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Biomedical subjects

O Trygstad

Publications and source records attributed to O Trygstad.

At least 37 records · Page 2Linked to original sources

Echocardiographic assessment of cardiac function and morphology in patients with generalised lipodystrophy.

Because cardiomegaly has been observed in lipodystrophic patients we studied cardiac morphology and function with one- and two-dimensional echocardiography in addition to general cardiologic examination in a series of seven patients. Muscular hypertrophy with increased chamber size and myocardial indentations were found. Two patients had asymmetrical septal hypertrophy (ASH), and two patients demonstrated systolic anterior movement (SAM) of the mitral valve. Wall motion analysis showed anomalities in four patients during contraction, in three during the early relaxation phase. Since pathological findings, probably increasing with age, were made in the majority of the patients, these findings add an additional unfavourable aspect to the syndrome.

Adolescent↗

Chromatographic profiles at E-280 nm for urinary precipitates in morbus Rett.

Previously we have observed different characteristic chromatographic ultraviolet absorbancy profiles at 280 nm for urinary protein precipitates from patients with behavioral disorders. The purpose with this study was to look for similar changes in urinary protein excretion from the grossly disabled patients with the Rett syndrome. Benzoic acid precipitates from the urine from 20 patients were chromatographed on a Sephadex G-25 gel column. There appeared 3 different profiles: 6 as observed in normal controls, 5 like that in compulsive behavior, and 9 were similar to those observed for autistiform-schizophreniform patients. The profiles did not mirror the behavioral observations for the patients, although the first group of 6 patients were the least disabled, whereas the last 9 patients were the most seriously ill. In conclusion the chromatographic profiles suggest a heterogeneous group of patients, but we need more observations for several patients over time, as there may be a change with the exacerbation of the disease.

Adolescent↗

Spinal deformities in tall girls.

In a prospective study, 62 girls who consulted the paediatric department because of tall stature were examined for spinal deformities. Thirteen cases of scoliosis measuring 10 degrees or more were found. Eighteen girls had a thoracic kyphosis of more than 40 degrees and 11 had additional vertebral abnormalities indicating Scheuermann's disease. The incidence of scoliosis and Scheuermann's disease was much higher in our material than normal.

Adolescent↗

Acute in vivo stimulatory effects of human pituitary lipid-mobilizing factor on plasma levels of glucagon and insulin in rabbits.

The lipid-mobilizing factor LMF is prepared from deep-frozen human pituitary glands by alkaline extraction, followed by acetone precipitation at pH 4.8, Sephadex gel filtration and DEAE-cellulose chromatography. In addition to its adipokinetic effect in rabbits, LMF also increased the plasma levels of glucagon and insulin in rabbits in doses of 15 to 100 micrograms. The LMF-induced increases in the plasma levels of glucagon were most pronounced in fasted rabbits, whereas the increases in the plasma levels of insulin were most pronounced in fed rabbits. Glucose infusions decreased the LMF-induced hyperglucagonaemia and increased the LMF-induced hyperinsulinaemia. Somatostatin did not inhibit the LMF-induced hyperglucagonaemia with statistical significance, but inhibited the LMF-induced hyperinsulinaemia. The plasma levels of glucose were slightly decreased by 20 and 40 micrograms LMF in fasted rabbits and were unchanged in fed rabbits. In fasted rabbits, LMF had a toxic effect and 100 micrograms LMF killed one rabbits. Human growth hormone (hGH), prepared from the pituitary glands after removal of LMF, also increased the plasma levels of glucagon and insulin in rabbits. It is possible that the observed effects of LMF and hGH were due to the presence of some biologically active substances from the pituitary gland. These postulated substances could be involved in the pituitary control of the endocrine pancreas, and work is in progress to isolate them from the LMF preparation.

Animals↗

Somatomedin levels in pregnancy: longitudinal study in healthy subjects and patients with growth hormone deficiency.

Somatomedin (Sm) levels throughout pregnancy were determined in a longitudinal study of four normal women and three patients with GH deficiency by use of the RIA for Sm-A, a newly developed RIA for insulin-like growth factor 2 (IGF-2), and the placenta RRA for Sm-A. In both normal women and those with GH deficiency, there was a continuous rise of immunoreactive Sm-A throughout pregnancy. During the third trimester the levels were 2-fold elevated above the level in nonpregnant age-matched normal subjects. No change of immunoreactive IGF-2 levels was found in the normal pregnant women, whereas an increase from low to normal levels was found in GH-deficient patients during pregnancy. The placenta RRA-Sm-A did not detect the increase of Sm-A immunoactivity in the normal pregnant women, whereas the levels were normalized in GH-deficient patients. After delivery a rapid fall of Sm levels occurred in patients with GH deficiency. The calculated half-lives for immunoreactive Sm-A and IGF-2 were 27 and 52 h, respectively. The birth weights of the seven children were significantly (P less than 0.05) correlated to both the individual peak and the mean maternal value of immunoreactive Sm-A during the last trimester. The present findings indicate that the production of both IGF-1 and IGF-2 related peptides during pregnancy is independent of maternal pituitary GH production.

Adult↗

Chromatographic analysis of urine and dialysate from schizophrenic patients undergoing hemodialysis.

Maladjustments in the central nervous system may result in altered concentrations of neuropeptides in cerebrospinal fluid and urine. Urinary protein-peptide precipitates from psychiatric patients have previously been observed to have different chromatographic profiles with regard to the ultraviolet absorbancy at 280 nm. Hemodialysis has previously been reported to improve some schizophrenic patients and has been recommended in treatment trials. Seven chronic schizophrenic patients were selected for hemodialysis in a double-blind crossover study. Specimens of 24-h urine collections were obtained the day before, the day of, and the day after active and sham dialysis. The chromatographic profiles obtained on Sephadex G-25 gel columns did not change with active dialysis. Materials with ultraviolet absorbancy at 280 nm also precipitated from dialysates and showed the same pattern following active and inactive dialysis. Thus, no biochemical evidence for removal of any pathogenetic humoral substance during hemodialysis could be provided.

Adult↗

Childhood psychosis and urinary excretion of peptides and protein-associated peptide complexes.

Twenty-four infantile autistic children were compared with 12 children with other kinds of childhood psychoses, 5 children with so-called minimal brain dysfunction syndrome, 5 children with attention deficit disorder, 14 children with mental retardation, and 19 normal children with regard to chromatographic profiles of urinary products that give ultraviolet absorbency at 280 nm. Six main types of chromatographic patterns emerged. Fifty-four percent of the autistic children and 17% of the children with other psychoses showed a distinct pattern that was not seen in any other cases. Only 8% of the autistic children showed the "normal" pattern seen in 95% of the normal and 93% of the mentally retarded children without psychosis. The ultraviolet absorbency peaks of the chromatograms possibly correspond to peptides and protein-associated peptide complexes. It is argued that these products are probably at least partly of CNS origin.

Adolescent↗

Pancreatic B-cell function and abnormal urinary peptides in a boy with lipoatrophic diabetes and stenosis of the aqueduct of Sylvius.

A boy with the classical clinical manifestations of acquired lipoatrophic diabetes has been studied for 5 years from the onset of diabetes at age 13. At the age of 15 a ventriculo-cisternal shunt operation was performed because of stenosis of the aqueduct of Sylvius, followed by a dramatic improvement in his diabetic state with a decrease of the 24 hr insulin requirement from 130 to 32 units. After 12 months there was a relapse with increased insulin requirement up to the preoperative level. Pimozide treatment was given for 7 months with no effect on the metabolic derangements. Extremely high basal levels of serum C-peptide and pro-insulin were found throughout the period of observation. A further increase occurred after i.v. arginine infusion tests, indicating hyperfunctioning B-cells. Repeated screenings of peptides in the urine by sephadex chromatography revealed pathological patterns similar to those observed in patients with other hypothalamic disorders, but different from that found in the urine of patients with congenital generalized lipodystrophy. Injection into mice of peptides extracted from the preoperative urine produced an acute hyperglycemia. The mechanisms behind this hypothalamic syndrome are unknown, but it is postulated that the abnormal urinary polypeptides originate from disorganized hypothalamic centres and that these peptides may be responsible for the disturbed carbohydrate and lipid metabolism.

Adolescent↗

Fat cell size and lipid content of subcutaneous tissue in congenital generalized lipodystrophy.

Fat cell size and lipid composition of subcutaneous tissue from 3 patients with congenital generalized lipodystrophy have been measured before and after treatment with either pimozide, fenfluramine or hypophysectomy. The fat cell volume before treatment ranged between 9.8 x 10(4) and 17.7 x 10(4) micron 3, compared to 9.0 x 10(4) and 85.3 x 10(4) micron 3 in 15 controls. The amount of lipids was only 1/10 to 1/50 of normal, triglycerides being the most heavily reduced lipid component. Neither the fat cell size nor the lipid content were affected by treatment with hypophysectomy, pimozide or fenfluramine.

Adipose Tissue↗

Transitory growth hormone deficiency successfully treated with human growth hormone.

This study was carried out in order to determine whether children with a transitory type of growth hormone deficiency showed an accelerated growth in height velocity on treatment with human growth hormone (HGH). Following careful diagnostic routine procedures 13 extremely short children were diagnosed as having isolated growth hormone deficiency, and were successfully treated with HGH. A true isolated growth hormone deficiency was present in 5 of the children, whereas 8 showed a normal increase in serum growth hormone on repeated growth hormone stimulation tests after their development of puberty and termination of HGH treatment. Three boys with bone ages of 5.5, 8.0 and 9.5 years showed an undisputable effect following HGH administration. They showed an initial growth at the start of treatment, and a second growth spurt during development of puberty. Two of the boys reached final statures of 14 cm taller than the predicted heights. The other patients, including the children with true isolated growth hormone deficiency showed an initial spurt of growth at the start of the HGH treatment immediately followed by a pubertal growth spurt. The mean accleration of height velocity for the children with true isolated growth hormone deficiency was from 3.4 cm during the year before treatment to 7.0 cm during the first year on treatment, as compared to 2.8 and 7.4 cm, respectively, for the children with transitory growth hormone deficiency. A girl with severe anorexia nervosa who had a transitory growth hormone deficiency, showed an accelerated high velocity from 1.1 cm to 7.6 cm during the first year following treatment with HGH.

Adolescent↗

Congenital generalized lipodystrophy and experimental lipoatrophic diabetes in rabbits treated successfully with fenfluramine.

Lipoatrophic diabetes has been produced in rabbits by injection of a fraction prepared from the urine from patients with congenital generalized lipodystrophy. Both these conditions are considered to be hypothalamic syndromes. The animals, and a patient with congenital generalized lipodystrophy and latent diabetes were treated with the dopamine receptor blocker, pimozide, for 4 and 17 months, respectively. The results were discouraging even though the patient got a daily dose of 16 mg pimozide. Fenfluramine has a lowering effect on brain serotonin, and peripheral effects on glucose and triglyceride metabolism. This drug improved the general condition of the rabbits with lipoatrophic diabetes, as well as that of the patient with congenital generalized lipodystrophy. The rabbits became normoglycaemic and insulin sensitive. In the patient a normalization of the urinary excretion of the serotonin metabolite 5-OH-indole acetic acid was observed. His voracious hunger and profuse perspiration were reduced, the hyperkeratotic layer of the skin peeled off, and the pigmentations of the skin decreased. There was observed an improvement of ALAT and ASAT, normalization of the fasting blood glucose, and increased sensitivity to exogenous insulin. After 11 months of 200 mg fenfluramine daily addtitional administration of 2 g clofibrate per day produced normalization of the serum triglyceride concentration and a marked reduction of the resistance to insulin. Three more patients with congenital generalized lipodystriphy, two of whom have manifest diabetes, have now started treatment with fenfluramine and are improving. The rabbits got relapse of their lipoatrophic diabetes when the fenfluramine treatment was stopped. It is suggested that a disturbance in the serotonin metabolism of the central nervous system may be of pathogenetic importance in congenital generalized lipodystrophy.

Adult↗

Lipodystrophic diabetes treated with fenfluramine.

Congenital generalized lipodystrophy is considered to be a diencephalic syndrome with disturbance of hypothalamic transmitters. After puberty and arrest of growth the patients develop a serious untreatable diabetes mellitus. One of our patients, a girl 15 years of age, developed a lipodystrophic diabetes with fasting blood glucose levels above 300 mg/100 ml, increased serum insulin with insulin resistance, and hyperlipidaemia. Daily administration of fenfluramine gave a dramatic improvement. The voracious hunger and profuse perspiration were reduced, the patient's serum lipids became normal, her blood glucose fell, and her sensitivity to exogenous insulin increased. A normalization of the urinary excretion of the serotonin metabolite, 5-OH-indole acetic acid, was observed.

Adolescent↗

Congenital contractural arachnodactyly.

Five cases of congenital contractural arachnodactyly (CCA) are reported. Three belong to the one family. CCA has often been mistaken for Marfan's disease and arthrogrypois multiplex. Because CCA has a more favourable prognosis, it is very important to be able to recognize this syndrome.

Adolescent↗