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more likely diagnosis than an arteriovenous malformation or dural arteriovenous fistula, which can, however, have the same appearance of dilated vessels on MRI scans. Selective spinal angiography is useful in the differential diagnosis and assessment of the vascularity. While small haemorrhages from a paraganglioma are common microscopically, a severe cauda equina paraganglioma haemorrhage on MRI is rare. Li et al. reported a case with a subarachnoid haemorrhage, but with no motor weakness or sphincter disturbance.1 One of our patients became paraplegic following a postoperative haemorrhage and did not recover despite haematoma removal. No residual tumour was found at the second operation. This haemorrhage was likely to have originated from one of the serpentine associated vessels. Although cauda equina paragangliomas are benign, slow-growing, well-defined tumours, their vascular nature must be considered, as for other tumours of paraganglia cell origin. Knowledge of their potential endocrine and vascular effects should alert the surgeon and anaesthesiologist to possible undesirable operative and post-operative events.
References 1. Li P, James SLJ, Evans N, et al. Paraganglioma of the cauda equina with subarachnoid haemorrhage. Clin Radiol 2007;62:277–80. 2. Boncoeur-Martel MP, Lesort A, Moreau JJ, et al. MRI of paraganglioma of the filum terminale. J Comput Assist Tomogr 1996;20:162–5. 3. Cabello A, Ricoy JR. Paraganglioma of the cauda equina. Cancer 1983;52:751–4. _ 4. Iliadis C, Kirgiannis K, Wozniak G, et al. Paraganglioma of cauda equina. Neurol Neurochir Pol 2008;42:463–6. 5. Aghakhani N, George B, Parker F. Paraganglioma of the cauda equina region— report of two cases and review of the literature. Acta Neurochir 1999;141:81–7. 6. Yoshida A, Umekita Y, Ohi Y, et al. Paraganglioma of the cauda equina. A case report and review of the literature. Acta Pathol Jpn 1991;41:305–10. 7. Pigott TJD, Lowe JS, Morrell K, et al. Paraganglioma of the cauda equina: report of three cases. J Neurosurg 1990;73:455–8. 8. Miliaras GC, Kyritsis AP, Polyzoidis KS. Cauda equina paraganglioma: a review. J Neurooncol 2003;65:177–90. 9. Miller A, Torack RM. Secretory ependymoma of the filum terminale. Acta Neuropath 1970;15:240–50. 10. Yang SY, Jin YJ, Park SH, et al. Paragangliomas in the cauda equina region: Clinicopathoradiologic findings in four cases. J Neurooncol 2005;72:49–55. 11. Djindjian M, Ayache P, Brugieres P, et al. Giant gangliocytic paraganglioma of the filum terminale. J Neurosurg 1990;73:459–61. 12. Horoupian DS, Kerson LA, Saıontz H, et al. Paraganglioma of cauda equina. Clinicopathological and ultrastructural studies of an unusual case. Cancer 1974;33:1337–48.
doi:10.1016/j.jocn.2009.12.026
Hypopharyngeal injury leading to subcutaneous emphysema after anterior cervical discectomy and artificial cervical disc placement Girija P. Rath a,*, Manish Marda a, Navdeep Sokhal a, Pankaj Singh b, P. Sarat Chandra b a b
Department of Neuroanaesthesiology, Neurosciences Center, 6th Floor, Room No. 9, All India Institute of Medical Sciences, New Delhi 110029, India Department of Neurosurgery, All India Institute of Medical Sciences, New Delhi 110029, India
a r t i c l e
i n f o
Article history: Received 29 September 2009 Accepted 4 January 2010
Keywords: Anterior cervical discectomy Hypopharyngeal injury Subcutaneous emphysema
a b s t r a c t Anterior cervical discectomy and fusion (ACDF) may lead to complications such as dysphagia, hoarseness, recurrent laryngeal nerve palsy, vocal cord palsy, dural tears with leakage of cerebrospinal fluid, haematoma, and oesophageal and vascular injuries. Hypopharyngeal injury in the early postoperative period is a rare, but life-threatening, problem. We present a patient with dyspnoea who developed subcutaneous emphysema in the early postoperative period after an iatrogenic hypopharyngeal injury following ACDF. These complications prolonged the postoperative course of the patient. The role of careful surgical intervention, and anticipation of potential complications, has been emphasized, especially when surgical exposure of the anterior spinal canal above C4 is required. Ó 2010 Elsevier Ltd. All rights reserved.
1. Introduction Management of a prolapsed intervetebral disc (PIVD) in the cervical spine involves anterior approach discectomy and fusion of related vertebrae. Robinson et al. first described this surgical technique in 1955, in which the trachea and oesophagus are retracted to expose the anterior spinal column.1 Various complications of this procedure have been reported, including dysphagia, hoarseness, recurrent laryngeal nerve palsy, vocal cord palsy, dural tears with leakage of cerebrospinal fluid, haematoma, and oesophageal and vascular injuries.2 However, injury to the hypopharynx during anterior cervical discectomy and fusion (ACDF) is rare.3,4 We describe a patient who presented in the early postoperative
* Corresponding author. Tel.: +91 98 68398204; fax: +91 11 26588663. E-mail address:
[email protected] (G.P. Rath).
period with dyspnoea that developed from subcutaneous emphysema occurring after an iatrogenic hypopharyngeal injury. 2. Case report A 72-year-old, 68 kg male was admitted with a history of progressive tingling sensations in all four limbs for the past 5 years and urinary incontinence for the past 6 months. The patient was known to have coronary artery disease and had undergone angioplasty 12 years earlier. His hypertension was controlled with oral amlodipine, atenolol, and nitroglycerin. He also had hypothyroidism that was managed with oral tri-iodothyronine supplementation. An MRI of the cervical spine revealed a prolapsed intervertebral disc (PIVD) at C3–C4 and C4–C5 with osteophytes (Fig. 1). We planned an anterior approach discectomy (from the right side) and artificial cervical disc (Discover, DePuy Spine Inc, Raynham, MA, USA) placement under general anaesthesia. We
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Fig. 2. Axial CT scan of swallow showing (A) the presence of air within the soft tissue of the neck and (B) oral contrast leaking into the prevertebral space. R = right.
Fig. 1. Sagittal T2-weighted MRI of the cervical spine showing a significant prolapsed intervertebral disc at C3–C4 and C4–C5, with osteophytes.
decided to perform arthroplasty in order to prevent adjacent disc disease. Anaesthesia was induced with thiopentone 150 mg and fentanyl 100 lg. Fiberoptic orotracheal intubation was facilitated with rocuronium 50 mg. Anaesthesia was maintained with isoflurane in an O2/N2O mixture combined with intermittent boluses of fentanyl and rocuronium. During surgery, a cervical retractor was placed to better visualize the spinal canal. It was released intermittently to prevent ischaemic injury to the adjacent soft tissues. The operation lasted for 5 hours and was uneventful. At the end of surgery, the residual neuromuscular blockade was reversed and the trachea was extubated. The patient was moved to the neurosurgical intensive care unit with a Philadelphia collar placed around the neck. One hour later, the patient complained of breathing difficulty and his neck collar was removed. A gross swelling was observed in the patient’s neck that extended from the surgical site to his upper chest and the other side of his neck. Crackling on palpation confirmed the presence of subcutaneous emphysema. The patient was given oxygen via a facemask and we prepared to perform a ‘difficult airway drill’ if necessary. We sought the opinion of a laryngologist as a tracheal injury was suspected. A direct laryngoscopy revealed an injury at the right hypopharynx. A CT scan after swallow of oral omnipaque was performed, revealing air within the soft tissue of the cheeks, neck, and parapharyngeal and prevertebral spaces. Contrast leak-
age was seen from the hypopharynx at the C4–C5 vertebral level into the prevertebral space and soft tissue of neck (Fig. 2). The patient was managed conservatively for possible spontaneous resolution. Although his respiratory problems improved, the hypopharyngeal injury remained unhealed after 2 weeks. Hence, re-exploration of the neck with debridement and repair of the defect was performed under general anaesthesia. The remainder of his 1-month hospital stay was remarkable for pseudopharyngocele formation at the operative site, which was resolved by applying an external pressure bandage for 4 days. At the 3-month follow-up, the patient’s preoperative symptoms and postoperative complications had resolved.
3. Discussion Hypopharyngeal injury after anterior cervical discectomy is a rare, but potentially life-threatening, complication. There are few reports of delayed pharyngeal injury occurring as a result of displaced bone graft or fixation devices.3,4 We report the first patient with hypopharyngeal injury after ACDF and artificial cervical disc placement who presented with breathing difficulty. The cause of his hypopharyngeal injury was attributed to direct trauma by sharp instruments used during extensive dissection and retractor (blade) application at a higher cervical level (C3–C4 and C4–C5 disc spaces) and required for artificial disc placement. A retractor was used to better visualize the anterior spinal column. It has been hypothesized that surgical manipulations above C4 which often lie underneath the mandible are more likely to cause pharyngeal injuries.5 Patients with pharyngeal and oesophageal injuries may present with pain and acute dysphagia.4 Our patient complained of dyspnoea in the postoperative period due to increasing subcutaneous emphysema and the presence of a tight neck collar. A rigid
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cervical collar can interfere with lymphatic and venous drainage of postoperative edematous tissue after ACDF, thereby causing airway obstruction.6 This phenomenon has been described as an acute ‘superior vena cava syndrome’. Although our patient’s respiratory difficulty decreased following removal of the neck collar, it persisted due to the presence of extensive subcutaneous emphysema. Subcutaneous emphysema commonly occurs following intubation, mechanical ventilation, and head and neck surgery7; it may also occur after trauma to the chest or aerodigestive tract if air is driven into the subcutaneous layer through the wound site. In our patient, air may have entered via the injury site at the right hypopharynx, which communicates to the sublingual and submandibular spaces. The sublingual space further communicates with the pterygomandibular, parapharyngeal, retropharyngeal and prevertebral spaces, facilitating the spread of air into these areas. Often, the subcutaneous emphysema course is benign and symptoms subside after 2–7 days with conservative management. In our patient, although his emphysema resolved over a period of time, the persistent hypopharyngeal injury formed a track in the soft tissue of his neck, requiring surgical re-exploration. As the injury was small, and without any symptoms of mediastinitis, the patient was managed conservatively. Possibly, an earlier exploration of the injury could have reduced the duration of the hospital stay.
4. Conclusion We present a patient with a hypopharyngeal injury that was complicated by subcutaneous emphysema and breathing problems. Optimum surgical precautions must be taken to prevent these complications. Anticipation of likely complications is necessary in order prevent life-threatening consequences, especially when extensive surgical exposure of the anterior spinal canal at or above C4 is required. References 1. Robinson RA, Smith GW. Anterolateral cervical disc removal and interbody fusion of cervical disc syndrome. Bull Johns Hopkins Hosp 1955;96:223–4. 2. Fountas KN, Kapsalaki EZ, Nikolakakos LG, et al. Anterior cervical discectomy and fusion associated complications. Spine (Phila Pa 1976) 2007;32:2310–7. 3. Kelly MF, Spiegel J, Rizzo KA, et al. Delayed pharyngoesophageal perforation: a complication of anterior spine surgery. Ann Otol Rhinol Laryngol 1991;100: 201–5. 4. Sharma RR, Sethu AU, Lad SD, et al. Pharyngeal perforation and spontaneous extrusion of the cervical graft with its fixation device: a late complication of C2– C3 fusion via anterior approach. J Clin Neurosci 2001;8:464–8. 5. Sagi HC, Beutler W, Carroll E, et al. Airway complications associated with surgery on the anterior cervical spine. Spine 2002;27:949–53. 6. Kreisler NS, Durieux ME, Spiekermann BF. Airway obstruction due to a rigid cervical collar. J Neurosurg Anesthesiol 2000;12:118–9. 7. Yang SC, Chiu TH, Lin TJ, et al. Subcutaneous emphysema and pneumomediastinum secondary to dental extraction: a case report and literature review. Kaohsiung J Med Sci 2006;22:641–4.
doi:10.1016/j.jocn.2010.01.020
Neuromyelitis optica associated with subacute human T-lymphotropic virus type 1 infection Stéphane Olindo a,*, Mickaël Bonnan a, Harold Merle b, Aïssatou Signate a, Didier Smadja a, Philippe Cabre a a b
Neurology Department, Centre Hospitalier Universitaire de Fort-de-France, Hôpital Pierre Zobda Quitman, BP 632, Fort-de-France 97261, Martinique, France Opthalmology Department, Centre Hospitalier Universitaire de Fort-de-France, Hôpital Pierre Zobda Quitman, Fort-de-France, Martinique, France
a r t i c l e
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Article history: Received 17 August 2009 Accepted 16 December 2009
Keywords: HAM/TSP HTLV-1 Neuromyelitis optica Optic neuritis
a b s t r a c t Although human T-lymphotropic virus type 1 (HTLV-1)-associated myelopathy, or tropical spastic paraparesis (HAM/TSP), is usually considered as a progressive myelopathy, a subacute variant has been described. It is unusual for optic neuritis (ON) to be associated with an HTLV-1 infection. Neuromyelitis optica (NMO) is characterised by severe attacks of acute transverse myelitis and ON of unknown aetiology. We report a 61-year-old Afro-Caribbean male patient with subacute HAM/TSP associated with bilateral ON that occurred 5 years previously. To our knowledge this is the first report of recurrent NMO syndrome associated with HTLV-1 infection.
1. Introduction Neuromyelitis optica (NMO) is an inflammatory syndrome of the central nervous system characterised by severe attacks of acute transverse myelitis and optic neuritis (ON) of unknown aetiology.1 Human T-lymphotropic virus-1 (HTLV-1)-associated myelopathy/ tropical spastic paraparesis (HAM/TSP) is usually a slowly progressive spastic paraparesis with bladder disturbances.2 Subacute progression has been reported, particularly in patients posttransfusion and post-transplantation.3 Although ocular manifesta-
* Corresponding author. Tel.: +33 596 596552261; fax: +33 596 596758442. E-mail address:
[email protected] (S. Olindo).
Ó 2010 Elsevier Ltd. All rights reserved.
tions such as uveitis, dry kerato-conjunctivitis and interstitial keratitis are common in HTLV-1 infection,4 ON is unusual.5 NMO has been associated rarely with viral infections such as hepatitis A,6 HIV-17 or dengue arbovirus.8 To our knowledge, this is the first report of NMO associated with HTLV-1 infection.
2. Case report A 61-year-old Afro-Caribbean man was admitted to the University Hospital of Fort-de-France (in the French West Indies) for a rapidly progressive paraplegia. He suffered from diabetes mellitus, which was treated with oral hypoglycaemics, and had experienced
