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PubMed1989

Histopathological heterogeneity and cytopathological similarity of findings in different muscles of two brothers affected by rigid spine syndrome.

The pathological changes in muscles biopsied from 2 brothers with rigid spine syndrome are reported. The findings ranged from marked fascicular atrophy and fibrosis to hypotrophy of small groups of fibres and vacuolation in most fibres. The presence of vacuoles and deposits of accumulated material seemed to be common to all the biopsies. These findings, compared with those reported in the literature, confirmed the histopathological heterogeneity of this syndrome but proposed also the hypothesis that similar elementary lesions of muscle fibres can account for the initiation of the pathological process, developing asynchronously in different muscles because of their different activity.

Adult↗
PubMed2003

Head retraction reflex in stiff-man syndrome and related disorders.

The head retraction reflex (HRR) is a vestigial withdrawal reflex of the face and is suppressed in healthy subjects. We investigated the prevalence and electrophysiological patterns of the HRR in patients suffering from stiff-man syndrome (SMS, n = 28) and related disorders, stiff-limb syndrome (SLS, n = 2), and progressive encephalomyelitis with rigidity and myoclonus (PERM, n = 20). In patients with a positive HRR, the electromyographic (EMG) pattern was analyzed with surface recordings from the orbicularis oculi, sternocleidomastoid, trapezius, and the paraspinal extensor muscles at midthoracic and lumbar levels. On clinical testing, 17 of 28 SMS patients, 10 of 20 PERM patients, and 0 of 2 SLS patients had a positive HRR, ranging from a brief contraction of the neck extensors to violent retropulsion of the upper body. In all muscles, EMG reflex patterns elicited by gentle taps to the face or by electrical stimulation of the trigeminal nerve branches consisted of two components: an early, synchronous, brief burst with the shortest latency in trapezius (12.5-20.0 msec) and a late, asynchronous, longer response with latencies between 44 and 70 msec. We conclude that the HRR is an abnormal cutaneomuscular brainstem reflex that occurs in a considerable proportion of patients with SMS and related disorders. Although neither specific nor particularly sensitive for SMS, presence of the HRR in a setting with otherwise unexplained stiffness and spasms might help to avoid the otherwise frequent misdiagnosis of a psychogenic motor disturbance in patients with SMS.

Brain Stem↗
PubMed1985

Anesthetic induction with fentanyl.

The efficacy of fentanyl, 30 micrograms/kg, was evaluated as an anesthetic induction agent in 72 ASA I-III patients scheduled for 2-4-hr operations. The effect of preinduction pretreatment with pancuronium and/or diazepam and the incidence of loss of consciousness (anesthesia), recall, rigidity, abnormal muscle movements, and hemodynamic changes were documented. Seventy-four percent of all patients became anesthetized. Diazepam pretreatment enhanced but did not ensure success of anesthetic induction. There was a significant correlation between age and the incidence of unconsciousness (P = 0.0287) and all patients over 60 yr old were anesthetized with 30 micrograms/kg of fentanyl. The incidence and severity of rigidity was reduced by pancuronium (P = 0.0002) but not by diazepam pretreatment. However, pancuronium plus diazepam produced a significant reduction in the incidence of rigidity when compared to pancuronium alone (P = 0.031). A significant positive correlation between age and the incidence of rigidity (P = 0.003) was found. Six patients had focal and one patient global tonic-clonic abnormal muscle movements. Diazepam but not pancuronium significantly decreased both heart rate (P = 0.05) and blood pressure (P = 0.04). Seventeen patients required reversal of narcotic effect to restore adequate spontaneous respiration after surgery. No patient required postoperative mechanical ventilatory assistance. The results of this study demonstrate that 30 micrograms/kg of fentanyl is not a reliable anesthetic induction dose in patients less than 60 yr old. Both age and premedication enhance the anesthetic capabilities of induction with fentanyl.(ABSTRACT TRUNCATED AT 250 WORDS)

Adolescent↗