Document Detail


Long-term follow-up of patients with Bartter syndrome type I and II.
MedLine Citation:
PMID:  20219833     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: Little information is available on a long-term follow-up in Bartter syndrome type I and II.
METHODS: Clinical presentation, treatment and long-term follow-up (5.0-21, median 11 years) were evaluated in 15 Italian patients with homozygous (n = 7) or compound heterozygous (n = 8) mutations in the SLC12A1 (n = 10) or KCNJ1 (n = 5) genes.
RESULTS: Thirteen new mutations were identified. The 15 children were born pre-term with a normal for gestational age body weight. Medical treatment at the last follow-up control included supplementation with potassium in 13, non-steroidal anti-inflammatory agents in 12 and gastroprotective drugs in five patients. At last follow-up, body weight and height were within normal ranges in the patients. Glomerular filtration rate was <90 mL/min/1.73 m(2) in four patients (one of them with a pathologically increased urinary protein excretion). In three patients, abdominal ultrasound detected gallstones. The group of patients with antenatal Bartter syndrome had a lower renin ratio (P < 0.05) and a higher standard deviation score (SDS) for height (P < 0.05) than a previously studied group of patients with classical Bartter syndrome.
CONCLUSIONS: Patients with Bartter syndrome type I and II tend to present a satisfactory prognosis after a median follow-up of more than 10 years. Gallstones might represent a new complication of antenatal Bartter syndrome.
Authors:
Elena Puricelli; Alberto Bettinelli; Nicolò Borsa; Francesca Sironi; Camilla Mattiello; Fabiana Tammaro; Silvana Tedeschi; Mario G Bianchetti;
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Publication Detail:
Type:  Journal Article; Research Support, Non-U.S. Gov't     Date:  2010-03-10
Journal Detail:
Title:  Nephrology, dialysis, transplantation : official publication of the European Dialysis and Transplant Association - European Renal Association     Volume:  25     ISSN:  1460-2385     ISO Abbreviation:  Nephrol. Dial. Transplant.     Publication Date:  2010 Sep 
Date Detail:
Created Date:  2010-08-25     Completed Date:  2010-12-23     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8706402     Medline TA:  Nephrol Dial Transplant     Country:  England    
Other Details:
Languages:  eng     Pagination:  2976-81     Citation Subset:  IM    
Affiliation:
Department of Pediatrics, San Leopoldo Mandic Hospital, Largo Mandic 1, Merate, Lecco, Italy.
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MeSH Terms
Descriptor/Qualifier:
Bartter Syndrome / classification,  drug therapy,  genetics*
Body Height
Body Weight
Child, Preschool
Female
Follow-Up Studies
Glomerular Filtration Rate
Heterozygote
Homozygote
Humans
Infant
Infant, Newborn
Male
Mutation / genetics*
Potassium Channels, Inwardly Rectifying / genetics*
Prognosis
Sodium-Potassium-Chloride Symporters / genetics*
Time Factors
Chemical
Reg. No./Substance:
0/KCNJ1 protein, human; 0/Potassium Channels, Inwardly Rectifying; 0/Sodium-Potassium-Chloride Symporters; 0/sodium-potassium chloride cotransporter 2 protein
Investigator
Investigator/Affiliation:
Silvio Maringhini / ; Paolo Porcelli / ; Marco Materassi / ; Maria Renata Proverbio / ; Nunzia Miglietti / ; Maria Gabriella Porcellini / ; Carla Navone / ; Giuseppe Ruffa / ; Aldo Rosini / ; Aurora Rossodivita /

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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