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H G Tiselius

Publications and source records attributed to H G Tiselius.

At least 19 recordsLinked to original sources

Identification of 12 novel mutations in the SLC3A1 gene in Swedish cystinuria patients.

Cystinuria is an autosomal recessive disorder that affects luminal transport of cystine and dibasic amino acids in the kidneys and the small intestine. Three subtypes of cystinuria can be defined biochemically, and the classical form (type I) has been associated with mutations in the amino acid transporter gene SLC3A1. The mutations detected in SLC3A1 tend to be population specific and have not been previously investigated in Sweden. We have screened the entire coding sequence and the intron/exon boundaries of the SLC3A1 gene in 53 cystinuria patients by means of single strand conformation polymorphism (SSCP) and DNA sequencing. We identified 12 novel mutations (a 2 bp deletion, one splice site mutation, and 10 missense mutations) and detected another three mutations that were previously reported. Five polymorphisms were also identified, four of which were formerly described. The most frequent mutation in this study was the previously reported M467T and it was also detected in the normal population with an allelic frequency of 0.5%. Thirty-seven patients were homozygous for mutations in the SLC3A1 gene and another seven were heterozygous which implies that other genes may be involved in cystinuria. Future investigation of the non-type I cystinuria gene SLC7A9 may complement our results but recent studies also suggest the presence of other potential disease genes.

Adolescent↗

Crystallisation properties in stone forming and normal subjects' urine diluted using a standardised procedure to match the composition of urine in the distal part of the distal tubule and the middle part of the collecting duct.

Using a standardised procedure, we assessed the crystallisation properties of calcium phosphate in urine with a composition matching that in the distal part of the distal tubules (DTd) and of calcium oxalate in urine with a composition matching that in the mid-collecting duct (CDm). We used 8-h urine samples collected between 2200 h and 0600 h with sodium azide as preservative. Urine from ten patients with recurrent CaOx stone formation and from ten normal subjects was used for the measurements. The DTd and CDm samples were obtained by diluting the voided 8-h urine to 3000 ml and 1750 ml per 1.73 m2 body surface area, respectively. The nucleation was studied in DTd urine following supersaturation with CaP. The crystal size distribution was assessed with a Coulter counter both following supersaturation of DTd urine with CaP and of CDm urine with CaOx. The crystallisation of CaP in DTd urine as well as that of CaOx in CDm urine, in the presence of CaP crystals that had been precipitated in DTd urine, was measured with the isotope technique. The inhibition of CaOx and brushite crystal aggregation in standardised diluted aliquots of DTd and CDm urine was assessed spectrophotometrically as the rate of sedimentation. There was a slightly increased sedimentation rate and a lower initial absorbance in DTd urine from stone formers supersaturated with CaP. Although these findings might reflect a state of increased crystal aggregation in stone formers' urine, this could not be confirmed by crystal size measurements in the Coulter counter. The inhibition of brushite crystal aggregation in DTd urine was significantly in stone formers' urine than in normal subjects' urine (P < 0.001). Moreover, all inhibition values in DTd samples from stone formers were negative, suggesting a promoter effect on crystal aggregation. The inhibition of CaOx crystal aggregation in CDm urine also was significantly higher in CDm urine from normal subjects than in CDm urine from stone formers (P < 0.05). For all other variables the level was similar when urine samples from the two groups were compared. Although this series of crystallisation assessments was carried out on a small number of standardised diluted urine samples only, the results nevertheless emphasise a defect in aggregation inhibition as one important determinant for an abnormal calcium salt crystallisation in patients with recurrent stone formations. This difference obviously includes aggregation of both CaP crystals in DTd urine and CaOx crystals in CDm urine. The results also show that assessment of crystallisation properties of this kind can be carried out in standardised, diluted 8-h night urine samples, which accordingly can be used in the routine work-up of patients with calcium stone disease. Such an approach might prove useful in order to get information on the combined effects of the driving force of supersaturation and crystallisation modifying properties accomplished by urinary macromolecules and other modifying agents.

Crystallization↗

A comparison of the effects of potassium citrate and sodium bicarbonate in the alkalinization of urine in homozygous cystinuria.

For many years, urine alkalinization has been one of the cornerstones in the treatment of homozygous cystinuria. Because of the relationship found between the excretion of urinary sodium and cystine, potassium citrate has emerged as the preferred sodium-free alkalizing agent. To evaluate the usefulness of potassium citrate for urine alkalization in cystinuric patients, sodium bicarbonate and potassium citrate were compared in 14 patients (10 on tiopronin treatment and four without treatment with sulfhydryl compounds). The study started with 1 week without the use of any alkalizing agents (Period 0) followed by 2 weeks with sodium bicarbonate (Period 1) and 2 weeks with potassium citrate (Period 2). Urinary pH, volume, excretion of sodium, potassium, citrate and free cystine, as well as the plasma potassium concentration, were recorded. Potassium citrate was shown to be effective as an alkalizing agent and, in this respect, not significantly different from sodium bicarbonate. Even though a normal diet was used, a significant increase in urinary sodium excretion was observed with sodium bicarbonate (Period 1). Urinary potassium and citrate excretion increased with potassium citrate (Period 2). A significant correlation was found between urinary sodium and cystine in the tio-pronin-treated patients. No significant differences in cystine excretion were recorded in Periods 0, 1 and 2. Plasma potassium was significantly higher during Period 2, but only one patient developed a mild hyperkalemia (5.0 mmol/l). The use of potassium citrate for urine alkalization in homozygous cystinuria is effective and can be recommended in the absence of severe renal impairment.

Adult↗

Cystine analyses of separate day and night urine as a basis for the management of patients with homozygous cystinuria.

Based on previous observations of the diurnal variation of urinary cystine excretion, the use of separate day and night urine collections was proposed. To improve the medical treatment of patients with cystinuria, this strategy was performed to guide the fluid intake and the administration of SH compounds (tiopronin, D-penicillamine, and MESNA).Twenty-six patients (19 treated with SH compounds and seven with alkalinization and hydration only) were followed during two 3.5-year periods. During Period 1, 24-h urine was collected and during Period 2, separate day and night urine was collected. There were 56 episodes of high urinary cystine supersaturation (> 1,200 micromol/l) during Period 2, 47% of which would have evaded detection with 24-h urine analysis. In comparison with Period 1, the urinary cystine concentration was lower (P < 0.05), and the urinary volume was higher (P < 0.05) during Period 2. Patients treated with tiopronin had reduced cystine excretion (P < 0.05) and at the end of Period 2, an increased dose of tiopronin, reflecting a more aggressive treatment. Furthermore, a reduced number of stone episodes and need of active stone removal (P < 0.05) was noted in the whole group of patients. Analyses of separate day and night urine samples can be used advantageously to reveal episodes of high supersaturation with cystine not detected in 24-h urine samples. Such a procedure might be useful for optimizing the treatment of patients with cystinuria.

Adult↗

Methods for measuring crystallization in urolithiasis research: why, how and when?

Whereas crystalluria does not distinguish between kidney stone formers and healthy people and thus can be considered a physiologic event, kidney stone formation is a pathologic incident and reflects a specific form of biomineralization. Both single urinary crystals as well as whole kidney stones form under exquisite control of organic macromolecules. Simple crystal formation in the urinary tract is distinguished from stone formation in the kidney by the process of particle retention. The latter occurs either because nucleated crystals strongly aggregate to particles too large to pass freely through the tubules ('free particle' theory), or because crystals become abnormally adherent to tubular cell surfaces ('fixed particle' theory). Since it is impossible to mimic all the processes involved in stone formation in vitro, it is highly important to carefully chose a specific crystallization process for in vitro studies, and to select the most appropriate experimental conditions for measuring the chosen process as reliably as possible. This overview aims at critically reviewing the principles of currently available assay systems for studying crystallization processes involved in stone formation. Consensus is reached by the experts that no in vitro system really mimics what happens in renal stone formation, but that carefully designed in vitro studies will always play an important part in urolithiasis research. For such studies, it is highly important to exactly control the appropriate experimental conditions that are relevant to a specific crystallization process under investigation. Practical guidelines for researchers working with crystallization systems are provided, and it is concluded that international efforts should be made to standardize the terminology, to agree on a set of basic experimental parameters (temperature, pH, artificial urine composition), and to adopt simple tests or conditions are reference points for quality and comparative control.

Calcium Oxalate↗

Guidelines on urolithiasis.

OBJECTIVES: A project was initiated by the Health Care Office of the European Association of Urology in order to formulate common recommendations and guidelines for the treatment of patients with urolithiasis. The basic task for the working group therefore was to extract and evaluate evidence from the literature in order to reach a consensus on how these patients could best be managed. METHODS: Extensive reviews of the literature together with a thorough and detailed discussion of the various topics, by a working group including of experts from several European countries, provided the basis for a consensus overview of urolithiasis and its management. RESULTS AND CONCLUSIONS: Recommendations are given for the management of patients with acute stone colic and for active removal of stones from the ureter and kidney. Moreover, the principles for risk evaluation of patients with recurrent stone formation and appropriate recurrence preventive treatment are given.

Calcium↗

Long-term follow-up of stone formers treated with a low dose of sodium potassium citrate.

We evaluated the clinical efficacy of long-term preventive treatment with a single evening dose of alkaline citrate. Information was collected from the files of 52 recurrent stone formers prescribed a daily intake of 3.75-5 g of sodium potassium citrate (SPC; 14-18 mmol of citrate). The annual and cumulative rates of stone formation and the rate of recurrence were compared before and during the treatment. A comparison was also made between the patients with (Group R) and without (Group NR) recurrent stone formation during treatment in terms of urine composition and previous history of the disease. For all patients who started the treatment, the number of stones was smaller during treatment (period tT) than during a period of the same length immediately before treatment (period tB), but greater than the number formed during a corresponding period immediately after the diagnosis (period tA). Via questionnaire we found low treatment compliance, with only 62% of the patients reporting consistent taking of their medication (Group T). The patients in Group T had a smaller cumulated number of stones during period tT than that during periods tA and tB, but the Kaplan-Meier curve of the fraction of patients remaining stone-free during treatment was almost identical to that recorded in 446 recurrent stone formers without medical treatment. No significant differences were recorded in terms of relevant pretreatment urinary risk factors between Groups T(R) and T(NR), but numerically higher values of calcium oxalate (CaOx) supersaturation and calcium/citrate quotients were observed in Group T(R). When 9 patients with a daily intake of SPC and a citrate excretion below 2.5 mmol/day were compared with 16 hypocitraturic patients only given drinking advice, the cumulated percentages of patients without recurrent stone formation in the 2 groups after 3 years were 44% and 48%, respectively. Although the number of patients in this study was small, our results indicate poor long-term protection from recurrent calcium stone formation when a single evening dose of only 3.75-5 g of SPC was taken. The rate of stone formation was apparently slightly reduced, but the fraction of patients free of recurrence was no different from that in patients without medical treatment.

Citrates↗

Metabolic evaluation and therapy.

The recurrent formation of stones in the urinary tract is a well-recognized clinical problem. Unfortunately, progress in the medical care of these patients has not paralleled that of the surgical care. There are, however, several steps that can be taken to reduce the recurrence rate. In this regard it is desirable to identify patients at risk and to find risk factors that need to be eliminated. The extent of biochemical evaluation and the form of stone preventive treatment in patients with calcium stone formation is presently a matter of debate. This review focuses on some current aspects on the analysis of risk factors for calcium stone formation. The present situation on recurrence prevention using dietary and pharmacological measures is also summarized.

Calcium↗

The effect of pH changes on the crystallization of calcium salts in solutions with an ion composition corresponding to that in the distal tubule.

The effect of pH changes on the crystallization in solutions with an ion composition assumed to correspond to that of urine in the distal part of the distal tubule was examined by recording the number and volume of crystals with a Coulter Multisizer and by studying the crystal morphology with scanning electron microscopy at different degrees of volume reduction. The experiments were carried out with 100 ml samples at different starting pH values without and with 20% of dialysed urine (dU). The number of crystals increased in response to the volume reduction. In solutions without dU, 100 or more crystals with a diameter in the range 2.4-45 microm were observed already at a volume reduction of 40% when the initial pH was 7.28. For solutions with a pH of 5.80 and 6.45 the corresponding values were 60% and 80%, respectively. In the presence of dU, an appearance of crystals was recorded at volume reductions of less than 20%. In solutions with an initial pH of 5.80 and 6.45, the crystal number was greater with dU than without; such a difference was not recorded at pH 7.28. In samples containing dU, the mean crystal volume (MCV) varied very little when the sample volume was reduced. The same was found in solutions without dU when the initial pH was 5.80 and 7.28, whereas the MCV was greater in the samples with pH 6.45. Scanning electron microscopy of solutions reduced to 30-40% of the original volume showed that calcium phosphate had formed in solutions with a starting pH of 7.28 and 6.45. In solutions with pH 5.80 calcium oxalate crystals were observed with calcium phosphate.

Calcium Oxalate↗

Crystallization during volume reduction of solutions with a composition corresponding to that in the collecting duct: the influence of hydroxyapatite seed crystals and urinary macromolecules.

To examine the effect of hydroxyapatite (HAP) seed crystals and urinary macromolecules on the crystallization under conditions similar to those in the collecting duct, we evaporated 100 ml samples of salt solutions with an ion composition assumed to correspond to that in the collecting duct without and with HAP seed crystals. The crystallization in seeded solutions was assessed both with and without dialysed urine (dU). After evaporation the number and volume of crystals were recorded in a Coulter Multisizer and the crystal morphology examined with scanning electron microscopy (SEM) and X-ray crystallography. Addition of HAP crystals was apparently followed by an approximately 15-20% increase in heterogeneous nucleation of calcium oxalate (CaOx). In these experiments SEM and X-ray crystallography showed a high percentage of CaOx in the precipitate. In samples reduced to 40-69 ml, addition of dU to the collecting duct solution containing HAP seed resulted in a greater mean (SD) number of crystals; 3895 (1841) in samples with dU and 1785 (583) in samples without. This was mainly explained by an increased mean (SD) number of small crystals. The mean crystal volume was 17.8 (1.1) and 34.3 (9.1) in samples reduced to 40 69 ml with and without dU, respectively. This might reflect the inhibitory effect of dU on the growth and/or aggregation of the CaOx-CaP precipitate or a promoted nucleation resulting in a large number of small crystals. It is concluded that calcium phosphate formed above the collecting duct might induce heterogeneous nucleation of CaOx at lower levels of the renal collecting system, and that urinary macromolecules are powerful modifiers of these processes.

Calcium Oxalate↗

Factors influencing the course of calcium oxalate stone disease.

OBJECTIVE: To assess the influence of previous stone formation, urine and stone composition on the further course of the disease in recurrent calcium stone formers without pharmacological treatment. METHOD: The course of the disease was analysed during a prospective follow-up period by means of Kaplan-Meier estimates. At the start of follow-up the patients were subgrouped with regard to their previous history of stone formation expressed as stone age index (SAI = 100 x number of stones/age), urine composition, stone composition, and sex. In 223 of the patients was it possible to calculate AP(CaOx) index(s), a standardized estimate of the ion-activity product of calcium oxalate. RESULTS: The 446 patients (329 men, 117 women) who were considered representative of an average population of recurrent calcium stone formers, had a 5-year recurrence risk of approximately 50%. Patients with an SAI <2 had a lower recurrence risk than those with an SAI >2 and a corresponding difference was recorded between patients with SAI levels <5 and >5. Furthermore, female patients had a lower risk of new stone formation than male patients. Patients with an AP(CaOx) index(s) of 1.5 or more had a significantly higher recurrence risk than those with a lower index, a difference that was most pronounced in female stone formers. A slightly higher risk of recurrent stone formation during the follow-up period could also be related to the presence of calcium phosphate in the stone, a high AP(CaP) index(s) (a standardized estimate of the ion-activity product of calcium phosphate) and a low concentration of citrate. CONCLUSION: AP(CaOx) index(s) and SAI were the most obvious predictors of the recurrence risk and these two variables, together with information on the sex distribution, might be useful for deriving an expected recurrence risk at a defined point of time in a group of recurrent stone formers. Such an estimate can be valuable for conclusions on the efficacy of different stone-preventive treatments when an appropriate control group is lacking.

Actuarial Analysis↗

Minimally invasive treatment of infection staghorn stones with shock wave lithotripsy and chemolysis.

We report the results in 118 patients with infection staghorn stones treated with an anaesthesia-free minimally invasive method that combined repeated shock-wave lithotripsy (SWL) sessions (unmodified Dornier HM3 lithotripter) and percutaneous chemolysis with Renacidin. The stone-free rate was 60%. In 27 consecutive patients with infection staghorn stones representative of patients with this stone type in the population, a stone-free rate of 77% was recorded. The latter figure is comparable with results reported for open surgery, percutaneous surgery and sandwich therapy, and superior to that recorded with SWL alone. During the study period, no patient referred to us with an infection staghorn stone was treated with percutaneous, ureteroscopic or open surgery, and all treatments were carried out without regional or general anaesthesia. The described treatment concept had a very low complication rate, but required a fairly long hospital stay, with a mean of 32 days (range: 5-82). The long period necessary for completing the treatment in the most complicated cases might render the procedure less attractive as a standard method, but it is nevertheless an excellent option in high-risk patients and in all those patients in whom other procedures are impossible.

Citrates↗

The effects of citrate and urinary macromolecules on the aggregation of hydroxyapatite crystals in solutions with a composition similar to that in the distal tubule.

The effects of citrate and dialysed urine (dU) on the aggregation of hydroxyapatite (HAP) crystals in solutions with different pH and otherwise with an ion composition assumed to correspond to that in the distal part of the distal tubule were studied by spectrophotometric assessment of the rate of crystal sedimentation. When the concentration of dU was increased from 1% to 20% we recorded an increased inhibition of HAP crystal aggregation at pH 6.5. There were no differences in the inhibition accomplished by 10% dU when the pH was varied between 5.5 and 7.0, but a lower inhibition was recorded at pH 7.5. Citrate in the range 0.05-4 mmol/l had a concentration-dependent inhibitory effect on HAP crystal aggregation. In the presence of 10% dU the net inhibitory effect of citrate was reduced at all pH levels. In the pH interval 5.5-7.0 a higher inhibition was recorded with 0.5 mmol/l citrate than with 10% dU, but in the presence of dU there was only a minor additional effect of citrate at concentrations below 0.5 mmol/l. These findings indicate that urinary macromolecules present in dU strongly inhibit HAP crystal aggregation under solution conditions corresponding to those in distal tubular urine. At the same nephron level citrate might have a direct inhibitory influence on the aggregation of HAP crystals, but in the presence of normal urinary macromolecules the additive inhibitory effect of citrate is probably only marginal.

Citric Acid↗

The influence of hydroxyapatite seed on the crystallization induced by volume reduction of solutions with an ion composition corresponding to that in the distal tubule at different pH levels.

OBJECTIVE: To simulate the crystallization process that might occur in the nephron segment from the distal part of the distal tubule (DTd) to the end of the collecting duct (CD), we evaporated salt solutions with an ion composition assumed to correspond to that in the DTd. MATERIAL AND METHODS: 100-mL samples of DTd solutions with and without dialysed urine (dU) and with hydroxyapatite (HAP) seed crystals were evaporated to a final volume of 10-20 mL at different starting pH levels. After evaporation, the number and volume of crystals were recorded in a Coulter Multisizer and the crystal morphology examined by scanning electron microscopy and x-ray crystallography. RESULTS AND CONCLUSIONS: Volume reduction of solutions with a pH of 6.5 resulted in a crystallization of calcium phosphate (CaP) and calcium oxalate (CaOx). An increased fraction of CaOx in the precipitate was observed when the pH was decreased. In solutions with an initial pH of 5.5, 6.0 and 6.5, the number and volume of crystals were smaller in samples analysed after 60 min than in those analysed immediately after the evaporation. This might reflect dissolution of crystals, as expected, most pronounced in the most acid solutions, but apparently counteracted by dU. The results obtained in these experiments suggest that CaP formed high in the nephron might induce heterogeneous nucleation of CaOx when subjected to the lower pH levels and increased supersaturation with CaOx encountered in the CD. With an alkaline pH in the CD, a pronounced crystallization of CaP can be anticipated.

Calcium Oxalate↗