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At least 19 recordsLinked to original sources
PubMed1975

A therapeutic trial of fresh plasma infusions over a period of 22 months in two siblings with Hunter's syndrome.

The clinical and biochemical changes following long-term treatment with infusions of fresh plasma over a period of 22 months are outlined in two siblings with the mild type of Hunter's syndrome. During the first six months of treatment, the clinical status of both siblings was characterized by accelerated growth, reduction in the size of liver and spleen, improvement in joint movement and diminution in the tendency to respiratory infections. During the remaining 16 months, these changes were less conspicuous. The first plasma infusion resulted in parallel, albeit transient, changes in pattern of urinary glycosaminoglycan excretion, manifested mainly by an increase in the cetylpyridinium-chloride-nonprecipitable glycosaminoglycan fraction. The main factor contributing to this increase was found to be heparan sulfate. After subsequent plasma infusions, however, the basic pattern of urinary glycosaminoglycan excretion remained the same as before treatment, although convincing clinical changes were still evident for at least the first six months in both siblings.

Blood Transfusion↗
PubMed1979

Treatment of mucopolysaccharidosis: clinical and biochemical aspects of leucocyte transfusion as compared with plasma infusion in patients with Hurler's and Scheie's syndromes.

The therapeutic effectiveness of leucocyte transfusion (LT) was compared with that of plasma infusion (PI) clinically by range of motion (ROM) of joints and biochemically from the standpoint of alpha-L-iduronidase activity and urinary excretion of acid mucopolysaccharides (AMPS) in 2 patients with Hurler's and Scheie's syndromes. Both syndromes are considered to be due to the lack of alpha-L-iduronidase activity, a congenital metabolic disorder. As a result, leukocyte transfusion surpasses plasma infusion with respect to enzyme content, the grade and duration of clinical improvement in the stiffness of joints. Clinical improvement in the stiffness of joints was correlated with the degradation of AMPS when the ratio of urinary AMPS fragments to the total large molecule AMPS has become 50% or more after the leucocyte transfusion and plasma infusion.

Adult↗
PubMed1975

Ultrastructure of the skin in mucopolysaccharidoses. Studies performed before and after plasma infusion therapy.

Skin biopsy specimens of two siblings with Hunter syndrome and two siblings with type B Sanfilippo syndrome were examined by electron microscopy, at six-month intervals, before and after plasma infusion therapy. Electron-lucent, membrane-bound vacuoles were seen in fibroblasts, Schwann cells, and in mononuclear cells both before and after the infusions. Based on the ultrastructural, clinical, and biochemical findings, it was concluded that there was no therapeutic benefit from these plasma infusions.

Child↗
PubMed1978

Limited effect of erythrocyte and plasma infusions in adenosine deaminase deficiency.

A 10-month-old child with a profound deficiency of adenosine deaminase and severe combined immunodeficiency was treated for a period of 17 months with red cell and plasma transfusions containing normal amounts of the deficient enzyme. Following each transfusion, the plasma adenosine, red cell and lymphocyte ATP, urinary adenine, and urinary deoxyadenosine decreased transiently. During this period, the absolute blood lymphocyte count rose and a limited increased in the response of the lymphocytes to PHA-P was observed. Delayed hypersensitivity skin tests remained negative during the transfusion periods. A quantitative elevation of serum immunoglobulins occurred, but specific antibody formation was not elicited. In contrast to a previous report of successful therapy of ADA deficiency with red cell and plasma infusions, this patient responded poorly to enzyme replacement therapy. The difference may be related to a more profound enzyme deficiency in our patient.

Adenine↗
PubMed1976

Plasma human calcitonin (hCT) levels in normal and pathologic conditions, and their responses to short calcium or tetragastrin infusion.

Plasma hCT levels were less than 50 pg/ml in 50 normal subjects. In 16 patients with medullary carcinoma of the thyroid (MCT), plasma hCT levels were distinctively elevated and they fell significantly after total thyroidectomy, but in 11 of them plasma levels were still high, indicating the presence of metastases. In 74 patients with the other types of malignancy, plasma hCT levels were found to be high in 9 cases (3 oat cell carcinoma of the lung, 4 malignant carcinoids, one malignant pheochromocytoma and one acute myelocytic leukemia). Except for the leukemic case, all these tumors were derived from neural crest. In 12 patients with primary hyperparathyroidism, plasma hCT levels were less than 20 pg/ml. In 13 hypoparathyroid patients, two with pseudohypoparathyroidism and one with pseudoidiopathic hypoparathyroidism, plasma hCT levels were slightly elevated. Some patients with uremia had elevated plasma hCT levels, but there was no relation between plasma levels of hCT and those of PTH, urea nitrogen or creatinine. In response to Ca (4.5 mg/kg/10 min) or tetragastrin (4 mug/kg/5 min) infusion, a marked increase in plasma hCT was observed in all patients with MCT, but not in normal subjects. In 5 hypoparathyroid patients, a significant increase to both stimuli was also observed in all cases. Two patients with pseudopseudohypoparathyroidism responded to the Ca load. These results indicate that the determination of plasma hCT levels especially after a short Ca or tetragastrin infusion is important to study various pathological conditions.

Calcitonin↗
PubMed2024

Hereditary thrombotic thrombocytopenic purpura mimicking immune thrombocytopenia was revealed by miscarriage-novel compound heterozygous mutations in hTTP.

We report a case of early-onset hereditary thrombotic thrombocytopenic purpura in a 16-year-old girl who suffered from thrombocytopenia and was misdiagnosed with immune thrombocytopenia for years until two failed gestations finally revealed the underlying cause. The novel compound heterozygous mutation c.2865G > A:p.Trp955X and c.721delG: p.Gly241fs in the ADAMTS13 gene were identified and are predicted to be associated with this disease. The patient responded to plasma therapy, including plasma infusion and plasma exchange, but renal dysfunction may be longstanding.

Adolescent↗
PubMed1976

Effect of angiotensin II on cyclic guanosine monophosphate and cyclic adenosine monophosphate in human plasma.

Infusion of alpha-adrenergic catecholamines increases plasma cyclic guanosine monophosphate (pcGMP), raising the possibility that the pressor effect of these agents may be mediated by cyclic GMP. We infused pressor doses of angiotensin II in 10 studies in 8 normal subject and measured pcGMP and plasma cyclic adenosine monophosphate (pcAMP) by radioimmunoassay. After 120 minutes of infusion, mean pcGMP was 128 +/- 31% (SE) higher than baseline values (P less than 0.01) while pcAMP was increased 30 +/- 10% (P less than 0.05).

Angiotensin II↗
PubMed1976

Plasma volume changes after infusion of various plasma expanders.

In the immediate post-operative period after moderate surgical procedures, 1 litre of a colloid solution or saline was given intravenously. The plasma volume expansion after infusion of dextran 70 (Macrodex), hydroxyethylstarch (Volex), polygelatin (Haemaccel), albumin and saline was found to be between 790 and 180 ml. The most efficent plasma expander was dextran, followed by hydroxyethylstarch. Polygelatin and saline did not give full restitution, although twice the calculated loss was infused. Total plasma protein concentration was lowered in all groups in proportion to the dilution, except for the patients given albumin, in whom the concentration of total protein increased. Calculation of the total circulating protein mass showed no decrease during the period immediately after the infusion. This investigation has demonstrated that the most efficient plasma volume expander is dextran but that hydroxyethylstarch offers an almost equal alternative in terms of volume expansion. Dextran, however, exerts an advantageous effect on the microcirculation. As the metabolic pathways of hydroxyethylstarch have not yet been further explored, dextran is preferred when using artificial colloids. Judged by its secondary effects alone, including the influence on plasma protein patterns, albumin seems to be the compound of choice. Polygelatin and saline are not efficient expanders when hypovolaemia is to be corrected rapidly.

Adult↗
PubMed1979

Recent therapeutic advances in thrombotic thrombocytopenic purpura.

Whole plasma infusion, in our experience, has been highly effective in the management of patients with thrombotic thrombocytopenic purpura. The effectiveness of plasma infusion in the treatment of this severe disorder implies the deficiency of a factor in the patient's plasma. Furthermore, we have observed that when platelets are suspended in plasma obtained during active, untreated thrombotic purpura, aggregation occurs. This effect is neutralized by preincubation of the thrombotic thrombocytopenic purpura plasma with normal plasma. Thus, there is both a correlation with the clinical pathogenic mechanism, disseminated platelet aggregation, and with the therapeutic response to plasma infusion. Based upon our experience and the concept that thrombotic thrombocytopenic purpura is a plasma factor deficiency state, we recommend initial infusion of a full plasma volume equivalent over the first 24 hours. This should be done under an intensive care setting. After this initial plasma infusion, we advise the infusion of 3 units of plasma daily until a full remission is obtained. When the clinical situation has stabilized, we stop the daily plasma infusions and cautiously observe for recurrence of the manifestations of thrombotic thrombocytopenic purpura. If there is a recurrence, plasma is again infused in substantial quantity during the first 24 hours and then 3 units daily until a full remission is again evident. Whether the plasma requirement might be attenuated or the course of the disease shortened by the concomitant use of antiplatelet agents, corticosteroids or other means remains to be determined.

Adolescent↗
PubMed1978

Degradation and clearance of methotrexate in children with osteosarcoma receiving high-dose infusion.

Plasma methotrexate (MTX) concentrations were quantitated in 34 patients after 127 high-dose (35--350 mg/kg) infusions with citrovorum factor rescue. Significant linear correlations have been obtained between methotrexate dosage and concentrations in plasma at 6 and 24 hours after the initiation of the therapy. However, similar trends have not been observed when 48- and 72-hour samples were analyzed. Clinical toxicity was not serious when the methotrexate level in plasma was less than 4.5 X 10(-6) M at 48 hours after the start of a six-hour infusion in children. A minimal four-hour steady-state methotrexate plasma level can be maintained during a six-hour infusion. Children excrete methotrexate at a faster rate than adults; the half-life of MTX during the first phase of plasma clearance curve is one hour shorter in children. Urinary analyses have indicated that substantial methotrexate is metabolized. The chemical nature of these components has not been identified. Further, the urinary metabolic profiles varied among patients.

Adolescent↗
PubMed1977

Effect of glucagon infusion on plasma cyclic AMP in patients with cholestatic hepatitis and obstructive jaundice. New test of hepatic cholestasis.

Plasma cyclic AMP concentration during glucagon infusion at various time intervals was determined in 8 normal subjects, 9 patients with extrahepatic obstructive jaundice and 10 patients with cholestatic hepatitis (hepatitis A and B). Plasma cyclic AMP concentrations (pmol/ml) during glucagon infusion in patients with both obstructive jaundice and cholestatic hepatitis were found to be greater than those in control subjects. In addition, a significant difference in plasma cyclic AMP concentrations was found between patients with cholestatic hepatitis and obstructive jaundice at the 10th minute of glucagon infusion. These results indicate that plasma cyclic AMP levels at the 10th minute of glucagon infusion represent a reliable diagnostic index of cholestatic jaundice.

Cholestasis↗
PubMed1976

Effect of norepinephrine infusion on plasma vasopressin levels in normal human subjects.

Norepinephrine was continuously infused for 30 minutes at a rate of 0.2 and 0.3 mug/kg/min into 3 normal human subjects, and plasma vasopressin levels, plasma osmolality, hematocrit value, blood pressure, and heart rate were determined simultaneously. In norepinephrine infusion, an elevation of mean blood pressure and a decrease in heart rate was seen, and the degree of these changes was greater with the infusion at a rate of 0.3 mug/kg/min. Plasma vasopressin levels were suppressed by the infusion, and a dose-response relationship was recognized between the infusion at rates of 0.2 mug/kg/min and 0.3 mug/kg/min, while plasma osmolality did not change, and the hematocrit value rose slightly. These results suggest that norepinephrine-induced water diuresis is related to the suppression of the vasopressin release.

Adult↗