A systematic review of Sandifer syndrome in children with severe gastroesophageal reflux
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In brief
In brief
This systematic review of 56 Sandifer syndrome cases demonstrates that all patients achieved symptom improvement with appropriate GER treatment. Patients with anatomical anomalies (hiatal hernia, malrotation) more frequently required surgical fundoplication, while 79% without anatomical anomalies improved with medical management alone within one month.
- Sandifer syndrome combines gastroesophageal reflux with abnormal posturing, often mimicking neurological disorders and delaying diagnosis.
- All 56 reviewed cases improved with appropriate GER treatment; 79% without anatomical anomalies resolved with medical management alone.
- Patients with anatomical anomalies (hiatal hernia, malrotation) significantly more likely to require surgical fundoplication (p<0.001).
- Median time to symptom resolution with conservative treatment is 1 month; consider surgery if no improvement after this period.
- Primary care providers should include Sandifer syndrome in differential for abnormal posturing when neuromuscular causes are excluded.
Written by the GCMD Library team from the article.
Abstract
Purpose
Sandifer syndrome (SS), which combines gastroesophageal reflux (GER) and a neurological or psychiatric disorder, is an uncommon condition that often takes a long time to diagnosis. We aimed to systematically review available papers regarding SS.
Methods
After presenting our two cases of SS, we systematically reviewed articles published in MEDILINE/PubMed, Cochrane Library, and Web of Science.
Results
The meta-analysis included 54 reported cases and 2 of our own cases. Our results showed that all cases achieved symptom improvement with appropriate treatment for GER. Notably, 19 of the 56 cases exhibited anatomical anomalies, such as hiatal hernia and malrotation. Significantly more patients with than without anatomical anomalies required surgery (p < 0.001). However, 23 of the 29 patients without anatomical anomalies (79%) achieved symptom improvement without surgery. Patients who did not undergo surgery had a median (interquartile range) duration to symptom resolution of 1 (1–1) month.
Conclusion
The primary care providers should keep SS in the differential diagnosis of patients presenting with abnormal posturing and no apparent neuromuscular disorders. Fundoplication may be effective especially for patients with anatomical anomalies or those whose symptoms do not improve after more than 1 month with nonsurgical treatment.