Published online Dec 28, 2016. doi: 10.4254/wjh.v8.i36.1637
Peer-review started: August 23, 2016
First decision: September 28, 2016
Revised: October 14, 2016
Accepted: November 1, 2016
Article in press: November 2, 2016
Published online: December 28, 2016
To describe one case of bilateral Tapia’s syndrome in a liver transplanted patient and to review the literature.
We report a case of bilateral Tapia’s syndrome in a 50-year-old man with a history of human immunodeficiency virus and hepatitis C virus child. A liver cirrhosis and a bi-nodular hepatocellular carcinoma, who underwent liver transplantation after general anesthesia under orotracheal intubation. Uneventful extubation was performed in the intensive care unit during the following hours. On postoperative day (POD) 3, he required urgent re-laparotomy due to perihepatic hematoma complicated with respiratory gram negative bacilli infection. On POD 13, patient was extubated, but required immediate re-intubation due to severe respiratory failure. At the following day a third weaning failure occurred, requiring the performance of a percutaneous tracheostomy. Five days later, the patient was taken off mechanical ventilation and severe dysphagia, sialorrea and aphonia revealed. A computerized tomography and a magnetic resonance imaging of the head and neck excluded central nervous injury. A stroboscopy showed bilateral paralysis of vocal cords and tongue and a diagnosis of bilateral Tapia’s syndrome was performed. With conservative management, including a prompt establishment of a speech and swallowing rehabilitation program, the patient achieved full recovery within four months after liver transplantation. We carried out MEDLINE search for the term Tapia’s syndrome. The inclusion criteria had no restriction by language or year but must provide sufficient available data to exclude duplicity. We described the clinical evolution of the patients, focusing on author, year of publication, age, sex, preceding problem, history of endotracheal intubation, unilateral or bilateral presentation, diagnostic procedures, type of treatment, follow-up, and outcome.
Several authors mentioned the existence of around 70 cases, however only 54 fulfilled our inclusion criteria. We found only five published studies of bilateral Tapia’s syndrome. However this is the first case reported in the literature in a liver transplanted patient. Most patients were male and young and the majority of cases appeared as a complication of airway manipulation after any type of surgery, closely related to the positioning of the head during the procedure. The diagnosis was founded on a rapid suspicion, a complete head and neck neurological examination and a computed tomography and or a magnetic resonance imaging of the brain and neck to establish the origin of central or peripheral type of Tapia’s syndrome and also the nature of the lesion, ischemia, abscess formation, tumor or hemorrhage. Apart from corticosteroids and anti- inflammatory therapy, the key of the treatment was an intensive and multidisciplinary speech and swallowing rehabilitation. Most studies have emphasized that the recovery is usually completed within four to six months.
Tapia’s syndrome is almost always a transient complication after airway manipulation. Although bilateral Tapia’s syndrome after general anesthesia is exceptionally rare, this complication should be recognized in patients reporting respiratory obstruction with complete dysphagia and dysarthria after prolonged intubation. Both anesthesiologists and surgeons should be aware of the importance of its preventing measurements, prompt diagnosis and intensive speech and swallowing rehabilitation program.
Core tip: Tapia’s syndrome is a rare entity characterized by the concomitant extracranial injury of the hypoglossal nerve (XII) and the recurrent laryngeal branch of the vagus nerve (X) at the base of the tongue and the pyriform fossa. Anesthesiologists, surgeons and otorhinolaryngologist should be aware of its presentation at any type of surgery as in the present case, after liver transplantation. The purpose of this study is to present our even rarer presentation of bilateral Tapia’s syndrome to the liver transplant community and to review the literature to update the current management and treatment. The most relevant common feature in most cases of bilateral syndrome was orotracheal intubation prolonged for more than 14 d.