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    • Manber et al evaluated the Behavioral

    2018-11-13

    Manber et al. evaluated the Behavioral Cognitive Therapy in patients with MD and insomnia, where they found improvement in insomnia symptoms as well as in productivity, energy, severity of the depressive symptoms, suicidal thoughts and self-esteem [48].
    Conclusions
    Introduction Duchenne Muscular Dystrophy (DMD) is a neuromuscular disease linked to the X chromosome which afflicts 1 in every 3500 born boys. DMD is caused by a mutation on the SR 3576 cost gene at locus Xp.21, leading to dystrophin absence or a production defect in the muscles. Affected individuals demonstrate progressive loss of muscle tone and respiratory, cardiac and orthopedic impairment. Loss of walking capacity usually occurs around 9–11 years of age and death between 20 and 30 years of age [1]. Neuromuscular diseases, in general, may be developed by chronic pain as a result of contractures, deformities and immobility, with great impact on the quality of life [2]. Pharmacological treatment with amytriptiline chloride constitutes one of the multimodal aspects of intervention for the pain treatment in patients with neuromuscular diseases [3]. There is a possible association between tricyclic antidepressants and the restless leg syndrome (RLS) [4].
    Case report Male patient with 27 years of age, with diagnostic of Duchenne Muscular Dystrophy at the age of 7 years. Beginning of symptoms at the age of 6, with myopathic walk and difficulty in climbing stairs. Loss of walking ability at 10. With 19 years old, evolved to alveolar hypoventilation syndrome and the need of ventilatory support with non-invasive mechanical ventilation. At this age, the patient already demonstrated movement restriction, with global tetraparesis. At the age of 23, initiated with diffuse and continuous chronic pain, worsening of passive limb movements and daily chronic migraine resulted from analgesic abuse. We then initiated treatment with a tricyclic antidepressant (amytriptiline chloride 25mg/night). After a few days of therapy the patient referred a feeling of undetermined discomfort in his inferior limbs, beginning every day in the evening, with intense will to move his legs, only relieved by passive movement of his legs (done by his caretaker since he was incapable of doing it himself). The phenomenon lasted for 2h, making it difficult to sleep and with spontaneous remission. The patient had demonstrated similar symptoms previously although with less intensity, duration and frequency. There was no evidence of periodic limb movements (patient with global tetraparesis and motor strength grade 1, making it impossible to move the limbs), peripheral neuropathy, spinal radiculopathy or other associated conditions. He had positive familiar history (mother refers some characteristically similar episodes, not related to any drug usage, with low frequency and intensity). Blood tests were considered normal. With the interruption of the medication, after approximately 30 days, the patient showed marked improvement of this condition, with decrease of the episodes and symptoms, maintaining its eventual frequency. The punctuation of the Naranjo and cols Drug Adverse Reaction (DAR) probability scale (1981) was of 6 points, indicating a probable side effect of the drug (Table 1).
    Discussion Patients with DMD show high risk for the development of respiratory disorders related with sleep, mainly the sleep obstructive apnea syndrome and alveolar hypoventilation [5]. Other sleep disorders include difficulty in initiating and maintaining sleep. Many patients show important sleep fragmentation with frequent awakenings for decubital changes (by the caretakers) due to pain or discomfort [6]. There is very little information about movement disturbances during sleep in these patients. RLS is a motor-sensitive disorder related to sleep, characterized by discomforting or painful paresthetic sensations in the legs, between the ankle and the knee at rest, accompanied by the intense need to move the affected limbs. The RLS physiopathology is still not established; however, there are some hypothesis related to the mechanisms of central and peripheral nervous system processing and some elements of the motor system, as well as the dopaminergic and iron metabolisms systems. RLS may be classified as idiopathic, genetic with a dominant autosomic heritage pattern, or secondary to a subjacent clinical condition. The RLS diagnosis is exclusively clinical, obeying the minimal criteria in accordance to the International Restless Legs Syndrome Study Group – IRLSSG and of the International Sleep Disorders Classification of 2005, being essential criteria the following: irresistible and intense need to move the legs, generally accompanied by discomfort or inconvenience; the symptoms worsen or are exclusively present at rest or during inactivity, sitting or laying down; the symptoms are alleviated totally or partially with movement; symptoms worsen or occur exclusively at night. The described symptoms are not best explained by any other diseases of conditions. The support criteria for diagnostic of RLS are: elevated index of periodic leg movement during sleep; positive familiar history; therapeutic response to dopaminergic agents; clinical course [7,8].