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Biomedical subjects

M Olarte

Publications and source records attributed to M Olarte.

14 recordsLinked to original sources

Molecular and clinical characterisation of three Spanish families with maternally inherited non-syndromic hearing loss caused by the 1494C->T mutation in the mitochondrial 12S rRNA gene.

Mutations in the 12S rRNA gene of the mitochondrial genome are responsible for maternally inherited non-syndromic hearing loss (NSHL), and for increased susceptibility to the ototoxicity of aminoglycoside antibiotics. Among these mutations, 1555A-->G is the most prevalent in all populations tested so far. Recently, the 1494C-->T mutation was reported in two large Chinese pedigrees with maternally inherited NSHL. In this study, sequencing of the 12S rRNA gene in a Spanish family with maternally inherited NSHL showed the presence of the 1494C-->T mutation. An additional screening of 1339 unrelated Spanish patients with NSHL allowed the authors to find two other families with the mutation. Audiological data were obtained from 17 confirmed 1494C-->T carriers, which showed that the hearing loss was sensorineural, bilateral and symmetrical, with a remarkable variability in age of onset and severity. Three carriers were asymptomatic. Three affected carriers had a history of treatment with aminoglycoside antibiotics. The mitochondrial genome of one affected person from each of these three families was entirely sequenced, and it was established that they belong to different mitochondrial haplogroups (H, U5b, U6a). The study results further support the pathogenic role of 1494C-->T on hearing, and show that this mutation can be found in different Caucasian mitochondrial DNA backgrounds.

Adult↗

Concurrent herpes simplex type 1 and varicella-zoster in the V2 dermatome in an immunocompetent patient.

A unique feature of herpesviruses is their ability to establish latent infection within the nervous system by colonizing peripheral sensory ganglia, which results in subsequent episodic outbreaks of infection triggered by precipitating events. Despite the latent nature of both herpes simplex virus type 1 (HSV-1) and varicella-zoster virus (VZV) within these sensory ganglia, simultaneous outbreaks of these viruses are uncommon. This is generally attributed to the differing reactivation features of these 2 viruses. Four cases of concurrent HSV-1 and VZV infection are described in the literature. We report concurrent infection of HSV-1 and VZV within the same V2 dermatome in an immunocompetent patient.

Acyclovir↗

Antisulfatide antibodies in neuropathy: clinical and electrophysiologic correlates.

OBJECTIVE: To investigate the clinical and electrophysiologic characteristics of the neuropathy associated with elevated serum antisulfatide antibodies. METHODS: Clinical, electrophysiologic, morphologic, and laboratory data of 25 patients with significantly elevated (>25,600) antisulfatide antibodies were reviewed. RESULTS: Four groups were distinguished based on clinical and electrophysiologic data: Group 1, eight patients with predominantly small fiber sensory neuropathy (32%); Group 2, five patients with mixed large and small fiber sensory neuropathy (20%); Group 3, seven patients with axonal sensorimotor neuropathy (28%); and Group 4, three patients with demyelinating sensorimotor neuropathy (12%). One additional patient had mononeuritis multiplex and one had ALS. An immunoglobulin M (IgM) monoclonal gammopathy was found in 30% of the patients tested, but not in any of the Group 1 patients with small fiber sensory neuropathy. Serum IgM level was elevated in 12 patients, of whom six had a concomitant monoclonal gammopathy. Morphologic studies in five patients showed predominantly axonal degeneration, with three of the patients also exhibiting additional features of demyelination. CONCLUSIONS: Antisulfatide antibodies are associated with several subtypes of peripheral neuropathy. Predominantly sensory or sensorimotor axonal neuropathies are most common in this series, with the sensory component either small fiber or mixed fiber type. A smaller demyelinating group indistinguishable from patients with chronic inflammatory demyelinating polyradiculopathy was also seen. One third of patients had a concomitant IgM monoclonal gammopathy, and approximately one half had elevated serum IgM.

Adult↗

Recurrent abrupt occlusion after transluminal angioplasty for basilar artery stenosis: case report.

OBJECTIVE AND IMPORTANCE: Angioplasty for basilar artery stenosis is often complicated by recurrent abrupt vessel closure. The clinical results can be catastrophic. In this case report, we assess the effects of intra-arterial papaverine (American Regent Laboratories Inc., Shirley, NJ) on rebound occlusion. CLINICAL PRESENTATION: The patient presented with crescendo transient ischemic attacks from atherosclerotic narrowing of the midbasilar artery despite maximal medical treatment. INTERVENTION: Angioplasty of the midbasilar artery was performed with serial balloon inflations. The patient was treated successfully with intra-arterial papaverine and achieved a nearly full recovery, with only mild dysarthria, by the time of the 7-month follow-up examination. CONCLUSION: Using intra-arterial papaverine, we were able to reverse the effects of this potentially life-threatening complication of basilar artery angioplasty.

Angioplasty, Balloon↗

Diagnostic biopsy of the motor nerve to the gracilis muscle. Technical note.

A biopsy specimen of the motor nerve to the gracilis muscle was obtained in 151 patients to aid in the diagnosis of neuromuscular disorders. The procedure is simple, safe, and has low morbidity and complication rates. The surgical technique is described. Indications for the use of this technique are motor neuropathy, motor neuron disease, Guillain-Barre syndrome, and mixed neuropathy with prominent motor symptoms.

Adult↗

Cisapride and metoclopramide in the treatment of gastroesophageal reflux disease.

In a double-blind, randomized study, the clinical effects of 5 mg and 10 mg of cisapride three times daily were compared with those of 10 mg of metoclopramide three times daily in 114 patients with symptoms of gastroesophageal reflux, mainly diurnal and nocturnal heartburn and regurgitation. The symptoms significantly (P less than 0.001) improved in the three groups; the mean severity score decreased by at least 78% after four weeks of treatment. Initial symptoms were more severe in the cisapride-treated patients, especially in those receiving 10 mg three times daily; however, the patients' condition after four weeks was similar in the three groups. Central nervous system side effects were reported by one patient from each of the cisapride-treated groups and by nine of the 43 metoclopramide-treated patients (P less than 0.02). Six metoclopramide-treated patients and one cisapride-treated patient dropped out of the study because of side effects. These findings favor the use of cisapride when prokinetic treatment of gastroesophageal reflux is considered.

Adult↗

Muscle phosphoglycerate mutase deficiency.

A 52-year-old man complained since adolescence of cramps and pigmenturia after 15 to 30 minutes of intense exercise. There was no family history of neuromuscular diseases, and strength was normal. The rise of venous lactate after forearm ischemic exercise was abnormally low. Histochemical and ultrastructural studies of a muscle biopsy showed mild increase of glycogen, which was confirmed by biochemical analysis. Studies of anaerobic glycolysis in vitro showed decrease lactate formation with glycogen and with all hexosephosphate glycolytic intermediates, suggesting a defect below the phosphofructokinase reaction. Muscle phosphoglycerate mutase (PGAM) activity was 5.7% of the lowest control, while all other enzymes of glycolysis had normal activities. Electrophoretic, heat lability, and mercury inhibition studies showed that the small residual activity of PGAM in the patient's muscle was represented by the brain (BB) isoenzyme, suggesting a genetic defect of the M subunit that predominates in normal muscle. The prevalence of the BB isoenzyme in other tissues, including muscle culture, may explain why symptoms were confined to muscle.

Adult↗

Emery-Dreifuss muscular dystrophy.

A man had weakness of humeroperoneal distribution associated with limited range of motion of the cervical spine and elbows. At age 25 he developed permanent atrial paralysis, and a cardiac pacemaker was inserted. Although this case was sporadic, most others have been transmitted as an X-linked recessive trait. Mixed patterns in electromyography and muscle histology have caused nosological confusion, but the unique clinical signs seem to define a distinct form of muscular dystrophy, warranting the designation "emery-Dreifuss" type.

Adult↗

Debrancher deficiency: neuromuscular disorder in 5 adults.

Five patients, 4 men and 1 woman, had adult-onset and slowly progressive weakness. There was distal wasting in 2, hepatomegaly in 3, and congestive heart failure in 2. Electromyography showed a mixed pattern with abundant fibrillations. Serum creatine phosphokinase was increased 5- to 45-fold. Blood glucose failed to respond to epinephrine or glucagon, and venous lactate did not rise after ischemic exercise. Muscle biopsy showed vacuolar myopathy affecting both fiber types. By electron microscopy the vacuoles corresponded to large pools of glycogen not limited by a membrane. Glycogen concentration was 3 to 5 times normal in muscle and 7 to 21 times normal in erythrocytes. In the presence of iodine, muscle glycogen showed a spectrum characteristic of phosphorylase-limit-dextrin. Debrancher activity was measured by a spectrophotometric assay and by a radioactive reverse reaction. The activity was lacking in muscle and erythrocytes of 4 patients according to both assays; in 1 patient the reverse reaction was not impaired. Though previously reported in only 5 patients, debrancher deficiency myopathy may not be rare and should be considered in the differential diagnosis of adult-onset hereditary myopathies.

Adolescent↗

Accessory nerve palsy.

After apparently uncomplicated excision of benign lesions in the posterior cervical triangle, two patients had shoulder pain. In one, neck pain and trapezius weakness were not prominent until one month after surgery. Inability to elevate the arm above the horizontal without externally rotating it, and prominent scapular displacement on arm abduction, but not on forward pushing movements, highlighted the trapezius dysfunction and differentiated it from serratus anterior weakness. Spinal accessory nerve lesions should be considered when minor surgical procedures, lymphadenitis, minor trauma, or tumours involved the posterior triangle of the neck.

Accessory Nerve↗

Biochemical abnormalities of erythrocyte membranes in Duchenne dystrophy. Adenosine triphosphatase and adenyl cyclase.

Adenosine triphosphatase (ATPase) activity in erythrocyte membranes from patients with Duchenne dystrophy was inhibited by ouabain less than in normal individuals in assay systems containing high or low contents of salt. Epinephrine and cyclic adenosine monophosphate increased total ATPase activity in all samples, and epinephrine restored ouabain sensitivity to the Duchenne membranes. Basal adenyl cyclase activity in about twice that of controls. Epinephrine stimulated adenyl cyclase activity of normal membranes two to three times, but did not stimulate the enzyme in Duchenne membranes. These differences may reflect a genetic abnormality of the membrane.

Adenosine Triphosphatases↗

Lipid storage myopathy, ichthyosis, and steatorrhea.

A 41-year-old man had ichtyrosis, ectropion, steatorrhea, and slowly progressive proximal limb weakness. Biopsies showed abnormal lipid accumulation in muscle, liver skin, leukocytes, and gastric mucosa. Lipid storage was particularly marked in cultures of skin and muscle, and it increased in subsequent cell generations. By electron microscopy, the lipid globules showed no limiting membranes. The stored lipid was identified by thin-layer chromatography as triglyceride; there was no excess of cholesterol or cholesteryl esters. Muscle carnitine concentration and activities of carnitine palmityltransferase and acid lipase were normal; 14CO2 production from labeled palmitate in leukocytes was not impaired. The excessive accumulation of triglyceride in different tissues and in the progeny of cells in tissue culture suggests a genetic error of lipid metabolism.

Adult↗