Literature DB >> 33807568

Simultaneous Homozygous Mutations in SLC12A3 and CLCNKB in an Inbred Chinese Pedigree.

Lijun Mou1, Fengfen Wu2.   

Abstract

Gitelman syndrome (GS) and Bartter syndrome (BS) type III are both rare, recessively inherited salt-losing tubulopathies caused by SLC12A3 and CLCNKB mutations, respectively. We described a 48-year-old male patient with fatigue, carpopedal spasm, arthralgia, hypokalemic alkalosis, mild renal dysfunction, hypomagnesemia, hypocalciuria, hyperuricemia, normotension, hyperreninemia and chondrocalcinosis in knees and Achilles tendons. His parents are first cousin. Genetic analysis revealed simultaneous homozygous mutations in SLC12A3 gene with c.248G>A, p.Arg83Gln and CLCNKB gene with c.1171T>C, p.Trp391Arg. The second younger brother of the proband harbored the same simultaneous mutations in SLC12A3 and CLCNKB and exhibited similar clinical features except normomagnesemia and bilateral kidney stones. The first younger brother of the proband harbored the same homozygous mutations in CLCNKB and exhibited clinical features of hypokalemia, normomagnesemia, hypercalciuria and hyperuricemia. Potassium chloride, spironolactone and potassium magnesium aspartate were prescribed to the proband to correct electrolytic disturbances. Benzbromarone and febuxostat were prescribed to correct hyperuricemia. The dose of potassium magnesium aspartate was subsequently increased to alleviate arthralgia resulting from calcium pyrophosphate deposition disease (CPPD). To the best of our knowledge, we are the first to report an exceptionally rare case in an inbred Chinese pedigree with simultaneous homozygous mutations in SLC12A3 and CLCNKB. GS and BS type III have significant intrafamilial phenotype heterogeneity. When arthralgia is developed in patients with GS and BS, gout and CPPD should both be considered.

Entities:  

Keywords:  Bartter syndrome; CLCNKB; Gitelman syndrome; SLC12A3; calcium pyrophosphate deposition disease; gout

Mesh:

Substances:

Year:  2021        PMID: 33807568      PMCID: PMC7999423          DOI: 10.3390/genes12030369

Source DB:  PubMed          Journal:  Genes (Basel)        ISSN: 2073-4425            Impact factor:   4.096


  17 in total

Review 1.  Miscellaneous non-inflammatory musculoskeletal conditions. Bartter's and Gitelman's diseases.

Authors:  Marta Favero; Lorenzo A Calò; Franco Schiavon; Leonardo Punzi
Journal:  Best Pract Res Clin Rheumatol       Date:  2011-10       Impact factor: 4.098

2.  Gout as a complication of Bartter's syndrome. A possible role for alkalosis in the decreased clearance of uric acid.

Authors:  W J Meyer; J R Gill; F C Bartter
Journal:  Ann Intern Med       Date:  1975-07       Impact factor: 25.391

3.  Simultaneous mutations in the CLCNKB and SLC12A3 genes in two siblings with phenotypic heterogeneity in classic Bartter syndrome.

Authors:  Alberto Bettinelli; Nicolò Borsa; Marie-Louise Syrén; Camilla Mattiello; Domenico Coviello; Alberto Edefonti; Marisa Giani; Maurizio Travi; Silvana Tedeschi
Journal:  Pediatr Res       Date:  2005-12       Impact factor: 3.756

4.  A novel mutation in the chloride channel gene, CLCNKB, as a cause of Gitelman and Bartter syndromes.

Authors:  Israel Zelikovic; Raymonde Szargel; Ali Hawash; Valentina Labay; Ihab Hatib; Nadine Cohen; Farid Nakhoul
Journal:  Kidney Int       Date:  2003-01       Impact factor: 10.612

5.  Gitelman syndrome: consensus and guidance from a Kidney Disease: Improving Global Outcomes (KDIGO) Controversies Conference.

Authors:  Anne Blanchard; Detlef Bockenhauer; Davide Bolignano; Lorenzo A Calò; Etienne Cosyns; Olivier Devuyst; David H Ellison; Fiona E Karet Frankl; Nine V A M Knoers; Martin Konrad; Shih-Hua Lin; Rosa Vargas-Poussou
Journal:  Kidney Int       Date:  2017-01       Impact factor: 10.612

6.  Mutations in the chloride channel gene CLCNKB as a cause of classic Bartter syndrome.

Authors:  Martin Konrad; Martin Vollmer; Henny H Lemmink; Lambertus P W J VAN DEN Heuvel; Nikola Jeck; Rosa Vargas-Poussou; Alicia Lakings; Rainer Ruf; Georges Deschênes; Corinne Antignac; Lisa Guay-Woodford; Nine V A M Knoers; Hannsjörg W Seyberth; Delphine Feldmann; Friedhelm Hildebrandt
Journal:  J Am Soc Nephrol       Date:  2000-08       Impact factor: 10.121

7.  The pharmacological characteristics of molecular-based inherited salt-losing tubulopathies.

Authors:  Kandai Nozu; Kazumoto Iijima; Kyoko Kanda; Koichi Nakanishi; Norishige Yoshikawa; Kenichi Satomura; Hiroshi Kaito; Yuya Hashimura; Takeshi Ninchoji; Hiroshi Komatsu; Koichi Kamei; Ritsuko Miyashita; Masaaki Kugo; Hiroshi Ohashi; Hajime Yamazaki; Hiroyo Mabe; Asa Otsubo; Takashi Igarashi; Masafumi Matsuo
Journal:  J Clin Endocrinol Metab       Date:  2010-09-01       Impact factor: 5.958

8.  Human CLC-KB gene promoter drives the EGFP expression in the specific distal nephron segments and inner ear.

Authors:  Katsuki Kobayashi; Shinichi Uchida; Hiro-Oki Okamura; Fumiaki Marumo; Sei Sasaki
Journal:  J Am Soc Nephrol       Date:  2002-08       Impact factor: 10.121

9.  The ClC-K2 Chloride Channel Is Critical for Salt Handling in the Distal Nephron.

Authors:  J Christopher Hennings; Olga Andrini; Nicolas Picard; Marc Paulais; Antje K Huebner; Irma Karen Lopez Cayuqueo; Yohan Bignon; Mathilde Keck; Nicolas Cornière; David Böhm; Thomas J Jentsch; Régine Chambrey; Jacques Teulon; Christian A Hübner; Dominique Eladari
Journal:  J Am Soc Nephrol       Date:  2016-06-22       Impact factor: 10.121

10.  Analysis of protein-coding genetic variation in 60,706 humans.

Authors:  Monkol Lek; Konrad J Karczewski; Eric V Minikel; Kaitlin E Samocha; Eric Banks; Timothy Fennell; Anne H O'Donnell-Luria; James S Ware; Andrew J Hill; Beryl B Cummings; Taru Tukiainen; Daniel P Birnbaum; Jack A Kosmicki; Laramie E Duncan; Karol Estrada; Fengmei Zhao; James Zou; Emma Pierce-Hoffman; Joanne Berghout; David N Cooper; Nicole Deflaux; Mark DePristo; Ron Do; Jason Flannick; Menachem Fromer; Laura Gauthier; Jackie Goldstein; Namrata Gupta; Daniel Howrigan; Adam Kiezun; Mitja I Kurki; Ami Levy Moonshine; Pradeep Natarajan; Lorena Orozco; Gina M Peloso; Ryan Poplin; Manuel A Rivas; Valentin Ruano-Rubio; Samuel A Rose; Douglas M Ruderfer; Khalid Shakir; Peter D Stenson; Christine Stevens; Brett P Thomas; Grace Tiao; Maria T Tusie-Luna; Ben Weisburd; Hong-Hee Won; Dongmei Yu; David M Altshuler; Diego Ardissino; Michael Boehnke; John Danesh; Stacey Donnelly; Roberto Elosua; Jose C Florez; Stacey B Gabriel; Gad Getz; Stephen J Glatt; Christina M Hultman; Sekar Kathiresan; Markku Laakso; Steven McCarroll; Mark I McCarthy; Dermot McGovern; Ruth McPherson; Benjamin M Neale; Aarno Palotie; Shaun M Purcell; Danish Saleheen; Jeremiah M Scharf; Pamela Sklar; Patrick F Sullivan; Jaakko Tuomilehto; Ming T Tsuang; Hugh C Watkins; James G Wilson; Mark J Daly; Daniel G MacArthur
Journal:  Nature       Date:  2016-08-18       Impact factor: 49.962

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  1 in total

1.  A case of Gitelman syndrome with membranous nephropathy.

Authors:  Xiafei Guo; Shanshen Yu; Jia Sun; Lijun Mou
Journal:  BMC Nephrol       Date:  2022-07-26       Impact factor: 2.585

  1 in total

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