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Vocal Fold Paralysis Secondary to Subclavian Venous Thrombosis Abdul-Latif Hamdan, and Helene Dabbous, Beirut, Lebanon Summary: The etiology of vocal fold paralysis is multifaceted. One of the rare causes is internal jugular vein thrombosis secondary to central venous catheterization. The palsy is usually ipsilateral to the site of indwelled catheterization, self-limited and reversible. The authors of this manuscript report a rare case of contralateral recurrent laryngeal nerve palsy induced by subclavian vein thrombosis following central venous catheterization. The patient developed irreversible vocal fold paralysis that necessitated office-based injection laryngoplasty. Key Words: Subclavian venous thrombosis−Vocal fold paralysis−Injection laryngoplasty−Dysphonia−Larynx.
INTRODUCTION Vocal fold paralysis is a common diagnosis in otolaryngology practice. Affected patients usually present with hoarseness, vocal fatigue, aspiration, dysphagia and at times shortness of breath. The symptoms depend primarily on the position of the paralyzed vocal fold as seen on laryngeal examination. The etiology of vocal fold paralysis is multifaceted. The main causes include iatrogenic injury following surgical intervention, and neoplasms compressing the recurrent laryngeal nerve along its course in the chest and neck. The surgeries most commonly reported are thyroidectomy, thoracic surgery such as lobectomy and mediastinal dissection, base of skull surgery, and cervical disk surgery using an anterior approach. Other reported etiologies include neck dissection, endotracheal intubation, and central venous catheterization. In almost one third of the cases, the etiology remains unknown.1,2 The authors of this manuscript describe a rare case of unilateral vocal fold paralysis secondary to subclavian venous thrombosis following the insertion of a central venous catheter. A review of the complications of central venous catheterization is presented. CASE REPORT A 70-year-old man, diagnosed with advanced stage IV colon mucinous adenocarcinoma, presented to the voice clinic with a chief complaint of dysphonia and occasional aspiration of several years duration. Medical history was positive for diabetes mellitus type II and hypertension. The patient reported the onset of dysphonia 4 months following central venous catheterization for administration of chemotherapeutic agent, a procedure that was performed 4 years prior to his presentation. The catheter was inserted through the left internal jugular vein and was threaded until the tip reached the right Accepted for publication December 30, 2019. There is no conflict of interest or financial support in relation to this paper. From the Department of Otolaryngology − Head & Neck Surgery, American University of Beirut Medical Center, Beirut, Lebanon. Address correspondence and reprint requests to Abdul-Latif Hamdan, American University of Beirut, Department of Otolaryngology, P.O. Box: 11-0236, Beirut, Lebanon. E-mail:
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subclavian vein. The central venous line was complicated few months later by thrombosis at the catheter tip located in the right subclavian vein, with evidence of opacification of the right brachiocephalic/subclavian vein and lack of contrast in the superior vena cava as shown by polysitogram. The diagnosis was further confirmed by computerized tomography of the chest which revealed occlusion of the proximal part of the superior vena cava reaching the catheter tip (Figure 1). It is worth noting that there was no evidence of pulmonary or mediastinal metastasis. Flexible fiberoptic laryngoscopy performed at that point in time showed right vocal fold paralysis in the paramedian position with incomplete closure of the vocal folds during phonation (documented in the medical record of the physician’s note). His laryngeal examination 4 years later did not show any interval changes. Repeated computerized tomography of the chest showed same previously described findings despite removal of the catheter. The patient underwent office-based injection laryngoplasty of the right vocal fold using hyaluronic acid (Figure 2).
DISCUSSION Central venous catheterization is commonly performed in cases of chemotherapeutic drug administration, whereby a chemo-port catheter (Polysite) is inserted below the skin. This latter is usually introduced on the right side and tunneled into the internal jugular vein, thereby offering a direct passage into the large venous system.1,3,4 Common complications encountered upon insertion of a tunneled central venous catheter include hematoma formation, venous thrombosis, air embolus, pneumothorax or hemothorax, as well as injury to major neck structures such as the great vessels, trachea, and esophagus.1,5,6 Injury to the recurrent laryngeal nerve during central venous catheterization has also been reported in view of the proximity of the nerve to the internal jugular vein behind the carotid sheath.1,3,7,8 Common risk factors for injury during catheterization include the number of attempts at cannulation, excessive catheter manipulation during insertion, and the duration of cannulation.1,4,5,8 An additional significant risk factor is venous thrombosis. Symptomatic venous thrombosis subsequent to central venous catheterization has
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FIGURE 1. Chest computer tomography with contrast showing the tip of the chemo-port catheter with surrounding vein thrombosis (arrow).
FIGURE 2. Endoscopic laryngeal view showing the tip of the fiberoptic injection needle during injection laryngoplasty using hyaluronic acid. been reported with a frequency reaching almost 10%.9 Most commonly, thrombosis occurs at the site of entry of the catheter into the vein, or at any point where there is contact or friction between the catheter and the wall of the vein.9 In 1993, Keidan RD reported three cases of recurrent laryngeal nerve palsy following central venous catheterization. The authors alluded to thrombophlebitis and mediastinal inflammation as potential causes.10 In 1995, Martin-Hirsch
and Newbegin described two cases of vocal fold paralysis following right central venous catheterization, and suggested several mechanisms for recurrent laryngeal nerve injury during the indwelling access to the central venous system.8 Herve et al reported internal jugular vein thrombosis as a result of migration of the chemo-port-catheter tip, which led to stagnation and extravasation of the administered chemotherapeutic agents with subsequent injury to the recurrent laryngeal nerve.7 The authors of this manuscript present another rare case of recurrent laryngeal nerve palsy as a result of right subclavian vein thrombosis. The presence of thrombosis at the level of the right subclavian vein as a consequence of left chemo-port insertion is the only significant event that could explain the patient’s vocal fold paralysis on the right side. The late onset of dysphonia (within 4 months) precludes or makes it very unlikely that the injury has occurred during the insertion of the catheter. Moreover, there was no note of extravasation of the chemotherapeutic medications administered to the patient during therapy. The chemotherapy regimen used included leucovorin calcium, fluorouracil, oxaliplatin, and bevacizumab (Avastin). Nevertheless, it is worth noting that though the radiologic findings and the temporal pattern of events strongly allude to venous thrombosis as the cause of the vocal fold paralysis, a viral etiology should not be disregarded. Of additional interest in this report is the permanent recurrent laryngeal nerve palsy necessitating surgical intervention, unlike previously reported cases where recurrent laryngeal nerve injury was reversible and self-limited. In this particular case, given the comorbidities of the patient, an office-based procedure was chosen as a treatment modality.
ARTICLE IN PRESS Abdul-Latif Hamdan and Helene Dabbous
Vocal Fold Paralysis Secondary to Subclavian Venous Thrombosis
Alternatively, affected patients may undergo laryngeal framework surgery for a more permanent solution. CONCLUSION The authors present a rare case of vocal fold paralysis secondary to right subclavian vein thrombosis. Recurrent laryngeal nerve palsy can occur late following central venous catheterization and may be irreversible. REFERENCES 1. Alazzawi S, Hindi K, Malik A, et al. Chemo-port insertion: a cause of vocal cord palsy. Laryngoscope. 2015;125:2551–2552. 2. Granato F, Martelli F, Comini LV, et al. The surgical treatment of unilateral vocal cord paralysis (UVCP): qualitative review analysis and meta-analysis study. Eur Arch Otorhinolaryngol. 2019;276:2649–2659. 3. Koduri S, Ng AK, Roy D, et al. Ipsilateral vocal fold paralysis: an unusual complication of internal jugular tunnelled dialysis catheter insertion. J Vasc Access. 2019;21:116–119.
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4. Sim DW, Robertson MR. Right vocal cord paralysis after internal jugular vein cannulation. J Laryngol Otol. 1989;103: 424. 5. Salman M, Potter M, Ethel M, et al. Recurrent laryngeal nerve injury: a complication of central venous catheterization-a case report. Angiology. 2004;55:345–346. 6. Arshad FA, Eng CY, Daw W, et al. Vocal cord paralysis following central line insertion in a neonate: a review of the literature. BMJ Case Rep. 2011;2011: pii: bcr1120103543. 7. Herve S, Conessa C, Desrame J, et al. Acute laryngeal paralysis induced by the migration of a totally implantable venous access device's catheter tip. J Laryngol Otol. 2004;118:237–239. 8. Martin-Hirsch DP, Newbegin CJ. Right vocal fold paralysis as a result of central venous catheterization. J Laryngol Otol. 1995;109:1107– 1108. 9. Kurul S, Saip P, Aydin T. Totally implantable venous-access ports: local problems and extravasation injury. Lancet Oncol. 2002;3: 684–692. 10. Keidan RD. Recurrent laryngeal nerve palsy secondary to massive venous thrombosis. J Laryngol Otol. 1993;107:54–57.