Ischemic mononeuropathy and mononeuropathy multiplex in diabetes mellitus.
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BACKGROUND: Mononeuropathy of a cranial nerve caused by brain metastases rarely occurs in patients with a malignant neoplasm. Even after the development of computed tomography (CT) and magnetic resonance imaging (MRI), few cases of brain metastases resulting in trigeminal mononeuropathy have been reported. We report a case of trigeminal mononeuropathy caused by a brain metastases that was detected on CT and MRI preoperatively, and was successfully treated by microsurgery and radiation. CASE DESCRIPTION: A 49-year-old woman with a history of breast cancer complained of right facial pain. CT and MRI revealed a brain tumor in the right Meckel's cave and cerebellopontine angle. She underwent surgery, and the diagnosis was metastatic breast cancer. No recurrence was detected on MRI performed 2 years after the resection and radiation of the tumor. CONCLUSIONS: Most of the reported cases of brain metastases causing a mononeuropathy occurred before the availability of CT; the mononeuropathy was diagnosed by neurologic or postmortem examination. Even after the development of CT and MRI, few cases of brain metastases resulting in trigeminal mononeuropathy have been reported. If such patients do not manifest symptoms of brain metastases other than the mononeuropathy, it is difficult to make the correct diagnosis without CT and MRI. Physicians must be aware of mononeuropathy as an uncommon presenting symptom of brain metastases.
Controversy exists with regard to the actual prevalence of compressive mononeuropathies at the wrist which may occur following chronic paraplegia. Thirty one chronic paraplegics, with a mean age of 37.9 years (range 20-68 years), and mean time since injury of 9.7 years (range 1-28 years), were studied with a comprehensive neurologic and electrodiagnostic (EDX) assessment. No patient had any clinical or EDX evidence of a peripheral polyneuropathy. The diagnosis of a median mononeuropathy at the wrist was determined by the following criteria: (a) prolonged median sensory distal latency greater than ipsilateral ulnar sensory distal latency greater than or equal to 0.5 msec; (b) a median mid-palmar sensory latency greater than ipsilateral ulnar mid-palmar sensory latency of greater than or equal to 0.3 msec; or (c) a median motor distal latency greater than or equal to 1.7 milliseconds as compared to the ipsilateral ulnar motor distal latency. Ulnar mononeuropathy at the wrist or across the elbow was also characterised. The EDX criteria for a median mononeuropathy at the wrist was met in 55% of subjects (24% of these with bilateral presentations). The location of ulnar mononeuropathies included: two at the superficial sensory branch at the wrist, one at the deep motor branch at the wrist, and three patients with a conduction block across the elbow. Overall, 67% of all patients tested had evidence of at least one mononeuropathy of the upper extremity. There was no association between prevalence of compressive mononeuropathies and age of the patient or time since onset of injury.
Fifty-one diabetic patients with mononeuropathies wereoneuropathy were studied to examine possible etiological factors, to determine the relationship with other diabetic complications, and to correlate with the presence and severity of background peripheral and autonomic neuropathy. The median, ulnar, and lateral popliteal nerves were most commonly affected and cranial neuropathy was relatively uncommon. When bilateral involvement of the same nerve was excluded, multiple mononeuropathies were found in only five patients. Median and ulnar mononeuropathy were gradual in onset and affected the dominant limb whereas other types of mononeuropathies were acute in onset with no predilection for either side. No consistent relationship was shown between the onset of mononeuropathy and age, sex, diabetic treatment, duration of diabetes, diabetic control, or other diabetic complications. In particular, there was no significant background peripheral and autonomic neuropathy, as assessed clinically and by objective tests, in almost one-half of the patients studied. It is concluded that diabetic mononeuropathy may occur independently of peripheral and autonomic neuropathy. It is possible, however, that a minimal degree of background damage, known to be present in all diabetic patients, may render them more susceptible than the general population to the various factors causing mononeuropathy.
OBJECTIVE: To determine which proposed risk factor, work activity (industrial v clerical), body mass index (BMI), or other demographic factors had the most influence on the prevalence of median mononeuropathy at the wrist, and if there was an interaction between the risk factors. METHODS: This was a cross sectional study of active workers at five different worksites; four were industrial sites and one was clerical. 527 workers were recruited--164 clerical and 363 industrial. The presence of a median mononeuropathy in either hand was measured by electrodiagnostic techniques comparing median and ulnar sensory latencies. RESULTS: 30% of workers had an abnormality of the median sensory nerve at the wrist (34% of the industrial v 21% of the clerical workers). The adjusted risk for industrial workers was twice that of clerical workers. Obese workers (BMI > 29) were four times more likely to present with a median mononeuropathy than workers who were normal or slender (BMI < 25). There was no significant interaction between BMI and worksite in relation to median mononeuropathy. Increasing age was also related to an increased risk of median mononeuropathy. CONCLUSIONS: Obesity, industrial work, and age are independent risk factors that influence the prevalence of median mononeuropathies among active workers.
Although diabetic mononeuropathy affecting the cranial and peripheral nerves is well recognized, there is little known or documented about diabetic mononeuropathy affecting the thoracic nerves, i.e., the truncal nerves. This series of 40 cases attests to its frequency; equal sex distribution; significance in differential diagnosis including coronary artery disease, intra-abdominal surgical diseases such as gallbladder pathology and appendicitis, pleurisy, and neoplasms. Truncal mononeuropathy has characteristics that differ from those of other diabetic mononeuropathies in that it is primarily sensory and typically not a first manifestation of clinical diabetes, whereas the other forms of diabetic mononeuropathy are primarily motor in effect and not infrequently may be the initial clinical presenting manifestation of diabetes. Finally, diabetic truncal mononeuropathy has a good prognosis.
The objective was to determine whether symptomatic workers with an abnormal sensory nerve conduction study consistent with carpal tunnel syndrome differed, in terms of electrophysiologic measures, psychosocial, demographic, anthropometric, or ergonomic variables, from workers with an asymptomatic median mononeuropathy. This was a cross-sectional study of active workers at six different work sites. Cases were defined as workers with electrodiagnostic findings of a median mononeuropathy in either hand, based on a 0.5-msec prolongation of the median sensory evoked peak latency compared to the ulnar latency. This group was stratified on the basis of symptoms of numbness, tingling, burning or pain in the hand. The two groups were compared in terms of demographic, anthropomorphic, psychosocial, electrophysiologic, and ergonomic risk factors. Active workers from six different sites were tested; five sites involved manufacturing workers, and one site represented clerical workers. One hundred eighty-four active workers with a median mononeuropathy were documented on nerve conduction studies. These workers represented a subset of more than 700 workers screened at six different locations. The main outcome measure was the patient's report of symptoms of pain, numbness, tingling or burning in the hand or fingers that lasted more than 1 week or occurred three or more times at the initial screening. Workers with a median mononeuropathy who complained of hand symptoms were more likely to be female, to have jobs with higher hand repetition levels, to have higher ratings of job security, not to have a history of diabetes, to use more force in their job with more abnormal postures of their wrist and fingers, and to have a trend toward a more prolonged median sensory distal latency. Most logistic regression models explained less than 15% of the variance (pseudo R2). Women with jobs that have higher ergonomic risks and no history of diabetes were more likely to have reported symptoms associated with carpal tunnel syndrome compared to other workers with a documented median mononeuropathy. Psychosocial variables were not particularly discriminatory. None of the models allows enough precision to predict on an individual basis.
We reported the first case of Rothmann-Makai syndrome associated with mononeuropathy in a 31-year-old woman. In June 1989, she noticed several small subcutaneous nodules in the bilateral upper arms. On July 2, she woke up because of severe pain below the knee in the right leg which lasted 15 hours. On admission, subcutaneous nodules were recognized on bilateral upper arms, abdomen, gluteal regions and thighs. None of these nodules were associated with local heat or pain. Muscle weakness and dysesthesia were observed in the right foot, but deep tendon reflexes were normal. Electrophysiological studies revealed a decrease of the amplitude of action potential and of the conduction velocity in the right tibial nerve. Microscopic examination of skin biopsy showed lipolysis and diffuse lymphocyte infiltration (panniculitis) and perivascular lymphocyte infiltration was also observed. Routine laboratory data including peripheral blood, ESR, blood chemistry, and CRP were in the normal range and antinuclear antibodies and LE cells were not detected. By those findings, she was diagnosed as Rothmann-Makai syndrome associated with mononeuropathy. Steroid treatment was effective on both skin lesions and mononeuropathy. Steroid treatment was effective on both skin lesions and mononeuropathy. We concluded that this mononeuropathy may be induced by a ischemic change which may be caused by panniculitis.
We report a case of localized hypertrophic mononeuropathy that involved the trigeminal nerve in a 61-year-old woman with a 5-year history of progressive drooping of the left eyelid, double vision, and left-sided temporal and retro-orbital pain. A mass that occupied the patient's left cavernous sinus was found to display localized hypertrophic mononeuropathy characterized by "onion bulbs" that were formed of concentrically proliferating Schwann cell processes around intact axons. This contrasted to the majority of previously reported cases of localized hypertrophic mononeuropathy, predominantly found in peripheral nerves, where the proliferating cell processes have been shown to be of a perineurial cell origin. Differences in architectural arrangement and degree of cellularity between the perineurial and schwannian forms of localized hypertrophic mononeuropathy were noted. These findings suggest important fundamental differences in the pathogenesis of various forms of onion-bulb mononeuropathies.
Mononeuropathies are unusual at birth, and electromyographic (EMG) definition the first day of life has not been reported previously. Although neonatal mononeuropathies may be related to obstetric complications, prenatal mechanisms also merit consideration. We report an infant, born with a peroneal neuropathy, whose EMG was performed 18 h after birth. An isolated peroneal nerve lesion with lack of compound muscle action potential and the presence of fibrillation potentials, confined to the tibialis anterior muscle, suggested a primary intrauterine mechanism for this mononeuropathy. Because of an infant's small size, the temporal profile used in adults for appearance of EMG signs of wallerian degeneration may not apply. Inaccurate conclusions may result if the EMG standards for timing adult nerve injury are applied to newborns. To our knowledge, previous published cases of neonatal mononeuropathies have not included babies whose first EMG was performed before age 4 days. Therefore, an EMG study shortly after birth needed to be accomplished if strong support for the hypothesis of a prenatal onset were to be generated. Our findings are compatible with an intrauterine onset of this baby's peroneal neuropathy.
In the current investigation we examined a peripheral mononeuropathy induced by four loose ligatures around the sciatic nerve. The nerve was treated with lidocaine or saline before implementing the ligatures (silk or chromic gut). The latency of the hindlimb withdrawal to noxious mechanical and radiant heat stimuli was tested under light pentobarbital anesthesia 3-10 days following the surgery. A latency/threshold difference > or = 15% between the hindlimbs was considered to represent a change in the nocifensive response. The adapting skin temperature of the hindlimbs was also measured. Pieces of hindpaw skin and the sciatic nerve were taken for histological evaluation. The results indicate that the incidence of hyperalgesia induced by a constriction mononeuropathy depended on the noxious submodality tested, and that the thermal and mechanical hyperalgesia were not coupled in all cases. Use of only one test modality led to false negative results. Furthermore, mononeuropathy-induced changes in the adapting skin temperature produced a considerable number of false positive results (an artefactual hyperalgesia) when radiant heat alone was used to determine the nocifensive withdrawal latency without paying attention to the abnormality of the skin temperature. A preemptive lidocaine treatment of the sciatic nerve before the nerve ligation significantly reduced the incidence of mononeuropathy-induced hyperalgesia. The nerve ligation material (silk vs chromic gut) was not a significant factor for the development of hyperalgesia induced by a constriction injury of a peripheral nerve. In histological evaluation the constriction injury-induced damage of the sciatic nerve was verified, and the inflammatory reaction caused by chromic gut was not stronger than that caused by silk ligatures. In general, immunohistochemical staining for substance P decreased whereas that for VIP increased. The results support the hypothesis that ligation-induced mechanical trauma and the afferent barrage induced by it, especially during the perioperative period, plays an important role in the development of postoperative hyperalgesia.
Using electrophysiological recordings, we studied a distal tibial mononeuropathy that involves the hind foot of rats reared in cages with wire grid flooring. In an initial set of experiments, serial sciatic-tibial motor conduction recordings were made in smaller or larger rats reared in cages with wire grid or sawdust flooring. Electrophysiological features of the neuropathy were loss in the amplitude of the distal tibial nerve M potential recorded over hind limb foot muscles, temporal dispersion of the potential, often into multiple peaks, and a prolonged distal latency of the response. The changes in M amplitude were more apparent in larger rats with a greater body weight. In a second series of experiments we studied sciatic-tibial conduction over 16 weeks in nondiabetic rats and rats rendered diabetic with streptozotocin raised and wire grid or plastic flooring. Tibial mononeuropathy developed in both wire grid-reared groups, but there was evidence that it appeared earlier in diabetic rats. Electrophysiological changes of distal mononeuropathy also obscured the expected slowing of sciatic-tibial motor conduction velocity from diabetics. Tibial mononeuropathy in rats reared on wire grid flooring may be a useful animal model of human entrapment neuropathy but its presence can confound studies of experimental neuropathy. Rats used in studies of experimental neuropathy should be housed in plastic cages with sawdust or shavings flooring.
OBJECTIVE: To report the clinical and pelvic CT findings in six patients with obturator mononeuropathy caused by cancer. DESIGN: A clinical case series of six patients followed for 2 months to 10 years (one patient lost to follow-up). SETTING: Three referral centers. PATIENTS: Three men and three women, ages 52 to 81 years. Three patients had transitional cell carcinoma of the bladder, and one patient each had pelvic papillary carcinoma, carcinoma of unknown origin, and lymphoma. MAIN RESULTS: In each patient, symptoms of obturator mononeuropathy were the sole presenting sign of new or recurrent pelvic cancer. Three patients had ipsilateral leg edema in addition to the typical sensory and motor findings of obturator mononeuropathy. Tumor sites detected on pelvic CT that correlated with obturator nerve compression or infiltration, singly or in combination, included the posterolateral wall of the upper pelvis or midpelvis, the anterior wall of the lower pelvis, and the external obturator and pectineus muscles extrinsic to the bony pelvis. Antineoplastic treatment provided symptomatic relief in four patients. CONCLUSIONS: Pelvic CT or MRI should be performed to exclude pelvic tumor in patients with obturator mononeuropathy if there is no temporal association with pelvic trauma or intra-abdominal, pelvic, or hip surgery.
The objective of this study was to investigate the central processing of dynamic mechanical allodynia in patients with mononeuropathy. Regional cerebral blood flow, as an indicator of neuronal activity, was measured with positron emission tomography. Paired comparisons were made between three different states; rest, allodynia during brushing the painful skin area, and brushing of the homologous contralateral area. Bilateral activations were observed in the primary somatosensory cortex (S1) and the secondary somatosensory cortex (S2) during allodynia compared to rest. The S1 activation contralateral to the site of the stimulus was more expressed during allodynia than during innocuous touch. Significant activations of the contralateral posterior parietal cortex, the periaqueductal gray (PAG), the thalamus bilaterally and motor areas were also observed in the allodynic state compared to both non-allodynic states. In the anterior cingulate cortex (ACC) there was only a suggested activation when the allodynic state was compared with the non-allodynic states. In order to account for the individual variability in the intensity of allodynia and ongoing spontaneous pain, rCBF was regressed on the individually reported pain intensity, and significant covariations were observed in the ACC and the right anterior insula. Significantly decreased regional blood flow was observed bilaterally in the medial and lateral temporal lobe as well as in the occipital and posterior cingulate cortices when the allodynic state was compared to the non-painful conditions. This finding is consistent with previous studies suggesting attentional modulation and a central coping strategy for known and expected painful stimuli. Involvement of the medial pain system has previously been reported in patients with mononeuropathy during ongoing spontaneous pain. This study reveals a bilateral activation of the lateral pain system as well as involvement of the medial pain system during dynamic mechanical allodynia in patients with mononeuropathy.
The present study was performed in rats with experimentally induced mononeuropathy after left common sciatic nerve ligation. The hindpaw withdrawal latencies to thermal and mechanical stimulation increased significantly after intra-periaqueductal grey injection of 2 or 3nmol, but not 1nmol of galanin in rats with mononeuropathy. Intraperitoneal administration of 4.5mg/kg morphine induced significant increases in hindpaw withdrawal latencies to both noxious stimulation, which were attenuated by following intra-periaqueductal grey injection of 2nmol of the galanin antagonist galantide. Furthermore, the antinociceptive effect induced by intra-periaqueductal grey injection of 26.6nmol of morphine was attenuated significantly by following intra-periaqueductal gray administration of 2nmol of galantide. The results demonstrated that in periaqueductal grey galanin plays an antinociceptive role in rats with mononeuropathy and galanin is involved in the mechanisms of opioid-induced antinociception.
A 31-year-old man from Myanmar with leprous neuropathy was reported. The progress of the disease was subacute but the painful symptom at the time of the onset was acute. Multiple mononeuropathy was diagnosed by the biopsy findings of the left superficial radial nerve. He was admitted to our hospital with the complaint of the weakness of his left hand and fingers which were very painful and got worse in several weeks. Motor palsy was observed in his left ulnar, median, and radial nerves, and there was the hypesthesia or anesthesia in his left hand, forearm and the medial side of his left upper arm. On nerve conduction studies, the amplitudes of CMAP and SNAP severely diminished or not detected. The pattern was compatible with multiple mononeuropathy. The biopsy of the left superficial radial nerve was performed. The pathological findings were the destruction of nerve fascicles, replacement of nerve fibers with inflammatory cells, and Mycobacterium leprae was found with the specific stain. These findings confirmed the diagnosis of the leprous neuropathy. Leprous neuropathy is one of the commonest causes of infectious neuropathy in the world, especially in Southeast Asia. These days many foreign workers from that area are staying in Japan, and the chances to see the disease are increasing. We have to recognize leprous neuropathy as a candidate for the multiple mononeuropathy of acute onset with painful dysesthesia similar to vascular neuropathy.
Mononeuropathies associated with orthotopic liver transplantation were evaluated in a prospective manner. Ten percent of liver transplant recipients were noted to have focal peripheral nerve lesions in the postoperative period. The ulnar nerve was most commonly involved, with intraoperative compression or postoperative trauma as possible mechanisms of injury. Other upper extremity mononeuropathies were likely a result of vascular cannulations. No brachial plexus injuries occurred. Diabetes and alcoholism were not risk factors for the development of a mononeuropathy.
Seventeen children with pediatric peroneal mononeuropathies evaluated between 1979 and 1991 are reported. Twelve boys and 5 girls, ranging in age from 1.5 months to 17 years, were referred for footdrop in 16 children (94%) or for lower extremity pain in 1 child (6%). Causes included compression in 10 children (59%), trauma in 3 children (18%), entrapment in 3 children (18%), and indeterminate in 1 child (5%). Based on nerve conduction studies and electromyography, the level of the pediatric peroneal mononeuropathic lesion was the common peroneal nerve in 10 children (59%), the deep peroneal nerve in 2 children (12%), and the superficial peroneal nerve in 1 child (5%). In 4 other children (24%), pediatric peroneal mononeuropathy at the knee was not more precisely identified. Surgical exploration in 3 children with progressive pediatric peroneal mononeuropathy was valuable. Improvement occurred in 13 of 17 children (76%).