A systematic review of Sandifer syndrome in children with severe gastroesophageal reflux

Pediatr Surg Int. 2024 Mar 25;40(1):91. doi: 10.1007/s00383-024-05683-3.

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.

Keywords: Fundoplication; Gastroesophageal reflux; Laparoscopic surgery; Sandifer syndrome.

Publication types

  • Systematic Review
  • Meta-Analysis

MeSH terms

  • Child
  • Fundoplication / methods
  • Gastroesophageal Reflux* / complications
  • Gastroesophageal Reflux* / diagnosis
  • Gastroesophageal Reflux* / surgery
  • Hernia, Hiatal* / complications
  • Hernia, Hiatal* / diagnosis
  • Hernia, Hiatal* / surgery
  • Humans
  • Laparoscopy* / methods
  • Torticollis* / diagnosis
  • Torticollis* / surgery
  • Treatment Outcome

Supplementary concepts

  • Sandifer syndrome