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J R Soc Med. 2004 February; 97(2): 99.
PMCID: PMC1079311

Atypical manifestations of gastrooesophageal reflux

The review by Mr Issing and Mr Karkos is extensive but not exhaustive (October 2003 JRSM1). Mention should also be made of Sandifer's syndrome.2 In this condition gastrooesophageal reflux (GOR) triggers dystonic posturing, primarily within the axial musculature, to produce torticollis, retrocollis and even opisthotonus. The condition occurs in children, particularly when there is coexistent developmental delay. The mechanism of Sandifer's syndrome is uncertain. In one case it was found that, when a 5-year-old girl with Sandifer's syndrome simulated her dystonic posturing during oesophageal manometry, oesophageal peristalsis increased in both amplitude and velocity.3 This did not occur in two other children with GOR but without dystonia, or in normal adult controls. Interestingly, similar dystonic posturing has been seen in a 2-year-old child without developmental delay after chronic oesophageal perforation by a foreign body. Earlier workers suggested that the process is reflex, noting that the upper cervical myotomes responsible for movements of the head and neck overlap segments thought to receive sensory afferents from the diaphragm. The observed absence of dystonic posturing during sleep in individuals with Sandifer's syndrome is difficult to reconcile with this idea.

Sandifer's syndrome has been considered rare, but its true prevalence is uncertain. It can be mistaken for a behavioural or seizural disorder but responds well to treatment of GOR by medical or surgical means.

References

1. Issing WJ, Karkos PD. Atypical manifestations of gastro-oesophageal reflux. J R Soc Med 2003;96: 477-80 [PMC free article] [PubMed]
2. Kinsbourne M. Hiatus hernia with contortions of the neck. Lancet 1964;i: 1058-61 [PubMed]
3. Puntis JWL, Smith HL, Buick RG, Booth IW. Effect of dystonic movements on oesophageal peristalsis in Sandifer's syndrome. Arch Dis Child 1989;64: 1311-13 [PMC free article] [PubMed]

Articles from Journal of the Royal Society of Medicine are provided here courtesy of Royal Society of Medicine Press