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Annals of Indian Academy of Neurology - October-December 2007

Annals of Indian Academy of Neurology

Annals of Indian Academy of Neurology is indexed/listed with Expanded Academic ASAP, Genamics Journal Seek, Health Reference Center Academic, DOAJ, MANTIS.The journal is offi cial publication of the Indian Academy of Neurology, India. Issues are published quarterly in the last week of March, June, September and December. Journal aims to publish articles in clinical neurology, related disciplines and basic neurosciences, to serve as a medium for dissemination of information and contribute to the ad-vancement of knowledge in neuroscienc-es. Journal follows the broad guidelines for good publications practice brought out by the Committee on Publication Ethics (COPE) and recommendations of World Association of Medical Editors.All the rights are reserved. Apart from any fair dealing for the purposes of research or private study, or criticism or review, no part of the publication can be reproduced, stored, or transmitted, in any form or by any means, without the prior permission of the Editor, Annals of Indian Academy of Neurology.Annals of Indian Academy of Neurology and/or its publisher cannot be held responsible for errors or for any con-sequences arising from the use of the information contained in this journal.The appearance of advertising or product information in the various sec-tions in the journal does not constitute an endorsement or approval by the journal and/or its publisher of the quality or value of the said product or of claims made for it by its manufacturer.The journal is published and distributed by Medknow Publications. Copies are sent to subscribers directly from the publisher’s address. It is illegal to acquire copies from any other source. If a copy is received for personal use as a member of the association/society, one can not resale or give-away the copy for commercial or library use.The Journal is printed on acid free paper.Editorial Offi ceSanjeev V. ThomasRoom 1409Department of NeurologySCTIMST, Trivandrum - 695011. IndiaEmail: editor@annalsofi an.orgPublished byMedknow Publications,A-108/109 Kanara Business Centre, Ghatkopar (E), Mumbai - 400075, India. Websiteshttp://www.annalsofi an.orghttp://www.journalonweb.com/aian

ISSN 0972-2327

Mathew Alexander, Vellore, India

Kameshwar Prasad, New Delhi, India

N. C. Borah, Gawahati, India

Geoffrey A. Donnan, Melborne, Australia

Sarosh M. Katrak, Mumbai, India

Venkateswarlu Kolichana, Vishakhapattanam, India

Mohan Madhusudanan, Kottayam, India

U. K. Misra, Lucknow, India

JMK Murthy, Hyderabad, India

Editorial Board - 2007

EDITORSanjeev V. Thomas

Apoorva Pauranik, Indore, India

Sudesh Prabhakar, Chandigarh, India

Sridharan Ramaratnam, Chennai, India

Trishit Roy, Kolkata, India

Aline JC Russell, Glasgow, UK

S. H. Subramony, Jackson, USA

Arun B. Taly, Bangalore, India

William H. Theodore, Bethesda, USA

Torbjörn Tomson, Stockholm, Sweden

Past Editors

M. Gourie Devi (1997-2002)

D. Nagaraja (2003-2005)

Asha KishoreAbraham KuruvillaP. S. Mathuranath

Muralidharan NairDinesh Nayak

Kurupath Radhakrishnan

Editorial Advisory Committee

INDIAN ACADEMY OF NEUROLOGY

President

R. S. Wadia (Pune)

Vice President

Madhuri Behari (New Delhi)

Past Presidents

Ambar Chakravarty (Kolkata)

S. Katrak (Mumbai)

C. Sarada

Associate Editors

Secretary

M. M. Mehndiratta (New Delhi)

Treasurer

P. Satish Chandra (Bangalore)

Executive Members

Arabinda Mukhkerji (Kolkata)

Ashok Panagariya (Jaipur)

C. M. Meshram (Nagpur)

Rakesh Shukla (Lucknow)

279


Annals of Indian Academy of NeurologyCONTENTS

Volume 10 - Issue 4 - October-December 2007

EDITORIAL

From muscular dystrophy to chickenpoxSanjeev V. Thomas ............. 197

PRESIDENTIAL ORATION

A neurotropic virus (chikungunya) and a neuropathic aminoacid (homocysteine)R. S. Wadia ............. 198

REVIEW ARTICLES

Limb girdle muscular dystrophies: The clinicopathological viewpointJ. Andoni Urtizberea, France Leturcq ............. 214What are relative risk, number needed to treatand odds ratio? Kameshwar Prasad ............. 225Neuromyelitis opticaAnu Jacob, Mike Boggild ............. 231

ORIGINAL ARTICLES

Neurological complications of chickenpoxA. S. Girija, M. Rafeeque, K. P. Abdurehman ............. 240Patterns and predictors of in-hospital aneurysmalrebleed: An institutional experience andreview of literatureGirish Ramachandran Menon, Suresh Nair, Ravi Mohan Rao, Mathew Abraham, H. V. Easwer, K. Krishnakumar ............. 247

SHORT COMMUNICATIONS

Serial nerve conduction studies of the tail of rhesus monkey (Macaca mulatta) and potential implications for interpretation of human neurophysiological studiesWilliam A. Graham, Richard Goldstein, Mansfi eld Keith, Shanker Nesathurai ............. 252Wilson’s disease: A study of 21 cases fromnorth-west IndiaAshok Panagariya, Rajender Kumar Sureka, Anjani Kumar Sharma, Amit Dev, Neeraj Agarwal ............. 255

CASE REPORTS

Niemann-Pick disease Type C - Sea-blue histiocytosis: Phenotypic and imaging observations and mini reviewK. S. Praveen, S. Sinha, T. C. Yasha, U. B. Muthane, S. Ravishankar, S. Sangeetha, K. T. Shetty, A. B. Taly ............. 259Why we missed an early diagnosis of cerebral aspergilloma: Lesson from a caseD. Goel, K. K. Bansal, C. Gupta, S. Kishor, R. K. Srivastav, S. Raghuvanshi, S. Behari ............. 263Reversal of acquired immunodefi ciency syndrome-dementia complex with antiretroviral therapyIyer Kamalam, S. R. Daga, Naresh Tayade ............. 266Kleine-Levin syndrome in tubercular meningitisAnup K. Thacker, Anupam Aeron, Jamal Haider, K. M. Rao ............. 270

IMAGES IN NEUROLOGY

Bilateral simultaneous hypertensive intracerebral hemorrhage in both putamenRavouf Parvez Asimi, Mushtaq Ahmad Wani, Feroze Ahmad ............. 272

LIGHTER MOMENTS ............. 273LETTERS TO EDITOR

Neurology of consciousness: Need for Indian impetus Ravi Prakash, Shashi Prakash ............. 274More collaborative studies: The need of the hour in IndiaSanjeev V. Thomas ............. 275

AUTHOR INDEX - 2007TITLE INDEX - 2007

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Why we missed an early diagnosis of cerebral aspergilloma: Lesson from a case

D. Goel, K. K. Bansal, C. Gupta, S. Kishor, R. K. Srivastav, S. Raghuvanshi, S. Behari1

Himalayan Institute of Medical Sciences, Swami Ram Nagar, Dehradun, Uttaranchal,1Sanjay Gandhi PGI, Lucknow, India

Abstract

Intracranial aspergilloma is a dreadful CNS infection with nonspecific clinical manifestation and radiological features. Therefore, delay in the diagnosis is common, resulting in a fatal outcome in almost all the cases. We present how the diagnosis of this condition is overlooked if we do not follow the conventional medical rules of taking history and thorough preoperative evaluation. These clinicoradiological points can be of help in early diagnosis and better outcomes.

Keywords

Aspergillosis, early diagnosis, squash smear

For correspondence:Dr. Deepak Goel, Neurology Department, Himalayan Institute of Medical Sciences, Swami Ram Nagar,

Doiwala, Dehradun, Uttaranchal, India. E-mail: [email protected]

Ann Indian Acad Neurol 2007;10:263-5

Introduction

Aspergilloma is unusually considered as the diff erential of intracranial mass lesion in the preoperative phase.[1] Their presentation is subtle, oft en without any diagnostic characteristics and they are frequently mistaken for brain tumors.[2] Therefore, these cases are frequently encountered as a clinical surprise after histological diagnosis.

Many authors had drawn att ention about the diffi culties in diagnosis of this dreadful condition with nonspeciÞ c clinical and radiological Þ ndings.[3] The early suspicion of diagnosis is the only method to reduce the high mortality.[3]

We discuss how we failed to initiate a prospective diagnosis in two cases of aspergilloma as an etiology of cerebral mass lesion.

Case Reports

Case 1A 32-year-old male patient with headache, proptosis, diplopia and progressive Visual loss in left eye for last 3 months was referred to the neurosurgery department. Referral diagnosis was malignant glioma on the basis of brain CT scan Þ nding, showing a lesion in left temporal lobe occupying a large space with contrast enhancement

and perifocal edema [Figure 1]. Preoperative MRI was done by the neurosurgeon for better delineation of the lesion and planning of resection. The surgery was performed with left temporal craniotomy and a bony hard avascular lesion with well-deÞ ned margins from the medial to middle temporal gyrus was observed. The tumor was att ached to the wall of cavernous sinus. Squash smear per-operatively showed fungal hyphae and histological diagnosis of aspergilloma was Þ nalized [Figure 2].

Case 2The second patient was admitt ed at around 6 months

Figure 1: Contrast CT scan showing moderately enhanced mass in the left frontotemporal region with perifocal edema at time of referral

Case Report

264


aft er the Þ rst case. This 50-year-old male presented with headache and progressive loss vision for Þ ve months. This headache became worse over the time and aft er the onset of visual symptoms, a CT scan was performed outside that showed large mass in right frontal area. He was referred as malignant glioma. We performed MRI which showed an enhancing right basifrontal lesion with a radiological possibility of anaplastic astrocytoma. Surgery was performed with right frontal craniotomy and excision of tumor. There was solid grayish, Þ rm to hard nonsuckable mass with deÞ nite plane of cleavage. Squash smear showed fungal hyphae; later on, it was proved to be aspergillosis.

Two cases of aspergilloma were diagnosed at the interval of 6 months and both were missed preoperatively. The points we missed in favor of diagnosis were very simple related to history taking and clinical examination. Retrospective clinical and radiological analysis was done in both cases aft er histological diagnosis of aspergilloma. In the detailed histories, Þ rst case had measles 6 months back and got some treatment for nasal block and recurrent sinusitis for many time in past, while the second case underwent some nasal surgery for epistaxis 13 years back. Second, the nasopharyngeal examination was not thought to be important. Cerebrospinal ß uid (CSF) examination was not done as we were biased for glioma ignored involvement of paranasal air and large venous sinus on MRI [Figure 3]. Both the patients were nondiabetic and HIV serology was negative. We could not perform other work-up for the assessment of the immune status. Both the patients were administered with amphotericin in postoperative period and both of them died with in 6 months.

Discussion

Aspergillosis is the most common among the fungal

infections of CNS; however, on an average, the reporting rates suggest one case per year.[4] The predominant symptoms involve headache, vomiting and cranial-nerve-related symptoms, while the rare symptoms are fever, nasal congestion and seizures. Common signs included papilledema with cranial neuropathy (I, III/IV/VI and V in 4, 7 and 2 patients, respectively), hemiparesis and meningismus. Two-third cases found to have some predisposing immunocompromised state, diabetes being the commonest, while one-third cases have no predisposition. The most common site of involvement is the frontal area followed by the parasalar region.[4]

Our cases are the best examples to reemphasize that taking the clinical history and examination is irreplaceable by technology for reaching a correct diagnosis. Although our patients had a number of clinicoradiological features indicating towards the diagnosis of fungal infection, we could only notice them aft er histological diagnosis.

Paranasal infections in the past and the topical use of decongestant with steroids might play an important role in predisposing fungal growth. Existing literature have no account for the history of old ENT problems in reported cases; however, radiologically, 40% patients have evidences of paranasal sinus involvement.[4] Fungal infection can directly extend intracranially from the paranasal sinuses.[5-7] Therefore, at times, intracranial lesions have continuation with paranasal sinuses. The proximity of cranial lesion to the nasal sinus is another point that we had failed to notice in our evaluation. Both the patients had abnormal intensity in the paranasal sinuses and lesions were in close proximity to these sinuses.

The MRI signal characteristics in aspergilloma were compared with the histologic Þ ndings. Irregular low-signal zones were demonstrated between the wall of the

Figure 3: T1W post-contrast MRI image showing the involvement of paranasal sinuses, cavernous sinus orbit and left temporal lobe

Figure 2: Squash smear showing hyphae of aspergillus branching at acute angle (H&E ×200)

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265


abscess and the central necrosis on T2-weighted images; the pathology specimen revealed concentrated iron in these transitional zones, but no hemosiderin. Iron is an essential element for the growth of fungal hyphae. The low-signal zones may represent the areas where there was active proliferation of Aspergillus and the unique location of the low signal may be a helpful imaging characteristic for the diagnosis of abscess caused by Aspergillus.[8] This MRI Þ nding was nicely demonstrated in our cases also [Figure 4].

Large venous sinus thrombosis is the common cause of presentation in fungal infection of CNS. The involvement of superior sagitt al and cavernous sinus is common.[9] This point also requires special att ention during the MRI evaluation of these patients. All our patients had venous sinus involvement.

We realized from our errors that for the cases with intracranial mass lesions, involving paranasal and venous sinuses and positive nasal problem in past,

a detailed nasopharyngeal and paranasal evaluation is more important than rushing for the surgery. This may give the diagnosis in the preoperative stage and the treatment with antifungal drugs can be started early. The opening of vascular channels and the use of corticosteroids in the postoperative period makes the prognosis worst in undiagnosed cases.

The Þ nal conclusion is that every patient with intracranial space occupying lesion should be subjected to a systemic approach of clinical history and examination rather than rushing to the operation theater.

References

1. Alapatt JP, Kutty RK, Gopi PP, Challissery J. Middle and posterior fossa aspergilloma. Surg Neurol 2006;66:75-9.

2. Nadkarni T, Goel A. Aspergilloma of the brain: An overview. J Postgrad Med 2005;51:37-41.

3. Fardoun R, Rao NK, Miskeen AK. Cerebral aspergilloma: Review of the literature apropos of a case. Neurochirurgie 1990;36:45-51.

4. Dubey A, Patwardhan RV, Sampth S, Santosh V, Kolluri S, Nanda A. Intracranial fungal granuloma: Analysis of 40 patients and review of the literature. Surg Neurol 2005;63:254-60.

5. Coulthard A, Gholkar A, Sengupta RP. Case report: Frontal aspergilloma: A complication of paranasal aspergillosis. Clin Radiol 1991;44:425-7.

6. Wilms G, Lammens M, Dom R, Boogaerts M, Marchal G, Demaerel P, et al. MR imaging of intracranial aspergilloma extending from the sphenoid sinus in an immunocompromised patient with multiple myeloma. J Belge Radiol 1992;75:29-32.

7. Swoboda H, Ullrich R. Aspergilloma in the frontal sinus expanding into the orbit. J Clin Pathol 1992;45:629-30.

8. Yamada K, Zoarski GH, Rothman MI, Zagardo MT, Nishimura T, Sun CC. An intracranial aspergilloma with low signal on T2-weighted images corresponding to iron accumulation. Neuroradiology 2001;43:559-61.

9. Goel A, Nadkarni T, Desai AP. Aspergilloma in the paracavernous region-two case reports. Neurol Med Chir (Tokyo) 1996;36:733-6.

Figure 4: T2W MRI showing heterogeneous mass with markedly hypointense areas medially toward sinuses probably due to high iron content

Received: 26-04-07, Revised: 11-06-07, Accepted: 12-07-07

Source of Support: Nil, Confl ict of Interest: Nil

Goel, et al.: Early diagnosis of cerebral aspergilloma

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