Introduction
Impairment of the protective function of the larynx can lead to intractable aspiration owing to repeated soilage of the tracheobronchial tract and result in frequent life-threatening pneumonia.Reference Bartlett and Gorbach1 While surgery is very effective, it often sacrifices the phonatory function. Hence, we propose a combined technique consisting of an open medialisation thyroplasty with intra-operative salivary botulinum toxin injection, which provides definitive treatment for intractable aspiration while preserving phonation. This paper aims to introduce our approach which offers these patients a better quality of life and highlights the importance of performing both procedures together to ensure a successful outcome.
Materials and method
Case report
A forty-three-year-old man diagnosed with a left pontomedullary cavernoma underwent excision via a left retrosigmoid approach. Post-operatively, he complained of a weak, breathy voice, swallowing difficulties and a chronic cough, particularly when drinking. Flexible nasopharyngolaryngoscopy revealed a left vocal fold palsy in cadaveric position with a large phonation gap and pooling of saliva.
Subsequently, he was hospitalised for aspiration pneumonia, and a tracheostomy was performed for prolonged ventilation. Fibreoptic endoscopic evaluation of swallowing further confirmed evidence of aspiration, and he was only allowed feeding via a nasogastric tube. His left glossopharyngeal (CN IX) and vagus (CN X) nerve injury following his surgery was a debilitating complication which significantly impacted his quality of life. His weak, breathy voice resulted in poor communication, and his prohibition of oral intake led to considerable weight loss.
He was referred to the speech-language pathologist, but there was only minimal improvement following six months of consistent therapy. Despite his intractable aspiration, the patient refused a PEG tube and was very adamant about weaning off both the nasogastric and tracheostomy tubes. This was a challenging case, as we would need to overcome his intractable aspiration and improve his vocal function.
Surgical approach
We devised a combined technique of open medialisation thyroplasty with intra-operative botulinum toxin type A injection into the salivary glands. The thyroplasty procedure medialises the left vocal fold to overcome glottic insufficiency. Botulinum toxin type A injection into the salivary glands reduces salivary flow and prevents excessive pooling caused by the dysfunction of his pharyngeal muscles. For this treatment, patients should be carefully selected and fully informed of the potential benefits and harm.
The operation was performed under general anaesthesia, as the patient could not tolerate lying in a supine position due to his constant coughing triggered by aspiration. He was ventilated via a cuffed tracheostomy tube. A 5 cm horizontal incision was placed in a skin crease at the level of the mid-thyroid cartilage. Subplatysmal flaps were raised superiorly to the hyoid and inferiorly to the upper cricoid. Division of the midline raphe between the strap muscles exposed the thyroid cartilage.
It was imperative to ensure optimal placement of the window in the thyroid cartilage to minimise the risk of poor voice outcome after medialisation. The vocal fold was medialised, and the effect of the displacement was visualised on the monitor via the flexible nasopharyngolaryngoscope. After taking the appropriate measurements, an implant was carved out of medium-grade silicon block. Once the implant was in place, the laryngoscopic image was observed to confirm adequate medialisation. The wound was irrigated with diluted povidone and closed in layers.
Next, we injected 20 units into each submandibular and parotid gland respectively, under ultrasound guidance in the same setting. The dilution protocol was as follows: 2 ml of 0.9 per cent normal saline to a vial of 100 units, creating a dilutional concentration of 5 units of toxin in every 0.1 ml of the reconstituted mixture. We believe the injection technique with a surgeon-performed ultrasound increases the accuracy and allows dynamic monitoring during the procedure.
The patient was discharged well the next day without any immediate post-operative complications.
Results
The patient was followed up for twelve months, during which he was seen monthly for the first six months and at three-month intervals thereafter. The patient’s voice quality exhibited significant improvement during post-operative day 1, while substantial salivary reduction was noticeable after two months. Flexible nasopharyngolaryngoscopy revealed a left vocal fold palsy in median position with no phonation gap and no obvious penetration or aspiration. He continued seeing the speech-language pathologist for intensive therapy. Eight weeks later, the video fluoroscopy demonstrated no evidence of aspiration. Following that, the patient was allowed a regular oral diet with removal of his nasogastric tube and successful decannulation.
Discussion
Recurrent aspiration heightens the risks of pneumonia and death, as well as adds to the financial burden in terms of treatment and frequent hospital admissions. Conservative measures such as nutritional support via nasogastric tube or gastrostomy, oral care and swallowing rehabilitation have been described. Surgical treatments are performed when there is unsatisfactory improvement, but they often result in the loss of vocal function. We seek an approach that provides effective treatment for intractable aspiration without damaging the phonatory function and offers the best quality of life. To our best knowledge, this is the first paper to report our coupled technique of open medialisation thyroplasty with intra-operative botulinum toxin type A injection into the salivary glands.
The three main methods to treat intractable aspiration are removal of the larynx including total laryngectomy and central-part laryngectomy,Reference Cannon and McLean2–Reference Kawamoto, Katori, Honkura, Kakuta, Higashi and Ogura4 altering the tracheal structure, such as tracheoesophageal diversion and laryngotracheal separation,Reference Lindeman5–Reference Eibling, Snyderman and Eibling8 and surgical closure of the larynx.Reference Remacle, Marza and Lawson9–Reference Takano, Goto, Kabeya and Tayama12 The first approach is rather invasive, especially total laryngectomy, and is associated with a lengthier operation time and a greater amount of bleeding. Our technique is advantageous as it is minimally invasive, has a shorter operation time and a low risk of bleeding. In addition, laryngectomy can only be carried out under general anaesthesia. Our approach can be performed under local anaesthesia, although in our case, it was not feasible owing to the patient’s constant coughing that was triggered by his aspiration. Nevertheless, it would be ideal in instances where the patient’s comorbidities are not suitable for general anaesthesia.
Post-operative sacrifice of vocal function is also an expected drawback of these surgeries. While post-operative voice rehabilitation, such as electrolarynx and oesophageal speech, can enhance the patient’s quality of life, the voice is less than ideal. For the first two methods, restoration of phonation can be achieved via a tracheoesophageal puncture and voice prosthesis insertion. However, this is an additional procedure and leads to a longer operating time. There are many complications related to tracheoesophageal puncture, and a favourable voice outcome is not guaranteed. Our approach does not require any extra steps and is perfect for patients with a strong demand for vocalisation. Our patient was able to phonate immediately, and his voice showed significant improvement in terms of quality and amplitude.
We recommend that our combined therapy should be considered as first-line surgical treatment for patients who have failed conservative therapy. The technique is the least invasive with minimal risks and provides the patient with an opportunity to improve his or her quality of life. The more aggressive surgeries can be performed if the patient continues to aspirate. Moreover, the presence of a tracheostoma is inevitable in all the other methods described. From the aesthetic point of view, our combined method has the best outcome, as it is the only one which offers patients a chance at tracheostomy decannulation.
Another factor that needs to be considered is the primary disease causing the intractable aspiration. Patients with high vagal lesions resulting from cerebrovascular accidents, skull base tumours or iatrogenic complications suffer from breathy dysphonia, dysphagia and aspiration. This is because of the impairment of vocal fold movement, loss of laryngeal sensation and poor co-ordination of the oropharyngeal and hypopharyngeal phases of swallowing. Hence, our coupled technique is ideal for these patients, and it must be emphasised that patient selection is critical to ensure a successful outcome. We would also like to highlight that performing only either procedure will likely lead to unsatisfactory results.
Several known side effects of botulinum include dry mouth, dysphagia and chewing difficulties. The action of this neurotoxin generally wears off after three to four months but can last up to six months. Nevertheless, our patients who have received botulinum, including those with pharyngocutaneous fistula and sialorrhoea, usually do not require repeat injections. In this case, the patient tolerated the procedure well, and another dose of botulinum may be considered if clinically indicated.
Nonetheless, our approach does have its own disadvantages. While suitable for patients with intractable aspiration caused by high vagal lesions, there is a risk of failure when multiple cranial nerves are involved. Our method does not restore sensation to the larynx and is not aimed directly at improving the swallowing function. The age of the patient will undoubtedly influence the outcome, as compensation from the unaffected side is more likely in younger patients. We believe that this may be overcome with intensive post-operative rehabilitation and the patient’s determination to resume oral digestion.
Conclusion
This paper demonstrates the feasibility of our combined approach as a viable treatment option for intractable aspiration, particularly in patients with a strong desire for vocalisation. Nevertheless, the cases must be selected carefully to ensure a favourable outcome.
Acknowledgements
None.
Financial support
This research received no specific grant from any funding agency, commercial or not-for-profit sectors.
Competing interests
The authors declare none.
Ethical statement and informed consent
The authors assert that all procedures contributing to this work comply with the ethical standards of our national and institutional guidelines on human experimentation and with the Helsinki Declaration of 1975, as revised in 2008. The authors assert that all procedures contributing to this work comply with our institutional and national ethical standards. The Institutional Ethical Committee has approved the study.
