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Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. References [1] Liubinas SV, Maartens N, Drummond KJ. Primary melanocytic neoplasms of the central nervous system. J Clin Neurosci 2010;17:1227–32. [2] Ramaswamy V, Delaney H, Haque S, et al. Spectrum of central nervous system abnormalities in neurocutaneous melanocytosis. Dev Med Child Neurol 2012;54:563–8. [3] Arsene D, Ardeleanu C, Balescu C, et al. Meningeal melanocytosis in a young patient–an autopsy diagnosis. Clin Neuropathol 2007;26:294–8. [4] Garg RK. Tuberculosis of the central nervous system. Postgrad Med J 1999;75:133–40. [5] Thompson 3rd GR, LaValle 3rd CE, Everett ED. Unusual manifestations of histoplasmosis. Diagn Microbiol Infect Dis 2004;50:33–41. [6] Mathison G, Shelub A, Truong J, et al. Coccidioidal meningitis: clinical presentation and management in the fluconazole era. Medicine (Baltimore) 2010;89:251–84.

[7] Bariola JR, Perry P, Pappas PG, et al. Blastomycosis of the central nervous system: a multicenter review of diagnosis and treatment in the modern era. Clin Infect Dis 2010;50:797–804. [8] Visvesvara GS, Moura H, Schuster FL. Pathogenic and opportunistic free-living amoebae: Acanthamoeba spp., Balamuthia mandrillaris, Naegleria fowleri, and Sappinia diploidea. FEMS Immunol Med Microbiol 2007;50:1–26. [9] Reske D, Petereit HF, Heiss WD. Difficulties in the differentiation of chronic inflammatory diseases of the central nervous system – value of cerebrospinal fluid analysis and immunological abnormalities in the diagnosis. Acta Neurol Scand 2005;112:207–13. [10] Chamberlain MC. Leptomeningeal metastasis. Curr Opin Oncol 2010;22:627–35. [11] Kaplan JG, DeSouza TG, Farkash A, et al. Leptomeningeal metastases: comparison of clinical features and laboratory data of solid tumors, lymphomas and leukemias. J Neurooncol 1990;9:225–9. [12] van Oostenbrugge RJ, Twijnstra A. Presenting features and value of diagnostic procedures in leptomeningeal metastases. Neurology 1999;53:382–5. [13] Sutton BJ, Tatter SB, Stanton CA, et al. Leptomeningeal melanocytosis in an adult male without large congenital nevi: a rare and atypical case of neurocutaneous melanosis. Clin Neuropathol 2011;30:178–82.

http://dx.doi.org/10.1016/j.jocn.2013.10.002

Salivoma after carotid endarterectomy Asif Bashir a,1, Mohammed Hussain a,1, Haitham Dababneh a, Deborah Rosin b, Aaron A. Cohen-Gadol c,⇑ a

New Jersey Neuroscience Institute, Department of Neurosurgery, JFK Medical Center, Seton Hall University, Edison, NJ, USA Department of ENT, JFK Medical Center, Edison, NJ, USA c Goodman-Campbell Brain and Spine, Department of Neurological Surgery, Indiana University School of Medicine, 355 W. 16th Street, Suite 5100, Indianapolis, IN 46202, USA b

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Article history: Received 18 September 2013 Accepted 19 October 2013

Keywords: Complications Endarterectomy Salivary glands Salivoma

a b s t r a c t Although the diagnosis and management of postoperative or traumatic fluid collections have been documented extensively in the literature, to our knowledge the occurrence of a salivoma after carotid endarterectomy has not been reported. We report an extra salivary glandular collection of saliva – a ‘‘salivoma’’ – in a 79-year-old patient who underwent a carotid endarterectomy with a high carotid bifurcation. He presented with serous watery drainage from the incision site that had started spontaneously 4 days after surgery. The patient was taken to the operating room for exploration and washout of the wound with presumption of an infectious source. As self-retaining retractors were placed under the platysma, a large release of serous fluid occurred. Copious irrigation allowed complete washout of the wound. On postoperative day 2, the patient re-exhibited neck wound fullness and a Penrose drain was placed in the incision with clear serous fluid flowing through the drain. The patient was given a scopolamine patch to decrease salivary secretions. Within 5 days, the drainage significantly decreased and the drain was removed. This diagnosis should be included in the differential diagnosis of an expanding neck mass following carotid endarterectomy to properly treat this complication. Ó 2013 Elsevier Ltd. All rights reserved.

1. Introduction The literature abounds with studies on the formation and treatment of postoperative fluid collections such as hematomas and seromas. They usually arise from insufficient wound healing or mechanical failure at the surgical site [1]. The otolaryngology literature also describes formation of ‘‘sialoceles’’ or subcutaneous cavities composed of saliva, usually resulting from trauma or infection of the parotid gland parenchyma, laceration of the parotid duct, or ductal stenosis with subsequent dilatation [2]. Characterized as swelling around the parotid region, these lesions are soft and mobile with patients experiencing no pain, fever, chills, or erythema unless secondarily infected [3]. These sialoceles usually result from trauma such as sharp penetrating wounds in the oral cavity or face or may be secondary to blunt trauma, such as zygomatic and man⇑ Corresponding author. Tel.: +1 317 362 8760; fax: +1 317 924 8472. 1

E-mail address: [email protected] (A.A. Cohen-Gadol). These authors have contributed equally to the manuscript.

dible fractures [4,5]. The history usually includes facial trauma or surgery days or weeks before the onset of swelling [2]. Extra-salivary pocket formations of saliva in patients who have undergone vascular procedures have seldom been reported. We report a patient after carotid endarterectomy with a high carotid bifurcation who developed a well-demarcated extra salivary glandular pocket of saliva or ‘‘salivoma’’. Diagnosis of salivoma after carotid endarterectomy is complex and requires clinical suspicion and a thorough history and assessment. Proper management involves initiating prophylactic antibiotics along with adequate drainage to prevent wound dehiscence and infection.

2. Case report A 79-year-old right-handed man with a history of coronary stents on Plavix (Bristol-Myers Squibb, New York, NY, USA) presented with acute onset of mild left hemiparesis and hemisensory loss. Brain MRI showed small multifocal cortical infarcts in the

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Fig. 1. (A) Diffusion-weighted axial MRI showing small multifocal cortical infarcts at the right posterior frontal convexity. (B) CT angiography of the neck showing greater than 70% stenosis at the right internal carotid artery origin. There is a high carotid bifurcation almost at the level of C2–3.

right posterior frontal convexity (Fig. 1A). MRI and CT angiograms demonstrated greater than 70% right internal carotid artery stenosis with ulcerated plaque and a high carotid bifurcation (Fig. 1B). This patient subsequently underwent a right carotid endarterectomy and the carotid bifurcation was exposed near the undersurface of mandible. No injury of the parotid or submandibular glands was noted. Under loop magnification, plaque was removed from the internal carotid artery (Fig. 2), followed by primary arteriotomy closure. The patient was discharged home the next day without complication. Five days later, the patient returned with complaints of a serous watery drainage from the superior margin of the incision site that had started 4 days after surgery. He denied fevers, chills, and jaw or mandibular pain. He was taken to the operating room for exploration and washout of the wound with presumption of an infectious source. As self-retaining retractors were placed under the platysma, a sudden release of copious serous fluid occurred. Liberal irrigation allowed complete washout of the wound. Infectious disease consultation and the results of the gram stain and cultures revealed no infectious etiology. On postoperative day 2, the patient re-exhibited neck wound fullness, but no active discharge. An ear, nose and throat (ENT) consultation was obtained and a Penrose drain was placed in the lower incision site, with clear serous fluid flowing through the drain. The

patient was started on a scopolamine patch to decrease salivary secretions. Within the next 3–5 days, the patient reported significant decrease in drainage and the drain was removed. At the 2 week follow-up, the patient did not report drainage or swelling (Fig. 3). He received oral antibiotics for 2 weeks postoperatively and did not suffer another complication.

Fig. 2. Intraoperative picture of the right internal carotid ulcerated plaque.

Fig. 3. Postoperative incision site 2 weeks following treatment of the salivoma.

3. Discussion Fluid collections such as hematomas and seromas are the most frequent complications of surgery, including endarterectomy. They infrequently require drainage or aspiration and usually resolve

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spontaneously. Seromas are typically seen after breast and axillary surgery [6]. Risk factors for seroma formation include obesity, extensive surgery, and the use of electrocautery for skin flap dissection [7–9]. Both hematomas and seromas can lead to wound dehiscence and infection [10,11]. Examination of the wound can provide clues about the character of the fluid collection, and ultrasound or CT scan can help identify these pockets. We had meticulously dissected the soft tissues during exposure of the high bifurcating carotid endarterectomy in this patient to avoid damage to the parotid or submandibular glands. Nonetheless, it is likely that retraction injury led to indirect damage to these glands, causing salivoma formation. Since the area of drainage involved the right aspect of the neck, vigorous or even passive motion of the head might have contributed to salivoma formation. If a salivoma is suspected after exclusion of an infectious etiology, we recommend placement of a Penrose drain and avoidance of unnecessary exploratory surgery. Early ENT consultation will assist with management of salivomas. Conflicts of Interest/Disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication.

References [1] Dodson MK, Magann EF, Meeks GR. A randomized comparison of secondary closure and secondary intention in patients with superficial wound dehiscence. Obstet Gynecol 1992;80:321–4. [2] Bater MC. An unusual case of preauricular swelling: a giant parotid sialocele. Int J Oral Maxillofac Surg 1998;27:125–6. [3] Canosa A, Cohen MA. Post-traumatic parotid sialocele: report of two cases. J Oral Maxillofac Surg 1999;57:742–5. [4] Burch RJ. Spontaneous closure of a parotid gland fistula with the aid of banthine; report of a case. Oral Surg Oral Med Oral Pathol 1953;6:1191–4. [5] Hutchison IL, Ryan D. A parotid fistula and sialocele complicating temporomandibular joint surgery. Br J Oral Maxillofac Surg 1989;27:203–8. [6] van Bemmel AJ, van de Velde CJ, Schmitz RF, et al. Prevention of seroma formation after axillary dissection in breast cancer: a systematic review. Eur J Surg Oncol 2011;37:829–35. [7] Bonnema J, Ligtenstein DA, Wiggers T, et al. The composition of serous fluid after axillary dissection. Eur J Surg 1999;165:9–13. [8] Petrek JA, Peters MM, Nori S, et al. Axillary lymphadenectomy. A prospective, randomized trial of 13 factors influencing drainage, including early or delayed arm mobilization. Arch Surg 1990;125:378–82. [9] Porter KA, O’Connor S, Rimm E, et al. Electrocautery as a factor in seroma formation following mastectomy. Am J Surg 1998;176:8–11. [10] Pogson CJ, Adwani A, Ebbs SR. Seroma following breast cancer surgery. Eur J Surg Oncol 2003;29:711–7. [11] Shamley DR, Barker K, Simonite V, et al. Delayed versus immediate exercises following surgery for breast cancer: a systematic review. Breast Cancer Res Treat 2005;90:263–71.

http://dx.doi.org/10.1016/j.jocn.2013.10.013

Behçet’s disease presenting as a peripheral vestibulopathy Alison S. White a, Rachael L. Taylor b, Celene McNeill c, Roger Garsia a, Miriam S. Welgampola a,⇑ a b c

Institute of Clinical Neuroscience, Royal Prince Alfred Hospital, Central Clinical School, University of Sydney, Missenden Road, Camperdown, NSW 2050, Australia Healthy Hearing and Balance Care, Sydney, NSW, Australia Department of Clinical Immunology, Royal Prince Alfred Hospital, Central Clinical School, University of Sydney, Camperdown, NSW, Australia

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Article history: Received 9 December 2012 Accepted 10 August 2013

Keywords: Behçet’s Hearing loss Vestibular

a b s t r a c t Prolonged acute spontaneous vertigo can be secondary to acute vestibular neuritis or posterior circulation ischaemia. We present a 66-year-old man who first developed an acute vestibular syndrome with profound unilateral hearing loss 34 years ago. First treated as vestibular neuritis, he subsequently developed manifestations of Behçet’s disease, including mouth ulcers, genital ulcers and erythema nodosum over a period of 10 years. Subsequently, sudden sensorineural hearing loss affecting his only hearing ear responded to immunomodulation, confirming an autoimmune cause for the audiovestibular symptoms. This report serves as a reminder that vestibular neuritis seldom causes hearing loss; ischaemic, infective and autoimmune causes should be sought when an acute vestibular syndrome is accompanied by hearing impairment. Ó 2014 Published by Elsevier Ltd.

1. Case report A 66-year-old man first presented in 1979 with acute spontaneous vertigo lasting 12 hours, right-sided hearing loss, tinnitus and imbalance. He had third degree left-beating spontaneous nystagmus. Bithermal caloric testing revealed a 78% right canal paresis. Audiometry revealed profound right sensorineural hearing loss. A diagnosis of vestibular neuronitis was made. His imbalance improved, yet the hearing loss was permanent. Ten years later, the patient developed mouth ulcers, genital ulcers and red macular coin-shaped lesions over both lower limbs,

⇑ Corresponding author. Tel.: +61 2 9515 8820; fax: +61 2 9515 8347. E-mail address: [email protected] (M.S. Welgampola).

which were consistent with erythema nodosum. A clinical diagnosis of Behçet’s disease was made. Recurrent ulcers were successfully treated with self-initiated oral thalidomide 100 mg twice daily for 3–4 days. A further 10 years later, he experienced a sudden left-sided hearing loss. He had a positive rightward bedside head impulse, left-beating head-shaking nystagmus and a rightward Unterberger test. Ice water caloric stimulation showed low amplitude responses for both ears with a persistent right canal paresis (33%) and video head impulse testing showed marked reduction in the vestibulo-ocular reflex gain for the right ear (0.3–0.6), with a shower of ‘‘catch up saccades’’ for rightward rotation and a small number of saccades for leftward rotation, implying a right, more than left horizontal canal dysfunction (Fig. 1A). Ocular and cervical vestibular evoked myogenic potentials (VEMP) to air and bone conducted sound were absent for right ear stimu-

Salivoma after carotid endarterectomy.

Although the diagnosis and management of postoperative or traumatic fluid collections have been documented extensively in the literature, to our knowl...
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