[Downloaded free from http://www.neurologyindia.com on Thursday, November 13, 2014, IP: 202.177.173.189]  ||  Click here to download free Android application for journal Letters to Editor

References 1.

Abdulrauf SI, Malik GM, Awad IA. Spontaneous angiographic obliteration of cerebral arteriovenous malformations. Neurosurgery 1999;44:280‑7. 2. Mabe H, Furuse M. Spontaneous disappearance of a cerebral arteriovenous malformation in infancy. Case report. J Neurosurg 1977;46:811‑5. 3. Krapf H, Siekmann R, Freudenstein D, Kuker W, Skalej M. Spontaneous occlusion of a cerebral arteriovenous malformation: Angiography and MR imaging follow‑up and review of the literature. AJNR Am J Neuroradiol 2001;22:1556‑60. 4. Hamada J, Yonekawa Y. Spontaneous disappearance of a cerebral arteriovenous malformation: Case report. Neurosurgery 1994;34:171‑3. 5. Mizutani T, Tanaka H, Aruga T. Total recanalization of a spontaneously thrombosed arteriovenous malformation: Case report. J Neurosurg 1995;82:506‑8. 6. Paillas JE, Berard M, Sedan R, Toga M, Alliez B. The relative importance of atheroma in the clinical course of arteriovenous angioma in brain. Prog Brain Res 1968;30:419‑25. Access this article online Quick Response Code:

Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.144500

Received: 28-09-2014 Review completed: 28-09-2014 Accepted: 28-09-2014

packed cell volume (PCV) 36% and thrombocytopenia with a platelet count 93,000/mm3 which further dropped to a low of 35,000/mm3 before convalescence. She was managed with intravenous fluids according to national guidelines. Once she was afebrile for more than 48 hours and platelet count was more than 50,000/mm3, anti‑platelet was started by the medical team. However, her symptoms of dysarthria and tongue deviation persisted. Therefore, the patient was referred to our otorhinolaryngology department for opinion. We proceeded further as no mass was seen or palpable on endoscopic examination of the nasopharynx, laryngopharnx, neck and oral cavity. Computed tomography (CT) scan and magnetic resonance imaging (MRI) of head and neck [Figure 2] excluded intracranial bleed or infarct but reported base of tongue mass. Magnetic resonance angiography (MRA) revealed no vascular pathology. She was managed expectantly for isolated right hypoglossal nerve palsy, reassured and was on our monthly follow‑up. The patient showed gradual improvement of swallowing and speech function over the next five months. Currently, her speech and swallowing is normal with tongue deviation to the right near normal [Figure 3]. This report highlights two interesting issues. Firstly, it reports a rare occurrence of isolated hypoglossal

Hypoglossal nerve palsy: A rare consequence of dengue fever Sir, Dengue is the most common arboviral disease caused by four antigenetically distinct dengue virus serotypes (DEN‑1 to DEN‑4). In recent years, neurological sequelae are being increasingly reported though exact incidence of various neurological complications is uncertain.[1] Hypoglossal nerve palsy usually appears as a sign rather than a symptom and the causes range from intracranial or extracranial space‑occupying lesion, head and neck trauma, vascular abnormality, infection and autoimmune diseases. [2] Herein, we report hypoglossal nerve palsy in a case of serologically confirmed dengue fever. A 53‑year‑old lady presented with sudden onset of mild dysarthria and difficulty in swallowing on day five of serologically confirmed dengue fever. Examination revealed deviation of tongue towards the left side at rest [Figure 1a] and deviation of tongue towards the right upon protrusion [Figure 1b] with normal pharyngeal sensation and bilateral gag reflex. Hematological studies showed total white blood cell count of 1800/mm3, hemoglobin of 12 g/dl, Neurology India | Sep-Oct 2014 | Vol 62 | Issue 5

a

b

Figure 1: (a) At presentation, deviation of tongue toward left side at rest (b) At presentation, deviation of tongue towards right upon protrusion

Figure 2: Image of MRI showing protrusion of right tongue into oropharynx

567

[Downloaded free from http://www.neurologyindia.com on Thursday, November 13, 2014, IP: 202.177.173.189]  ||  Click here to download free Android application for journal Letters to Editor

tongue onto oropharynx, variable fatty infiltration.[6] Therefore, the radiologist should be aware of various appearances of tongue denervation and not confuse it with base of tongue mass to avoid unnecessary biopsy in search of a tumor.

Shantini Jaganathan, Rajagopalan Raman Department of Otorhinolaryngology, Faculty of Medicine, University Malaya, Kuala Lumpur, Malaysia E‑mail: [email protected]

References 1. Figure 3: After 5 months, tongue deviation to right near normal

nerve palsy following dengue infection. Secondly, the imaging studies of the patient were misinterpreted as tongue base mass and literature search also report similar mistakes. The pathogenesis of neurological manifestations of dengue include: Direct neurotropic effect of dengue virus, related to systemic or metabolic complication of dengue or immune mediated.[1] Literature search shows demyelinating type of conduction defects with axonal components on nerve conduction studies also has been associated with monoenuropathies following dengue.[3] Thirteen cases of cranial palsy have been documented which included Bell’s palsy and long thoracic nerve palsies till 1996. [4] Since then, few more Bell’s palsy, diaphragmatic hernia secondary to phrenic nerve palsy.[3] lateral rectus palsy, occulomotor nerve.[5] palsy have been documented. Isolated unilateral hypoglossal nerve palsy is rare and the infective causes of hypoglossal nerve palsy reported to date are infectious mononucleosis streptococcal and even common cold.[2] In this patient, dengue serology was positive and all other causes for hypoglossal nerve palsy have been excluded.[2] Moreover, she gradually obtained near full recovery of her symptoms by five months just as most reported mononeuropathies associated with dengue have shown.[3,5] To date, there is no established treatment for mononeuropathies following dengue though expectant management has shown to have a favorable outcome.[3,5] This clear temporal relationship with dengue infection establishes the relationship between hypoglossal nerve palsy and dengue fever in our patient. Retrospective review of MRI belonging to seven patients of clinically and/or radiologically suspected tongue base mass were studied and all patients actually showed MRI findings typical of tongue denervation: T2‑weighted hyper intensity of involved hemitongue, protrusion of 568

Murthy JM. Neurological complication of dengue infection. Neurol India 2010;58:581‑4. 2. Yoon JH, Cho KL, Lee HJ, Choi SH, Lee KY, Kim SK, et al. A case of idiopathic isolated hypoglossal nerve palsy in a Korean child. Korean J Pediatr 2011;54:515‑7. 3. Ratnayake EC, Shivanthan C, Wijesiriwardena BC. Diaphragmatic paralysis: A rare consequence of dengue fever. BMC Infect Dis 2012;12:46. 4. Lam SK. Dengue infection with central nervous system manifestations. Neurol J Southeast Asia 1996;1:3‑6. 5. Shivanthan MC, Ratnayake EC, Wijesiriwardena BC, Somaratna KC, Gamagedara LK. Paralytic squint due to abducens nerve palsy: A rare consequence of dengue fever. BMC Infect Dis 2012;12:156. 6. Learned KO, Thaler ER, O’Malley BW Jr, Grady MS, Loevner LA. Hypoglossal nerve palsy missed and misintepreted: The hidden skull base. J Comput Assist Tomogr 2012;36:718‑24. Access this article online Quick Response Code:

Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.144501

Received: 09-08-2014 Review completed: 17-08-2014 Accepted: 09-10-2014

Post‑traumatic blepharocele: A rare manifestation of head injury Sir, Dural defect associated with skull base fracture following head injury may lead to cerebrospinal fluid (CSF) leak in the form of CSF rhinorrhea or otorrhea. In rare circumstances, it might lead to CSF leak and brain herniation into the orbit[1] (orbital encephalocele), lid[2,3] (blepharocele) or CSF lacrimation[4] (pseudo lacrimation) [Table 1]. There are very few case reports of blepharocele in literature. We report here an interesting case of blepharocele and review of literature. Neurology India | Sep-Oct 2014 | Vol 62 | Issue 5

Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.

Hypoglossal nerve palsy: a rare consequence of dengue fever.

Hypoglossal nerve palsy: a rare consequence of dengue fever. - PDF Download Free
730KB Sizes 3 Downloads 7 Views