Bilateral recurrent laryngeal nerve and unilateral hypoglossal nerve paralysis (Tapia’s Syndrome) secondary to intubation in Burns ITU.

Authors: Daniel Craig Hathaway, Nader Ibrahim, Sarah Hemington-Gorse, Jonathan Cubitt.

Welsh Centre for Burns and Plastic Surgery, Morriston Hospital, Swansea, UK.

Objectives
Tapia’s syndrome was first described by Spanish otolaryngologist Antonio Garcia Tapia and is reported as an ipsilateral larynx and tongue hemiplegia with the cause originating extracranially. (1, 2) This eponymous syndrome has since been applied to other derivatives of similar pathology. The proposed mechanism of injury to these nerves are as a result of tracheal intubation, abnormal head/neck positioning and high cuff pressures.(3, 4) 

Methods
We report a case of a 16-year-old male with 42% total body surface area mixed-depth burns to his legs, abdomen, back and hands following an explosion. He was intubated on presentation to A&E with minimal tracheal soiling not requiring lavage. He underwent a total of 10 theatre sessions during his ITU admission. 

Whilst intubated, he received treatment for a ventilator-acquired pneumonia and a malodourous tonsillar exudate grew a positive culture for fusobacterium necrophorum. After two failed extubations, he was successfully extubated on day 19. He was found to be aphonic with minimal laryngeal elevation, no anterior hyoid movement and a weak cough causing a dysfunctional swallow. Laryngoscopy identified sluggish vocal cords bilaterally. On day 32, a left hypoglossal palsy was also noted.

Neuroradiological imaging of his cervical spine and skull base showed no structural cause for the patient’s symptoms which resulted in a diagnosis of Tapia’s syndrome.

Results
There has been some improvement in hyolaryngeal elevation although the patient is still heavily reliant on Yankauer suctioning to manage his secretions. Tongue denervation persists and His voice remains aphonic and airway protection remains severely compromised requiring NG feeding.

Conclusion
Six weeks after extubation, there has been limited spontaneous recovery in the patient’s symptoms. The patient underwent multiple intubations and visits to theatre which may have contributed to his presentation with Tapia’s syndrome. Compared with other reported cases of Tapia’s syndrome, our patient should expect to have resolution of his symptoms within 3-6 months.(5, 6)
The growth of fusobacterium necrophorum which can rarely cause Lemierre’s syndrome (and subsequent CN IX, X, XII palsy) may be coincidental. 

1.	Tapia AG. Un caso de paralisis del lado derecho de la laringe y de ungue, con paralisis del externo-cleidomastoidea y trapecio del mismo lado; accompañado de hemiplejia total temporal de lado izquierdo del cuerpo. . El siglo médico. 1905;52:211-3.
2.	Schoenberg BS, Massey EW. Tapia's syndrome. The erratic evolution of an eponym. Arch Neurol. 1979;36(5):257-60.
3.	Yavuzer R, Basterzi Y, Ozkose Z, Yucel Demir H, Yilmaz M, Ceylan A. Tapia's syndrome following septorhinoplasty. Aesthetic Plast Surg. 2004;28(4):208-11.
4.	Gelmers HJ. Tapia's syndrome after thoracotomy. Arch Otolaryngol. 1983;109(9):622-3.
5.	Tan R, Sieunarine K. Case Report: Tapia’s Syndrome after Prolonged and Repeated Intubations. Anaesthesia and Surgery. 2018.
6.	Boisseau N, Rabarijaona H, Grimaud D, Raucoules-Aime M. Tapia's syndrome following shoulder surgery. Br J Anaesth. 2002;88(6):869-70.

3 - Bilateral recurrent laryngeal nerve and unilateral hypoglossal nerve palsy (Tapia’s Syndrome) secondary to intubation in Burns Intensive Therapy Unit (ITU).