Extremely Late Recovery of Unusual Cervical Ankylosing Spondylitis-Related Dysphagia: Anesthesiologic, Surgical, and Pathophysiological Considerations, and Review of the Literature

BACKGROUND: Ankylosing spondylitis (AS) is an inflammatory disease that involves above all the spine and the pelvis. In the spine, the inflammatory processes cause the formation of syndesmophytes between the vertebral bodies and the ossification of ligaments, with bony overgrowth. In this setting, dysphagia is a rare but severe complication and only a few cases have been reported in the literature.CASE DESCRIPTION: We describe the case of a 50-year-old man suffering from AS, with a 6-month history of severe dysphagia caused by bone compression of the esophagus at the C3-5 level. Because the patient underwent a 10-kg weight loss 2 months after clinical onset, a gastrostomy tube placement was needed. Complete surgical excision of the bone overgrowth via an anterior cervical approach was performed, but despite continuous intensive swallowing rehabilitation therapy, the patient failed to improve in the first following months. Surprisingly, the patient started to improve 18 months after the operation, with a complete recovery from dysphagia 24 months after.CONCLUSIONS: Among the 5 reports (including the present case) available in the current literature with a clear dysphagia recovery follow-up, the present case (the second one harboring gastrostomy) is associated with the slowest complete recovery published so far. This unusually late recovery suggests a possible role not only of the mechanical decompression of the esophagus but also of the degeneration/regeneration ratio of the myenteric plexus, along with local neurotransmitters sensitivity changes, to better understand the dysphagia recovery time course of this unique patient.