Six-year follow-up of a survivor of cervical spine fracture and dislocation with oesophageal perforation following long scarf syndrome - a case report and literature review

BACKGROUND: Accidental strangulation due to scarf getting caught in the wheels of a vehicle or machine was called “Isadora Duncan Syndrome” or “Long Scarf Syndrome”. Survival of concomitant fracture dislocation of cervical spine and oesophageal perforation following Long Scarf Syndrome was rarely described and medium-term follow-up for this lesion has not been reported. CASE PRESENTATION: We present a 39-year-old female who suffered accidental strangulation caused by the scarf around her neck getting trapped in the wheels of the a vehicle and was referred to our hospital forty days post injury. The CT examination showed a fracture dislocation at C5/6 levels with complete dissociation of the supporting structures. She developed paravertebral abscesses, cutaneous fistulas and oesophageal perforation confirmed by oesophagoscope. The patient was treated conservatively because of poor general condition and inappropriate initial treatment. Halo-vest was used to immobilize the cervical spine. The oesophagus-cutaneous fistula was managed with enteral tube feeding and repeated local care. The patient survived despite such severe injury. Nine months after the injury, the oesophageal perforation closed spontaneously and fixed malunion of the cervical spine was achieved. Six-year follow-up demonstrated that the patient survived with complete C5 tetraplegia. Literature associated with this lesion was reviewed and factors contributing to the survival were discussed. CONCLUSIONS: Concomitant fracture dislocation of cervical spine and oesophageal perforation following Long Scarf Syndrome is extremely rare with high risk of mortality. Though surgical intervention is always necessary, the optimal management for this kind of lesion should be made on an individual basis through a multidisciplinary approach.