Cases reported "Lymphangiomyoma"

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1/2. Failure of buserelin-induced medical castration to control pulmonary lymphangiomyomatosis in two patients.

    Two women, aged 44 and 29 years, respectively, were admitted to the hospital in early 1987 for recurrent pneumothorax, dyspnea and a diffuse reticulonodular pattern evidenced on the chest x-ray film. lung biopsy confirmed LAM in both patients. Both were treated sequentially with medroxyprogesterone and a LHRH agonist (buserelin) to achieve reversible medical castration. Neither subjective nor objective improvement was noted after 13 and 5 months, respectively, of buserelin therapy (900 micrograms/day, nasal spray) despite an effective suppression of the pituitary-gonadal axis. medroxyprogesterone also was ineffective. buserelin thus failed to control pulmonary LAM in these two patients, in spite of effective medical castration.
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2/2. Pulmonary lymphangiomyomatosis: treatment with castration.

    Pulmonary lymphangiomyomatosis (PLM) is a rare disease of smooth muscle proliferation that generally leads to an early death due to respiratory insufficiency. The disease only affects women. Exacerbations with pregnancy and after hormonal manipulation have been documented. There is presently no successful treatment of the disease. castration as a means of early therapy has been associated with a stable respiratory picture in our patient. Peritoneal-jugular shunt can benefit women with significant ascities.
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