09 December 2023: Articles
Long-Term Indomethacin Treatment in a Chinese Child with Gitelman Syndrome: Case Report and Literature Review on its Efficacy and Tolerance
Challenging differential diagnosis, Unusual or unexpected effect of treatment, Rare disease
Xiaoyan Peng A , Chaoying Chen A* , Juan Tu D , Yuan Lin B , Huarong Li B , Haiyun Geng BDOI: 10.12659/AJCR.941627
Am J Case Rep 2023; 24:e941627
Table 2. Literature review of applications for Gitelman syndrome patients.
| Year | Nation | Type | Main points | Author | Ref. |
|---|---|---|---|---|---|
| 2019 | China | Retrospective study (8/16 pts used indomethacin) | Treatment with triamterene or indomethacin(50–100 mg/d) significantly increased serum potassium concentration. Two patients (25%) stopped the treatment due to intolerance (one for abdominal discomfort and another for sleepiness) | Cui Y | []8 |
| 2015 | French | Open-label, randomized, crossover study (30 pts) | Indomethacin (75 mg/d for 6 weeks) was the most effective but can cause gastrointestinal (GI) intolerance and decreased eGFR. Indomethacin caused GI intolerance (heartburn, dyspepsia, or gastric irritation) in 6 patients (20%), resulting in early discontinuation | Blanchard, A | []5 |
| 2015 | China | Case report (a 48-year woman with proteinuria) | Indomethacin (150 mg/d) treatment significantly increased serum potassium concentration and no adverse effects were mentioned | Zeng D | []9 |
| 2015 | China | Cases (Two sisters, 42 and 37 years old) | Two sisters had inappetence and pain after indomethacin (25 mg/d) treated for 1–2 weeks. Their symptoms were relieved after stopping indomethacin | Ji W | []10 |
| 2014 | Caucasian | Cases (two sisters) | Both children were treated with indomethacin (2–3.8 mg/kg/d) resulting in improved linear growth and polyuria. They were treated for 5–7 years but no adverse effects were reported | Larkins N | []3 |
| 2013 | China | Retrospective study (4/11 used indomethacin) | The symptoms of GS patients were relieved by supplementation with potassium alone or in combination with indomethacin (25–150 mg/d). Self-discontinuation was common in follow-up | Yang C | []11 |
| 2012 | China | Retrospective study (4/17 pts used indomethacin) | Indomethacin (75 mg/d), spironolactone, and other potassium-magnesium asparaginase helped to relieve the symptoms. No adverse effects or long-term outcomes were mentioned | Qu L | []12 |
| 2011 | China | Retrospective study (1/4 pts use indomethacin) | All symptoms resolved after treatment with potassium or combined magnesium supplementation, indomethacin (1.5–2.5 mg/kg), spironolactone, and captopril. Hypokalemia was corrected but hypomagnesemia was not | Fan S | []13 |
| 2010 | Turkey | Case report (mental retardation) | Indomethacin and triamterene were administrated and helped the plasma levels of magnesium and potassium normalization | Tuhta GA | []14 |
| 2006 | China | Retrospective study (6/9 pts used indomethacin) | Treatment combination with indomethacin (75–150 mg/d) helped to improve hypokalemia. Serum magnesium levels were still lower than normal range | Yang GQ | []15 |
| 2005 | China | Case report (a 63-year-old woman) | GS may be present with severe hypocalcemia and hypokalemic periodic paralysis; the combined use of indomethacin (50 mg tid) with triamterene has good therapeutic effect | Ran XW | []16 |
| 2003 | China | Case report (39-year-old man) | Indomethacin (25 mg tid) treatment significantly increased serum potassium concentration | Tao H | []17 |
| 2002 | Israel | Case report | Rofecoxib instead of indomethacin promptly elevated serum potassium concentration with normalization of plasma aldosterone and near normalization of renin. Rhabdomyolysis was also ameliorated by COX 2 inhibition | Mayan H | []18 |
| 2001 | Germany | Cases report| (3/5 children used indomethacin) | Indomethacin (1–2 mg/kg/d) failed to fully correct hypokalemia and hypomagnesemia, but markedly improved growth velocity and normalized IGF-1 levels in the 3 patients with short stature. For the longest, indomethacin was treated for 7 years. The adverse effects were not mentioned | Schmidt H | []19 |
| 2001 | Japan | Case report (20-year-old man) | Treated with potassium, spironolactone, and indomethacin for over 9 years, no abnormal signs. The adverse effects were not mentioned | Tsuchiya H | []20 |
| 1999 | Europe | Cases report (3 sisters) | Hypotension and polyuria were eliminated by taking 2 mg/kg/day indomethacin. Increasing the indomethacin dose to 4 mg/kg/day improved their growth significantly, without changing their symptoms or biochemistry. Gastrointestinal hemorrhage was reported in the oldest sister while on high-dose indomethacin (5 mg/kg/d) | Liaw LC | []4 |
| pts – patients; * paper in Chinese. | |||||