concepts in management.

Myasthenia gravis is a disorder of autoimmunity in which neuromuscular transmission is impaired by autoantibodies to the acetylcholine receptor (AChR). There is evidence for more than one form of the disorder with differing genetic susceptibilities. The aetiology is unknown, but thymic involvement is suggested by abnormal histology and by the beneficial response of the disorder to thymectomy in more than two-thirds of patients. Thymectomy is indicated in most patients unless the symptoms are minimal or are confined to the extraocular muscles alone, or the patient is elderly. Thymectomy alone results in remission in about one-third of patients, but, in addition, most patients require symptomatic anticholinesterase drugs to prolong the action of acetylcholine at the muscle end-plate. Over-dosage of these drugs can also cause weakness. Immunosuppression with corticosteroids or azathioprine may also improve myasthenia; at present, these drugs are used mainly in patients who do not respond to thymectomy or in those patients considered unsuitable for operation. Plasma exchange can cause a rapid, though temporary, involvement in myasthenia, but it probably has no long term place in its treatment. Future therapy will probably involve specific immunotherapy, such as anti-idiotype antibodies.

Cholinesterase Inhibitors↗

PubMed2001

Myasthenia gravis: options and timing of immunomodulatory treatment.

The autoimmune pathogenesis of myasthenia gravis is relatively well understood. The current options for treatment of this disease are acute and long term immunotherapies, acetylcholinesterase inhibitors and thymectomy. Many factors influence the timing of initiation of immunomodulatory therapy in myasthenia gravis and both disease factors, such as stage and severity, and patient factors, such as age, pregnancy and intercurrent illness, must be considered. Decisions regarding the choice of therapy can be difficult because of the limited number of randomised controlled trials that have been performed in myasthenic patients. In general, acetylcholinesterase inhibitors alone are used only in mild ocular disease, and in the majority of other patients immunomodulatory therapy is begun early. Corticosteroids are the most commonly used initial therapy, followed by azathioprine. In refractory cases, the available options include immunosuppressants such as cyclosporin, mycophenolate mofetil and cyclophosphamide. Plasmapheresis and intravenous immunoglobulin are important in the treatment of acute exacerbations and myasthenic crisis and in the perioperative setting. Despite many years of experience, the role of thymectomy in improving long term outcome in nonthymomatous myasthenia gravis remains controversial.

Adjuvants, Immunologic↗

PubMed1996

Myasthenia-like syndrome induced by overdosage of cibenzoline.

A case of cibenzoline-induced myasthenia-like syndrome is reported. A 67-year-old woman with renal failure and no previous disorder of neuromuscular junction complained of fatigue during climbing up a flight of stairs and experiencing heavy eyelids after administration of 100 mg/day of cibenzoline. Repetitive nerve stimulation tests revealed decrement at 5-10 Hz. After reduction of the dosage, myasthenia-like signs and symptoms disappeared. The peak cibenzoline concentration was still high even after the dose reduction (666.4 ng/ml). In conclusion, cibenzoline, at a high plasma level, may induce myasthenia-like syndrome without any disorder of the neuromuscular junction.

Aged↗