Tapia’s Syndrome After Cardiac Surgery: A Case Report and Review of Literature

Introduction

Tapia’s syndrome was coined by Spanish Otolaryngologist Antonio Garcia Tapia in 1904 and describes symptoms of hoarseness and dysphagia believed to be caused by mechanical trauma to cranial nerves (CN) X and XII. ¹ The theoretical etiology can be described as compression from intubation with flexion of the neck imposing the ramus of the mandible on the anterior branch of the recurrent laryngeal nerve and hypoglossal nerve.^2-4 In Tapia’s syndrome, neuropraxia due to stretch or crush type injury is generally treated with steroids ⁴ ; multiple studies also suggest speech language therapy (SLT) improves prognosis within 3 to 6 months. ⁵

We present a unique case of Tapia’s syndrome following aortic valve replacement (AVR) and coronary artery bypass grafting (CABG) which was treated with SLT and vocal cord injection of carboxymethylcellulose gel implant, leading to partial recovery of function. We additionally provide a review of literature and previously documented cases of Tapia’s syndrome to summarize contemporary diagnosis and management of this condition.

Case Report

A 70-year-old male with a past medical history of aortic valve disease, coronary artery disease, hyperlipidemia, and hypertension underwent AVR and CABG. He was placed in supine position, was administered general anesthesia, and was intubated orotracheally. He tolerated the procedure well, and was transferred to the surgical intensive care unit (SICU) postoperatively in stable condition.

Upon admission to the SICU, his exam was notable for a normal neurologic exam with intact cough and gag reflexes and an endotracheal tube in place. He was quickly weaned off sedation and extubed to nasal cannula oxygen. Following extubation, the patient noted hoarseness in his voice. This hoarseness continued, and on postoperative day 4, Otolaryngology was consulted.

Patient was found to have a tongue deviation to the left with protrusion (Figure 1A) and mild difficulty swallowing. Flexible laryngoscopy showed left vocal cord immobility lying in the paramedian position and a fair glottic gap with attempted adduction. Modified barium swallow was performed by SLT, which showed mild oral and moderate pharyngeal dysphagia with aspiration of thin and nectar thick liquids. A computerized tomography (CT) scan of the neck was performed and showed mild prominence of the left base of the tongue without discrete mass. Given the above symptoms and evaluation, the patient was diagnosed with Tapia’s Syndrome. Patient was discharged with follow-up of Otolaryngology and SLT.OPEN IN VIEWER

At a 3 week follow-up visit with Otolaryngology (postoperative day 29), the patient reported continued symptoms. Decision was made for vocal cord injection with carboxymethylcellulose gel implant which occurred 1 week later (on postoperative day 36). Patient was injected with 1.0 cm³ of the implant into the left vocal cord at the level of the vocal process and just anterior at the superior arcuate line (Figure 2A).OPEN IN VIEWER

Modified barium swallow was re-performed 1 week following vocal cord injection, which showed no penetration or aspiration with independent swallow. He was then cleared by SLT for a regular diet with thin liquids, and continued with outpatient swallow and voice therapy.

The patient then followed up with Otolaryngology 6 months later, at which time he reported mild gradual improvement in his voice quality. Flexible laryngoscopy was performed, and showed continued left vocal cord paralysis with the vocal cord fixed in the near median position with near complete glottic closure, with no frank aspiration of saliva or pooled saliva. At this point, the patient elected for observation moving forward. Patient was again re-examined at 9 months postoperative with tongue protrusion about 90% back to midline (Figure 1B) and slight weakness of left true cord along with mild atrophy (Figure 2B and C).

Discussion

Cases of Tapia’s syndrome have been reported most commonly in procedures that require orotracheal intubation during general anesthesia in a wide array of surgical procedures including cardiac surgery, neurological surgery, orthopedic surgery, and otolaryngologic surgery (Table 1). There have also been cases of Tapia’s syndrome unrelated to surgery.^16,24 Tapia’s syndrome is a clinical diagnosis which requires interprofessional workup with a thorough Neurology and Otolaryngology exam along with SLT. After review of literature summarized in Table 1, the most common presenting symptoms after extubation are hoarseness and dysphagia.

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Table 1.

Tapia’s Syndrome Literature Review of Cases.

Reference	Age (years) and Sex	Procedure	ETT or LMA	Symptom onset	Symptoms	Treatment	Recovery	Reported recovery time
Mayer et al., 1974 6	51 M	Hiatal hernia surgery and PNA	ETT	Immediate post-op	Hoarseness, dysphagia, ecchymosis on right soft palate, right vocal cord paralysis, right soft palate weakness, weakness of right side of tongue	Cricothyroid catheter	Partial	1 week
Gelmers, 1983 7	36 M	Thoracotomy for bronchiectasis	ETT	Immediate post-op	Hoarseness, hemiplegia and wasting of tongue with ipsilateral paralysis of vocal cord	-	Unchanged	12 months
Gelmers, 1983 7	41 M	CABG	ETT	Immediate post-op	Hoarseness, hemiplegia and wasting of tongue with ipsilateral paralysis of vocal cord	-	Unchanged	12 months
Boisseau et al., 2002 3	42 M	Arthroscopic surgery of glenohumeral joint	ETT	Immediate post-op	Throat and neck pain, hoarseness, dysphagia, tongue deviation to left, left vocal cord palsy, unable to lift left shoulder	Corticosteroids, vit B1/6 for 2 weeks, SLT	Complete	6 months
Yavuzer et al., 2004 8	42 F	Septorhinoplasty	ETT	Immediate post-op	Hoarseness, difficulty in tongue movement and swallowing, dysarthria, tongue deviation to left, submucosal hemorrhage of left vocal cord with paralysis	Prednisolone 60 mg/day tapered down over 10 days, vit B12 500 mcg/day and vit B6 100 mg/day for 3 months	Partial	6 months
Cinar et al., 2005 9	20 M	Rhinoplasty	ETT	Immediate post-op	Laryngospasm, inability to swallow, unable to move tongue, bilateral vocal cord paralysis	Post-op IV high dose steroid, then Prednisone 1 mg/kg/day with 2 week taper, NGT	Complete	4 weeks
Tesei et al., 2006 10	30 F	Rhinoplasty	ETT	Day 1	Hoarseness, dysphonia, dysphagia, deviation of tongue to right, right vocal cord paralysis	Steroids, vitamins, SLT	Complete	4 months
Sotiriou et al., 2007 11	52 F	CABG	ETT	Day 1	Upper respiratory airway obstruction after extubation, bilateral hypoglossal and RLN palsy	Re-intubation and tracheostomy	Complete	5 months
Kashyap et al., 2009 12	41 M	Mandibular surgery	ETT	Immediate post-op	Hoarseness, dyspnea during speech, dysphagia, left tongue deviation, left vocal cord palsy	-	Partial	16 months
Boga et al., 2010 13	35 M	Septorhinoplasty	ETT	Immediate post-op	Unilateral tongue swelling, unilateral tongue palsy, slowed and cracked speech, unilateral cord paralysis	Methylprednisolone 20 mg/day IV for 3 days, followed by oral corticosteroids 5 mg/d for 10 days	Complete	15 days
Rotondo et al., 2010 14	72 M	AVR	ETT	Day 2	Dyspnea, dysphagia, dysphonia, hoarseness, left tongue deviation, paresis of left hemilarynx, hematoma of left posterior rhinopharynx	Methylprednisolone 200 mg IV, followed by oral therapy for 10 days	Partial	3 months
Nalladaru et al., 2011 15	49 M	CABG	ETT	Few hours after extubation	Dysphonia, hoarseness, inability to swallow, tongue deviation to right, right vocal cord paralysis, paralysis of soft palate	Dexamethasone 4 mg 3 times daily IV, NGT, PEG	Complete	10 weeks
Turan et al., 2012 16	15 M	PNA and ARDS	ETT	Day 21	Hoarseness, dysphagia, left vocal cord paralysis, tongue immobility	Prednisolone 1 mg/kg/day for 2 weeks	Partial	2 weeks
Lykoudis et al., 2012 2	32 M	Rhinoplasty	ETT	Day 1	Hoarseness, ipsilateral paralysis of right tongue and vocal cord, contusion of right hypopharynx	Oral steroids, SLT	Complete	4 months
Wadelek et al., 2012 17	57 M	Arthroscopic acromioplasty of right shoulder joint	ETT	Immediate post-op	Hoarseness, dysphagia, numbness of half of tongue, slight tongue deviation to right, lowering of left palatine arch, hematoma of left posterior wall of nasopharynx, impaired mobility of right vocal fold	Symptomatic treatment	Partial	Several months
Varedi et al., 2013 18	27 M	ZMC fx repair	Nasotracheal tube	Day 1	Hoarseness, disturbed speech, trouble in tongue movement and swallowing, deviation of tongue to left, paralysis of left vocal cord	Oral prednisolone 60 mg/day w tapering for 14 days, vit B complex, SLT	Complete	9 months
Kang et al., 2013 19	47 M	C3-6 Laminectomy	ETT	Immediate post-op	Throat and neck pain, hoarseness, dysphagia, tongue deviation to left, paralysis of left soft palate, paralysis of left vocal cord	Corticosteroids for 2 weeks, SLT	Partial	8 months
Gevorgyan et al., 2013 20	48 F	Liposuction	-	-	-	-	Unchanged	3 years
Park et al., 2013 21	53 M	C4-5 posterior foraminotomy/discectomy	ETT	Day 1	Hoarseness, hypesthesia of left tongue, buccal cavity and left tongue deviation, absent action potentials on EMG of left thyroarytenoid muscle	-	Complete	6 months
Park et al., 2013 21	56 M	Right C2 Hemilaminectomy/C3-5 laminoplasty	ETT	Immediate post-op	Hoarseness, hypesthesia of left tongue, deviation of tongue to left	-	Complete	2 months
Lim et al., 2013 22	64 M	C3-7 laminoplasty	ETT	Day 1	Dysphagia, aspiration, deviation of tongue left, left vocal cord paralysis	IV corticosteroids, electrical stimulation therapy, NGT, balloon dilation of esophagus	Complete	3 months
Ghorbani et al., 2014 23	27 M	Septorhinoplasty	ETT	Day 1	Hoarseness, dysphagia, dysarthria, aspiration, tongue deviation left, left true vocal cord paralysis	Prednisolone 50 mg, vit B1/12 for 7 days	Complete	6 months
Coninckx et al., 2015 24	49 M	Cardiopulmonary resuscitation	ETT	Immediate post-extubation	Hoarseness, swallowing problems, absent gag reflex and paralysis of tongue musculature without deviation, bilateral vocal cord paralysis	Prednisolone 48 mg for 8 weeks, PEG, SLT	Complete	4 months
Coninckx et al., 2015 24	64 M	PNA and ARDS	ETT	Swallowing difficulties immediately after extubation, formal diagnosis of hoarseness and dysphagia day 8, FFL diagnosis made at day 40	Hoarseness, dysphagia, left deviation of tongue, left vocal cord paralysis	SLT, PEG	Complete	22 months
Noroozi et al., 2016 25	29 M	Liver transplant	LMA	Day 1	Right tongue deviation, hoarseness, swallowing difficulty, tongue fasciculations	-	Complete	2 weeks
Cariati et al., 2016 5	36 M	I&D right neck abscess w cervical approach	ETT	Immediate post-op	Dysphonia, tongue deviation toward affected side, lingual motility disturbance, swallowing difficulty	SLT	Complete	3 months
Cariati et al., 2016 5	42 M	Shoulder fracture reduction w direct anterolateral approach	ETT	Immediate post-op	Dysphonia, tongue deviation toward affected side, lingual motility disturbance, swallowing difficulty, insensibility of lateral border of tongue, lingual bite wounds	SLT	Complete	3 months
Cariati et al., 2016 5	61 M	I&D right neck abscess w cervical approach	ETT	Immediate post-op	Dysphonia, tongue deviation toward affected side, lingual motility disturbance, swallowing difficulty	SLT	Complete	3 months
Silva et al., 2017 26	41 F	Right C5-6 and left C6-7 posterior foraminotomy	ETT	Day 1	Hoarseness, left tongue swelling, dysphagia, cough, tongue deviation left, left vocal cord paresis	SLT, vocal cord medialization injections	Partial	1 year
Mumtaz et al., 2018 27	23 M	Control of oral bleeding and epistaxis after upper right wisdom tooth removal	ETT	Day 11	Deviation of the tongue to the right side and dysphonia	SLT and dietary support	Complete	3 months
Jee et al., 2018 28	41 F	Total mastectomy and breast reconstruction w latissimus dorsi flap	ETT	Day 1	Left sided tongue deviation, subtle hoarseness, swallowing difficulty, immobility of true left cord	Prednisolone 5 mg daily for 3 weeks	Complete	1 months
Kerolus et al., 2018 29	48 F	C2-3 ACDF	ETT	Day 7	Left facial numbness, facial droop, tongue deviation, dysphagia, dysphonia	SLT	Complete	6 weeks
Ma et al., 2020 30	28 M	Repair of nasal septal perforation and septoplasty	Anesthesia cannula?	???	Hoarseness, disturbed speech, dysphagia, tongue deviation to right, right vocal cord paralysis	Ginkgo leaf extract and dipyridamole and methylcobalamin (patient refused glucocorticosteroids), mouse nerve growth factor inj. 1 week post-op, acupuncture, local physiotherapy	Partial	1 months
Sehara et al., 2020 31	70 M	Cervical laminoplasty	ETT	Immediate post-op	Dysphagia, hoarseness, and tongue deviation to the right side, limitation in the abduction of the right vocal cord and insufficient glottal closure on utterance	IV methylprednisolone 1gm for 2 days tapered over 2 weeks	Complete	3 months
Waits et al., 2020 4	52 M	Multilevel cervical laminoplasties for stenosis	ETT	Immediate post-op	Left unilateral vocal cord paralysis, unilateral tongue deviation, and dysphagia	IV dexamethasone every 6 hr for 24 hr followed by another 24 hr oral dosing at lower dose, PEG tube, 21 day prednisone taper at 1-month follow-up	Complete	3 months
Jose et al., 2022 32	72 M	Mitral valve repair	ETT, TEE	Immediate post-op	Weak voice, choking, deviation of tongue to the right, paramedian position of right cord	SLT	Complete	6 months
Steehler et al., 2022	70 M	AVR and CABG	ETT	Immediate post-op	Hoarseness, dysphagia, aspiration, tongue deviation left, left vocal cord paralysis	SLT, carboxymethylcellulose gel implant injection on postoperative day 36	Partial	9 months

The wide variety of cases in the literature in which Tapia Syndrome was found hints to multiple possible underlying etiologies. Incorrect laryngoscope use, overinflation of the endotracheal tube cuff, neck hyperextension, and prolonged packing of the throat. ²⁷ Tapia’s syndrome has also been shown by use of a laryngeal mask airway (LMA) ²⁵ and nasotracheal intubation. ¹⁸ Cranial nerve XII, in relation to LMA use, runs in close proximity to the greater horn of the hyoid, at the location of the superior portion of the LMA. ³³ In addition, CN XII and CN X both course closely together at the level of the first cervical vertebra.^17,34

Patients with Tapia Syndrome present in the acute postoperative setting. The initial evaluation of the patient should include a thorough head and neck physical exam to differentiate between central and peripheral lesions. Boga et al. described a classification system of Tapia syndrome, termed the Aktas and Boga Classification, that represents the array of symptomatology seen depending on the severity of nerve injury. Grade I (mild type) consists of unilateral cord and tongue paralysis, no uvula distortion, minimal slowdown in speaking, no tongue swelling and lack of dysphagia. Grade II (moderate type) consists of unilateral cord and tongue paralysis, no uvula distortion, mild slowdown in speaking, tongue swelling, pharynx dryness, dysphagia, cracked speech and normal feeding and drinking. The most severe type, grade III, consists of unilateral cord and tongue paralysis, uvula distortion, dysarthria, tongue swelling, dryness in the pharynx, dysphagia, and difficulties with both feeding and drinking. ¹³

Management of Tapia syndrome is theorized in some cases as treatment of neuropraxia (e.g., stretch or crush type injury)—often involving steroids. ⁴ Boga et al. grading scale suggests corticosteroids and IV corticosteroids for grades II and III, respectively. ¹³ For grade I cases, supportive treatment and close follow-up is recommended. Supportive treatment can be composed of anti-inflammatory drugs and warm air inhalation, with most cases resolving in 4–6 months. Literature also shows the advantages of interdisciplinary care and the role of speech language pathology to hasten recovery and prevent adverse outcomes. ⁵

Outside of our study, there is only 1 other case reported in the literature with follow-up of a patient that underwent vocal cord injection. Silva et al. had a partial recovery after 1 year with excellent vocal cord closure and normal voice despite continued paralysis of unilateral true cord. Our patient also did not experience a complete recovery; however, we believe the vocal cord injections assisted in resolving comorbidities associated with penetration and aspiration risk inherent to Tapia’s syndrome.

Lykoudis et al. described several preventive measures that have been discussed in previous reviews of Tapia Syndrome, including careful head and neck maneuvers during intubation, gentle intubation with the laryngoscope, proper cuff inflation, proper tube fixation to decrease excessive tube motion, and careful positioning of throat packing. ²

Conclusion

Reported cases of Tapia’s syndrome are rare; however, it is important to recognize the common presenting signs, symptoms, and management of Tapia syndrome, along with potential preventative techniques. The clinician should consider this diagnosis in those presenting with symptoms of CN XII and CN X neuropraxia in the postoperative or post-extubation setting as prompt diagnosis and management are warranted. Vocal cord injection is a safe procedure in managing Tapia’s syndrome to prevent comorbidities of vocal cord paralysis.