TONGUE PARALYSIS AFTER OROTRACHEAL INTUBATION IN A PATIENT WITH PRIMARY MEDIASTINAL TUMOR

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1 TONGUE PARALYSIS AFTER OROTRACHEAL INTUBATION IN A PATIENT WITH PRIMARY MEDIASTINAL TUMOR Esther Uña (1), Francisco Gandía (2), Jose Luis Duque (3) (1) Medical Oncology Service. Clinical Universitary Hospital. Valladolid (Spain) (2) Intensive Care Unit. Clinical Universitary Hospital. Valladolid (Spain) (3) Thoracic Surgery Service. Clinical Universitary Hospital. Valladolid (Spain) Correspondence author: Dr Esther Uña Cidon Medical Oncology Service. Clinical Universitary Hospital. C/ Ramon y Cajal s/n Valladolid (SPAIN). Phone number: aunacid@hotmail.com

2 ABSTRACT Hypoglossal nerve injury is a very rare complication of anaesthesia airway management. The cause argued to this lesion is neuropathy of this nerve provoked by its compression due to inflation of the cuff within the larynx or damage secondary to hyperextension of the neck during a difficult intubation. Although most cases describe Tapia s syndrome with hypoglossus and recurrent laryngeal nerve paralysis, the cases which describe only bilateral hypoglossus palsy are very unfrequent. We report here a case of bilateral palsy of hypoglossus nerves in a 28-year-old man undergoing a diagnostic anterior mediastinotomy as a part of the study of a mediastinal mass.

3 INTRODUCTION Several lesions have been described as post-intubation complications. Most frequent are injuries of the pharynx and larynx such as oedema or ulcerations of both of them, chondromalacy of the larynx, oesophago-tracheal fistula, stenosis of the larynx or trachea and paralysis of the vocal cords (1). Although etiologic factors are not very well known, it is supposed that the chemical material of the tube or sterilization products used are the main causes. But also the pressure on the adjoining tissues secondary to the oro-tracheal tube has been described as another etiologic factor (1). Few cases, however, have been reported with hypoglossal nerve injury secondary to anaesthesia airway management. The cause argued to this lesion is neuropathy of this nerve provoked by its compression due to inflation of the cuff within the larynx or damage secondary to hyperextension of the neck during a difficult intubation (2). We report here a case of bilateral palsy of hypoglossus nerves in a 28-year-old man undergoing a diagnostic anterior mediastinotomy as a part of the study of a mediastinal mass. CASE REPORT A 28-year-old man was referred to our hospital due to dry cough for 2 months associated with dyspnea in the previous week. He has been previously healthy and without a history of smoking. Physical examination revealed that on thoracic auscultation heart and lung sounds were clearly heard. Chest imaging studies, including X-ray and computed tomography (CT), revealed a mass, measuring 15x12x10 cm in the anterior mediastinum with pericardial and pleural effusions. Fine-needle puncture of this lesion showed indifferentiated cells suggesting lymphoma. The patient underwent a diagnostic anterior mediastinotomy to obtain a biopsy of this lesion to complete the study before the treatment. Following awake fiberoptic tracheal intubation, general anaesthesia was administered. Diagnostic mediastinotomy was performed without incident. After that the patient with

4 peripheral desaturation to 60%. The patient had to be intubated again and he remained in the intensive care unit. Pathological analyses revealed Yolk sac tumor and laboratory data showed a marked elevation of serum alfa-fetoprotein (AFP) up to ng/ml with normal serum levels of β- human chorionic gonadotropin (β-hcg). The patient thus received chemotherapy with EP (VP-16 or etoposide and cisplatin) without bleomycin to decrease the risk of pulmonary toxicity. He showed a very slow improvement so he underwent a tracheostomy to maintain tracheal intubation. There were necessary 3 courses of chemotherapy and 8 Gy of radiotherapy to ensure that tumor size was reduced sufficiently to allow extubation and discharge to the plant. The tumor shrank to 10x7x5 cm and serum AFP dropped to 3750 ng/ml. Globally the patient remained a total of 70 days in intensive care unit and 62 days intubated. After discharging the patient complained of inability to swallowing and difficulty of speaking with normal vibration of vocal cords. This problem resulted in loss of 5 kg of weight. The patient needs to be fed by nasogastric tube. Examination revealed no structural alterations of the soft palate. The tongue showed bilateral hypotrophy with inability to drive forward the tongue suggesting a bilateral hypoglossus palsy. Meticulous neurological examination, including also magnetic resonance imaging (MRN) revealed no evidence of central or cranial nerve involvement. On account of the spatial distance, there is no evidence of injuring the hypoglossal nerve during this type of surgery. We atributted the etiology of this palsy to a neuropathy after a prolonged compression of the neighboring tissues secondary to the tube which could be transferred to these nerves. With conservative management including steroids and speech-swallowing reeducation therapy the patient achieved full recovery of lingual functions within four months after orotracheal intubation. Five months after the surgery, progressive tumor in the anterior mediastinum was showed in chest CT. The patient received 4 additional courses of chemotherapy with TIP (ifosfamide, paclitaxel and cisplatin) with partial response. In the last control he was waiting for a complete excisional surgery.

5 DISCUSION The hypoglossal nerve is a pure motor nerve that innervates all the muscles of the tongue. This nerve is divided into five segments: medullary, cisternal, skull base, nasopharyngeal and oropharyngeal, carotid space, and sublingual. Each segment might be affected by different disorders (3). The atrophy or hypotrophy of the tongue muscles is only seen when the nuclear or peripheral segments of the nerve are involved. In its course distal to the base of skull this nerve may be affected by vascular aneurysm, local infections, surgical procedures such as carotid endarterectomy, accidental trauma or tumors (4). There are many series describing cases of hypoglossal nerve palsy. In the Tommasi- Davenas (5) serie with 32 patients with paralysis of the hypoglossal nerve and tongue atrophy, they found only eight cases of isolated nerve XII palsy without neurologic involvement of other cranial nerves and in most cases the cause was tumor, especially bone metastasis. However, few cases have been described related to surgical procedure or anaesthesia airway management and most of them were unilateral paralysis without tongue hypotrophy and combined with palsy of other cranial nerves such as recurrent laryngeal branch of the vague nerve (Tapia s syndrome) (6-9). Although any surgical procedure might have complications, which may vary from mild to very unexpected and severe episodes, it is very rare this neuropathy (10). Gelmers HJ described two cases of Tapia s syndrome after a thoracotomy procedure (10). The cause argued was a structural lesion localized at the point of crossing of the vagal and hypoglossal nerves but another cause may be neuropathy of both nerves due to inflation of the cuff within the larynx with a neuropathy secondary. But an alternative explanation was related with the damage to these nerves secondary to traction of the esophagus that may be transferred to both nerves which are closely connected in many sites. Yavuzer at al. described another case of Tapia s syndrome after septorhinoplasty. They believed that the picture was caused by pressure neuropathy of nerves due to inflation of the cuff within the larynx (11). Globally after a careful review of the literature, two probable mechanisms of nerve injury are considered: compression by the endo-tracheal tube upon the throat pack in the oropharynx or a stretching mechanism of these nerves due to excessive flexion of the head (2). These two mechanisms of nerves injury are believed to be related with

6 neuropraxic origin due to pressure to the lateral roots of the tongue during routine transoral intubation (2, 12, 13). It has been described another case of Tapia s syndrome secondary to direct injury of the soft palate and oro-pharynx by a foreign body (2). Our patient presented with clinical signs that localized the lesion to the peripheral segment of cranial nerve XII. Neuroimaging revealed no lesions in central nervous system or cranial nerves. Although we are not describing a complete Tapia s syndrome, we could describe our case as a bilateral incomplete Tapia s picture with isolated bilateral paralysis of the muscles of the tongue secondary to hypoglossus damage, without evidence of other craneal nerves affected. In our case, the clinical picture can not be related with the surgical procedure or tumor compression because the spatial distance between the surgical or tumoral area and the hypoglossus nerve and its branches course. We have attributed this clinical picture to a neuropathy secondary to prolonged oro-tracheal intubation although it was not difficult or complicated. To our knowledge there are very few cases in the literature describing cases similar to ours with postoperative bilateral Tapia s syndrome with isolated hypoglossal paralysis without other nerves affected secondary to airway management in a surgical procedure with oro-tracheal intubation (8). The progressive recovery of the functions in this patient and in the majority of the cases reported in the literature support the neuropraxic type of nerve damage (2). Although cranial nerves injury following routine endo-tracheal intubation appears to be rare, this complication should be considered by the otolaryngologist and anaesthesiologist (2). Special attention should be paid to correct positioning of the head during surgery to avoid such problems and early start of language/swallowing reeducation is advisable (2).

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