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Congenital insensitivity to pain remains without a satisfactory physiopathological explanation. In an electrophysiological study on a nociceptive flexion reflex of the lower limb, the effects of naloxone and of placebo were compared in 8 normal subjects and in a patient with congenital insensitivity to pain. In normal subjects, no significant change in the reflex threshold was observed with naloxone or with placebo. In contrast, two electrophysiological abnormalities characterized the patient: (1) spontaneous elevation in the nociceptive reflex threshold of 350% as compared to control, and (2) a large (67%) and rapid (2–3 min) fall of this threshold for about 10 min following the administration of naloxone.These results raise the problem of the relationship between congenital insensitivity to pain and an hyperactivity of a naturally occurring “morphine-like” pain-inhibitory system.