G Model NEUCHI-1008; No. of Pages 15 ARTICLE IN PRESS Neurochirurgie xxx (2019) xxx–xxx Disponible en ligne sur ScienceDirect www.sciencedirect.com Short clinical case Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis A. Vienne a,b , R. Dulou b,c , F. Bielle d , M. Baruteau e , F.-L. Maison b , Y. Nikolova Yordanova b,∗ a Defense Health Service, Cognition and Action Group, Cognac-G, CNRS UMR 8257, Paris Descartes University, 45, rue des Saints-Pères, 75006 Paris, France Department of Neurosurgery, ‘Percy’ Military Hospital, 101, avenue Henri-Barbusse, 92140 Clamart, France c Val de Grâce Military Medical Corps Academy, 74, boulevard du Port-Royal, 75005 Paris, France d Department of Neuropathology, Pitié-Salpêtrière Hospital, 41-83, boulevard de l’Hôpital, 75013 Paris, France e Department of Neurology, ‘Percy’ Military Hospital, 101, avenue Henri-Barbusse, 92140 Clamart, France b a r t i c l e i n f o Article history: Received 11 May 2019 Received in revised form 5 July 2019 Accepted 28 July 2019 Available online xxx Keywords: Awake surgery Cystic lesion Differential diagnosis Neurocysticercosis Tuberculoma Tumor a b s t r a c t Differential diagnosis of isolated single neurocysticercosis can be difficult, and management is controversial. We report here an original surgical strategy, and review previous studies reporting misdiagnosis, using the PRISMA guidelines. A 24-year-old man was admitted to our hospital for recent memory impairment, hypoesthesia of the right hand, and recurrent focal seizures without loss of consciousness. Brain MRI revealed a single ring-enhancing parenchymal lesion in the left superior postcentral gyrus, with large perilesional edema. Since exhaustive systemic exploration was negative, surgical resection of the lesion was decided on in a multidisciplinary team meeting. To preserve eloquent brain areas, surgery was performed in awake condition. It allowed complete resolution of clinical manifestations. The diagnosis of neurocysticercosis was confirmed on pathology. This case illustrates the utility of awake surgery in degenerating neurocysticercosis in functional areas, and emphasizes the importance of including it in differential diagnosis of cystic ring-enhancing brain lesions. © 2019 Elsevier Masson SAS. All rights reserved. 1. Introduction Single ring-enhancing parenchymal lesions of the central nervous system have multiple differential diagnoses, including degenerating neurocysticercosis and tuberculoma. Differentiation between these two conditions can be a real dilemma: many cases of neurocysticercosis are seronegative, being already degenerated or because antibody response is not high enough, while many cases of tuberculoma are isolated, with no other mycobacteria sites [1–3]. Cystic brain metastases or primary tumor can also be incriminated, and misdiagnosis between neurocysticercosis and neoplasia has frequently been reported in the literature [4–43]. Managing such equivocal clinical conditions can be difficult, notably in case of large lesions located in eloquent brain areas. Surgery in awake condition can enable safe lesion resection and definitive diagnosis. Here we demonstrate, for the first time, the role of this technique in the ∗ Corresponding author. E-mail address: yn.yordanova@gmail.com (Y. Nikolova Yordanova). management of isolated neurocysticercosis. We also performed a review of studies investigating misdiagnosis. 2. Case report A 24-year-old soldier, native of Madagascar but living in France for 5 years, was admitted to the emergency department after recurrent aware focal seizures. The patient was right-handed, with no neurological history. He had had the first aware focal sensory seizure (paresthesia of the right upper limb, subsequently irradiating to the rest of the right hemi-body)two months earlier, with no triggering factor. On first examination, he had right hand hypoesthesia and was subfebrile (37.8 ◦ C), but without signs of meningitis. Brain MRI disclosed a solitary multilobular cystic ringenhancing lesion, located at the gray/white matter junction of the superior left postcentral gyrus. The center of the lesion was hypointense on T1-weighted images, hyperintense on T2-weighted images, with increased apparent diffusion coefficient, suggesting necrosis and ruling out pyogenic abscess. There was large perilesional edema and heterogeneous enhancement of the lesion periphery on the contrast-enhanced T1-weighted images. MR https://doi.org/10.1016/j.neuchi.2019.07.002 0028-3770/© 2019 Elsevier Masson SAS. All rights reserved. Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 G Model NEUCHI-1008; No. of Pages 15 2 ARTICLE IN PRESS A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Fig. 1. Preoperative MRI findings. A. T1-weighted imaging showing single lesion in the superior left postcentral gyrus. B. T1-weighted gadolinium-enhanced imaging showing irregular rim enhancement of the lesion. C. T2-weighted imaging showing a multilobular cystic structure of the lesion, and large perilesional edema. D. Diffusion-weighted imaging showing increased apparent diffusion coefficient of the lesion. E. MR perfusion-weighted imaging showing slightly increased perfusion of the lesion. F. Long Echo Time MR spectroscopy showing increased choline peak with preserved choline/N-acetyl aspartate ratio. spectroscopy found marked elevation of the lipid/lactate peak and depression of the creatine and N-acetyl aspartate peaks. MR perfusion-weighted imaging revealed slight neo-angiogenesis, which was difficult to interpret because of the proximity of the brain cortex. Altogether, the MRI aspect of the lesion was rather in favor of non-pyogenic infection, but a neoplastic lesion was not entirely ruled out (see Fig. 1). Daily antiepileptic treatment was started and evidence of parasitic infection, systemic disease or secondary brain tumor was explored. Thoraco-abdominopelvic CT did not find any progressive Fig. 2. Intraoperative photographs and gross examination of the lesion. A. Photograph of the brain surface, taken after cortical mapping and before lesion resection. The lesion is visible on the cortical surface as whitish area (arrow). Direct electrical stimulation led to dysarthria (tag 5), speech arrest/negative motor response of the right upper limb (tags 3 and 10), arrest of the fine motor skills of the right hand (tags 2 and 7), and paresthesia of the right hand (tags 1 and 4). B. Photograph, taken after lesion resection, showing the previously identified cortical functional areas and the responsive sites found at the bottom of the surgical cavity. Paresthesia and arrest of the regular movement of the right arm were systematically induced by the stimulation of each of the subcortical eloquent sites indicated by sterile number tags. C. Macroscopic aspect of the removed parasitic abscess. D. Gross examination of the abscess after cutting in half. A thick wall surrounds cystic cavity, filled with gelatinous content. AM: anteromedial; PL: posterolateral. Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 G Model NEUCHI-1008; No. of Pages 15 ARTICLE IN PRESS A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx or infectious lesions. The results of all other investigations–lumbar puncture, ophthalmologic examination and biological tests (blood culture and parasitological stool examination, serology for human immunodeficiency virus, hepatitis B or C virus, syphilis, taenia infection, and interferon-gamma-release assay) came back negative. Exhaustive neurocognitive assessment revealed mild deficits of visual spatial processing speed, working memory, executive functions and attention. Given the clinical manifestations, the atypical radiological features and impossibility of ruling out tumor, and the volume of the lesion, surgical resection was decided on. Because of the location of the lesion in the primary somatosensory area, surgery was performed in awake condition in order to detect, and thus conserve, functional cortical and subcortical structures. Once the craniotomy was completed and the dura mater opened, the lesion was partially visualized on the brain surface (see Fig. 2A). It was delineated on intraoperative ultrasound and landmarked with sterile empty tags. Direct electrical stimulation was then performed, using a bipolar electrode probe with 5-mm inter-electrode gap (NIMBUS Stimulator, Newmedic, France), delivering a biphasic electric current (pulse frequency 60 Hz, 1 ms pulse width). Functional disturbances were evaluated online by a speech therapist and responsive sites (i.e., areas in which repetitive stimulation provoked at least three identical functional disturbances) were marked with sterile number tags. At current amplitude of 3.75 mA, the primary somatosensory and motor cortices were first identified by recording any sensory disturbance and by asking the patient to continually count up to ten while performing regular movements with the right arm. Then picture-naming (DO80) [44] and right arm movements were used as intraoperative tasks until the end of resection. Once the exposed brain surface was entirely mapped, lesion removal was started. A firm, whitish lesion, well delineated but strongly adherent to the arachnoid mater, was found and completely removed. Since perilesional tumor infiltration could not be ruled out, resection 3 was extended into the modified adjacent brain tissue. Continuous subcortical stimulation was performed, using the same current amplitude of 3.75 mA, and surgery was suspended after identification of eloquent areas in the depth of the operative cavity (see Fig. 2A and B). Intraoperative photographs were systematically taken before and after resection. The removed lesion was cut in half, and a cystic cavity with gelatinous content, surrounded by a thick wall, was found (see Fig. 2C and D). The histopathological examination revealed a degenerating parasitic cyst, containing a cysticercus with wavy cuticle external layer covered by microtriches and associated with aggregated subcuticular cells, and internal layer composed of loose myxoid tissue. The cyst was surrounded by a large inflammatory cell infiltrate (plasma cells, lymphocytes, macrophages, and rarer eosinophils, neutrophils and giant cells) in a background of fibrosis and gliosis (see Fig. 3). On postoperative radiographs of the limbs, no calcified lesions were found in the soft tissue. Treatment with albendazole (400 mg twice daily), simultaneous to corticosteroid therapy, was performed for two 2 weeks. The neurological deficit recovered progressively, and neurocognitive assessment at 1 year was normal. At the 1-year follow-up, brain MRI found total edema resorption without any new or residual infectious lesion. There had been no further epileptic seizures were recorded, and from the 13th postoperative month antiepileptic treatment was gradually stopped, allowing the patient to return to normal social and occupational life. 3. Discussion Neurocysticercosis is the most common parasitosis of the central nervous system worldwide [45]. It is caused by the larval stage of the tapeworm Taenia solium after fecal-oral contamination with Fig. 3. Histopathological findings (hematoxylin and eosin stain). A. Cysticercus parasite composed of wavy cuticle external layer and loose myxoid internal content (original magnification ×20). B. Intermediate- (×100) and high-power (×400, upper right corner) magnifications of the cyscticercus, showing a superficial layer covered with microtriches, an intermediate cellular layer, and an inner loose reticular layer. C. Abscess wall and central cavity, seen at low (×20) magnification. D. High-power (×400) magnification of the abscess wall, showing large inflammatory cell infiltrate containing lymphocytes, plasma cells, eosinophils, neutrophils and giant cells. Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Provisional diagnosis Neoplastic history Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis ArismendiMorillo 2004 Bouillot 2003 NA (NR) NA (NR)/Venezuela NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) 14/NA (NR) Seizure 1 NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) 29/NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) 12/M No Seizure, left facial paralysis Left-eye vision loss without exophthalmos 1 Bousquet 1996 High grade glioma High grade glioma Retrobulbar optic nerve tumor NA (NR) Bouillot 2003 Endemic areas/France Endemic areas/France Reunion Island/France 1 Retrobulbar portion of the left optic nerve NS NS Surgical removal Bruschi 2006 NA (NR) NA (NR)/Italy Tumor NA (NR) Seizures 1 NS NA (NR) Colli 1984 22/F NA (NR)/Brazil Tumor NA (NR) Seizures, right hemiparesis, dysphasia 2 Surgical removal Surgical removal Colli 1986 15/F Brazil/Brazil Tumor NS Multiple Colli 1986 13/M Brazil/Brazil Tumor NS Headache, dysosmia, hydrocephalic hypertension, cerebellar syndrome Left brachio-facial paresis Right-frontotemporal Left frontotemporoparietal, and other, more diffuse Right temporal, medial frontal CT: hypodense center, hyperdense periphery, ring enhancement, no perilesional edema MRI: cystic lesion CT: cystic lesion Coulibaly 2008 70/F Guadeloupe/FranceMetastasis Multiple Dao, 2015 37/F Congo/Morocco Metastasis Breast adenocarcinoma Gait disturbance, right hemiparesis, bradypsychia Headache, gait disorder 1 Left internal temporooccipital. 57 × 51 mm (CT), 32 × 30 mm (MRI) Kaw 1994 NA (NR) Meningioma NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) Kim 2006 67/M NA (NR)/NA (NR) NS/Korea Dizziness, nausea, vomiting 1 Left cerebellar 55 × 40 × 40 mm NS HemangioblastomaNS Multiple Left fronto-parietal and right parietal Diffuse 70 × 77 mm NA (NR) NS CT: cystic lesion (+) in serum (retrospectively) Ventriculography NS CT: enhancing lesions, perilesional edema CT/MRI: enhancing lesions, no perilesional edema CT: hyperdense, ring enhancement, perilesional edema NA (NR) (+) in serum Serology, spontaneous disappearance Western blot (+) in serum and CSF Biopsy (−) in serum Biopsy NA (NR) Biopsy (+) in serum (retrospectively) Surgical removal Small MRI: cystic lesion with thin wall, internal septations and mural nodule, ring enhancement except of the associated nodule, subtle perilesional edema ARTICLE IN PRESS Patient residence/Country of publication G Model Age (yrs)/gender A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Authors & Date NEUCHI-1008; No. of Pages 15 4 Table 1 Literature review of studies reporting neurocysticercosis mimicking neoplasia. Table 1 (Continued) Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis L’Ollivier 2012 69/M France/France Glioma NS Asthenia and headache 1 Right frontal 40 × 30 mm NS Surgical removal Matyja 1998 Matyja 1998 McDowell and Harper 1990 McDowell and Harper 1990 Michael 1990 NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) 1 NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) Tumor No NA (NR) NA (NR) Seizures, headache Seizures, headache Headache NA (NR) NA (NR) Multiple 39/F NA (NR)/Poland NA (NR)/Poland NA (NR)/Australia NA (NR)/Australia USA/USA CT/MRI: internal necrotic or cystic septations, ring enhancement, perilesional edema NA (NR) NA (NR) NA (NR) 1 Peripheral NS NS Surgical removal Michael 1990 32/F USA/USA Tumor No Seizure 1 Right frontal Small NS Surgical removal Mittal 2014 1 ½/M NS/India Tumor no NA (NR) 1 Right thalamus 43 × 45 mm NS New MRI 4 months later Nandhagopal 2010 Ohsaki 1999 6/M NA (NR)/NA (NR) NS (but lived in India for 5 years)/Japan NA (NR) Tuberous sclerosis No NA (NR) 2 NA (NR) NA (NR) CT/MRI: isodense, heterogeneous medium intensity nodule, ring enhancement, large perilesional edema CT/MRI: isodense, medium intensity, perilesional edema CT/MRI: hypodense lesion with eccentric mural scolex, T1 hypointense, minimal enhancement, T2 heterogeneously hyperintense, increased choline peak and decreased N-acetyl aspartate, moderate perilesional edema NA (NR) NA (NR) NA (NR) Seizures 1 Right parietal 6 mm MRI: T1 hypointense, ring enhancement, no perilesional edema ELISA and EITB (-) (after surgical removal) Surgical removal NA (NR) 19/F Tumor ARTICLE IN PRESS Neoplastic history G Model Provisional diagnosis NEUCHI-1008; No. of Pages 15 Patient residence/Country of publication 5 Age (yrs)/gender A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Authors & Date Sabel 2001 47/M Silver 1996 NA (NR) Song 2013 Provisional diagnosis Neoplastic history Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis Greece/Germany Malignant tumor NS Speech difficulties, dyscalculia, alexia 1, multilobular Left frontal NS NS Surgical removal High grade glioma glioma No NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) 68/F Born in Korea/US NS/China CT/MRI: hypodense center and hyperdense periphery, other small calcified lesions, T1 hypointense, ring enhancement, T2 hyperintense, mild perilesional edema NA (NR) NS Facial numbness, left hearing impairment, headache 1 Left parietal 42 × 20 × 25 mm Surgical removal Surgical removal Umredkar 2009 17/F NS/India Abscess or glioma NS HEADACHE, cognitive deficit 1 Left insular Umredkar 2009 40/M NS/India Abscess or glioma NS Headache, seizures, cognitive deficit 1 Left frontal MRI: T1 hypointense, ring enhancement, T2 hyperintense, no perilesional edema 53 × 43 × 35 mm MRI: T1 hypointense, ring enhancement, T2 hyperintense, perilesional edema NS MRI: cystic lesion, ring enhancement, perilesional edema (-) in serum NS Surgical removal NS Surgical removal ARTICLE IN PRESS Patient residence/Country of publication G Model Age (yrs)/gender A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Authors & Date NEUCHI-1008; No. of Pages 15 6 Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Table 1 (Continued) G Model NEUCHI-1008; No. of Pages 15 Age (yrs)/gender Patient residence/Country of publication Provisional diagnosis Neoplastic history Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis VasiljevicVuckovic 2011 30/F NS/Serbia Low grade tumor NS Seizure 1 Left occipital 35 × 20 mm NS Surgical removal Wada 2004 74/M Philippines/ Honolulu Primary or secondary tumor Gastric carcinoma Seizures 1 Right frontal 20 × 25 × 30 mm NS Surgical removal Yong and Warren 1994 Yong and Warren 1994 Yong and Warren 1994 Yong and Warren 1994 NA (NR) South America/Australia South America/Australia South America/Australia Cyprus/Australia NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) MRI: low-grade tumor spectrum, ring enhancement, no perilesional edema Irregular lesion, ring enhancement, perilesional edema NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA (NR) NA: not available; NR: full article not retrieved; NS: not specified in article; F: female; M: male; CT: computed tomography; MRI: magnetic resonance imaging; CSF: cerebrospinal fluid; EITB: enzyme-linked immunoelectrotransfer blot; ELISA: enzyme-linked immunosorbent assay. ARTICLE IN PRESS Authors & Date A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Table 1 (Continued) 7 Final diagnosis Neoplastic history Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis Agapejev 1992 32/M Brazil/Brazil Oligodendroglioma No Headache, cognitive deficit 1 Supra-sellar and fronto-parietal NS (+) in CSF and punctured fluid Surgical removal after therapeutic test (albendazole) BagoRozankovic 2013 55/F NS/Croatia Papillary thyroid carcinoma metastases No Headache, dizziness Multiple Supra- and infra-tentorial NS (+) in serum Biopsy of brain lesions after therapeutic test (albendazole) Bick 2012 62/F NS/US Breast carcinoma metastases Breast carcinoma Seizures Multiple Diffuse NS NS NS Choi 2012 50/M Korea/Korea Adenocarcinoma No metastases (primitive not found) Headache Multiple Prepontine and premedullary cisterns, cerebellar NS CT: hypodense center, hyperdense periphery, ring enhancement, perilesional edema CT/MRI: hypodense center, hyperdense periphery, cystic lesion, solid intraluminal component, ring enhancement, perilesional edema CT/MRI: punctate peripheral calcifications, minimal enhancement CT/MRI: cystic lesion, partial ring enhancement, no perilesional edema (−) in serum and CSF Costa 2014 61/F Brazil/Brazil Lung adenocarcinoma metastases No Seizures Multiple Diffuse NS Partial surgical removal of brain lesions after therapeutic test (praziquantel) Biopsy of brain lesions after therapeutic test (albendazole) Fantini 2017 41/F NA (NR) (caucasian)/Italy Multiple NA (NR) NA (NR) 56/M South America/USA Uterine cervical carcinoma No NA (NR) Graber 2011 Uterine cervical cancer metastases Pleomorphic sarcoma of malignant fibrous histiocytoma type Right sided weakness Multiple Diffuse NS Hoang 2017 64/M NA (NR)/USA Lung adenocarcinoma metastases NA (NR) Phototopia Multiple NA (NR) NS MRI: T1 hypo-intense, ring enhancement, perilesional edema Calcifications NS NA (NR) NA (NR) CT/MRI: hypodense center, hyperdense periphery, cystic lesion, ring enhancement, perilesional edema for some lesions NA (NR) (−) in serum Biopsy of new lesion in muscle NA (NR) NA (NR) ARTICLE IN PRESS Patient residence/Country of publication G Model Age (yrs)/genderex A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Authors & Date NEUCHI-1008; No. of Pages 15 8 Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Table 2 Literature review of studies reporting neoplasia mimicking neurocysticercosis. Patient residence/Country of publication Final diagnosis Neoplastic history Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis Kim 2017 47/M NS/Korea Non-small cell lung cancer metastases No Dysarthria Multiple Diffuse bilateral ? (−) in serum and CSF Therapeutic test (albendazole) Lam 2016 60/F NA (NR) (caucasian)/Canada Small-cell neuroendocrine carcinoma metastases No Ataxia, otalgia Multiple Supra- and infra-tentorial Varying sizes (−) serum Biopsy of brain lesions Li 2016 68/M China/China Anaplastic astrocytoma No Right lower limb weakness, headaches 1 Left frontal 6 mm (+) in serum Surgical removal after therapeutic test (albendazole) Mota 2011 47/M NS/Brazil Lung adenocarcinoma metastases Lung cancer found concomitantly Bilateral hearing loss, tinnitus, dizziness, imbalance Multiple Supra- and infra-tentorial NS NS Surgical removal of a brain lesion after therapeutic test (albendazole) Ruppert 2010 55/M NS/USA Lung adenocarcinoma metastases Lung adenocarcinoma Cerebellar ataxia Milliary Supra- and infra-tentorial NS MRI: smooth/thinwalled cystic lesions, little or no ring enhancement, no perilesional edema CT/MRI: cystic lesions, mostly thin-walled, some calcified, fluid levels, ring enhancement, no perilesional edema MRI: cystic lesion, smooth wall, ring enhancement, T1 hypointense, T2 hyperintense CT/MRI: hypodense center, hyperdense periphery, T2 hyperintense, T1 hypointense, no enhancement, high apparent diffusion coefficient, no perilesional edema MRI: T1 barely detectable (−) in serum and CSF Salomao 2006 15/M NS/Brazil Anaplastic oligoastrocytoma No Seizures Multiple Left parietotemporal, right hypothalamus NS Brain autopsy after therapeutic test (albendazole) Surgical removal after therapeutic test (albendazole) CT: hypodense center, hyperdense periphery with mural nodule, perilesional edema, calcified mass in the right hypothalamic (+) in the punctured fluid ARTICLE IN PRESS Age (yrs)/genderex G Model NEUCHI-1008; No. of Pages 15 Authors & Date A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Table 2 (Continued) 9 G Model NEUCHI-1008; No. of Pages 15 10 Age (yrs)/genderex Patient residence/Country of publication Final diagnosis Neoplastic history Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis Troiani 2011 45/F NS/Brazil Breast cancer metastases Breast cancer Headache Multiple Diffuse NS NS Surgical removal after therapeutic test (albendazole) Tuchman 2009 16/M NS/USA Choroid plexus papilloma No Depression, memory deficit 1 Septum pellucidum NS CT: hypodense, cystic lesions some with small mural nodules, T1 hypointense, ring enhancement, more intensely in the region of mural nodules, T2 hyperintense MRI: T1 hypo-intense, ring enhancement with associated enhancing nodule, T2 hyperintense, no perilesional edema NS Surgical removal NA: not available; NR: full article not retrieved; NS: not specified in article; F: female; M: male; CT: computed tomography; MRI: magnetic resonance imaging; CSF: cerebrospinal fluid. ARTICLE IN PRESS Authors & Date A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Table 2 (Continued) G Model NEUCHI-1008; No. of Pages 15 Age (yrs)/gender Patient residence/Country of publication Provisional diagnosis Neoplastic history Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis Dayananda 2011 20/M India/India Tuberculoma NS Headache, seizures Conglomerate Right frontal 29 × 17 mm NS Surgical removal Maeda 2011 NS/F India/Japan Tuberculoma No Headache, right forearm paresthesia Multiple NS NS MRI: hypointense periphery, hyperintense center, no diffusion restriction, ring enhancement, perilesional edema CT/MRI: scattered calcifications, T1 hyper-intense, enhancement, peripheral edema Positive (serum) Surgical removal (mitochondrial DNA PCR) NS: not specified in article; M: male; F: female; MRI: magnetic resonance imaging. ARTICLE IN PRESS Authors & Date A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx 11 Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Table 3 Literature review of studies reporting neurocysticercosis mimicking tuberculoma. G Model NEUCHI-1008; No. of Pages 15 12 Age (yrs)/gender Patient residence/Country of publication Final diagnosis Neoplastic history Symptoms Number of lesions Location Size Imaging characteristics Cysticercosis serology Diagnosis Lu 2011 41/M NS/Japan Tuberculoma NS Vertigo, vomiting Multiple Supra- and infra-tentorial, ventricles Small (+) in serum, (-) in CSF Failure of therapeutic test, MR spectroscopy Seth 2010 4/F India/India Tuberculoma NS Seizures 3 Left frontal, bi-parietal 9.6–13 mm (+) in serum Failure of therapeutic test, MR spectroscopy Tosomeen 1998 Verma 2014 26/F India/Lebanon Tuberculoma NS Seizures 1 NA (NR) NA (NR) MRI: T2hyperintense, ring enhancement, perilesional edema CT/MRI: enhancing excentric foci, perilesional edema NA (NR) NA (NR) NA (NR) 18/F India/India Tuberculoma NS Headache, seizure Multiple Supra- and infra-tentorial Small (−) in serum PCR tuberculosis MRI: increased choline peak, increased choline to creatinine ratio, normal Nacetylaspartate peak, ring enhancement NA: not available; NR: full article not retrieved; NS: not specified in article; F: female; M: male; CT: computed tomography; MRI: magnetic resonance imaging; CSF: cerebrospinal fluid; PCR: polymerase chain reaction. ARTICLE IN PRESS Authors & Date A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 Table 4 Literature review of studies reporting tuberculoma mimicking neurocysticercosis. G Model NEUCHI-1008; No. of Pages 15 ARTICLE IN PRESS A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx the eggs of the parasite. The embryos of the eggs reach the brain after hematogenous spread from the bowel, actively crossing the wall. After a long asymptomatic period, the initially viable cysticerci degenerate and the resulting inflammation causes specific symptoms, such as seizures and focal neurological and/or cognitive deficit, which depend on the topography and the size of the lesion(s). The difficulty of differentiating neurocysticercosis from tumor or tuberculoma has often been discussed in the literature and will most likely continue to be in the future. Because of the completely different management, such cases can be a real moral dilemma for the clinician. On the one hand, anti-parasitic drugs used in neurocysticercosis have potential side-effects, can be ineffective in bulky lesions, and can delay urgent treatment if the true diagnosis is neoplasia. On the other hand, brain surgery is invasive and associated with specific risks, in particular that of inducing or irreversibly worsening a neurological/cognitive deficit. Such a risk is greater for lesions located in eloquent brain areas and removed in a classical manner (under general anesthesia). Several studies have reported misdiagnosis of brain neurocysticercosis, and we performed a literature review of these, focusing on the differential diagnoses considered in the present case: tumor (studies listed in Tables 1 and 2) [4–43], and tuberculoma (studies listed in Tables 3 and 4) [46–51]. The PRISMA guidelines used to select articles are presented in Fig. 4. Twenty-four studies reported 33 cases with neurocysticercotic brain lesions that mimicked neoplasia (see Table 1), and full-text papers were retrieved for 16. For patients with available data, mean age was 34.4 ± 22.7 years (range, 1.5–74 years). Definitive diagnosis of neurocysticercosis was made by biopsy in 3 patients, surgical removal of the lesion in 15 patients (inconclusive biopsy having been performed in 1), suggestive ventriculography (in 1983) in 1, and repeat imaging in a child whose parents refused an invasive procedure. Tumor lesions initially diagnosed as neurocysticercosis were reported in 16 articles (see Table 2), and 14 full-text papers were retrieved. Sixteen patients were reported; for those with available data, mean age was 48.4 ± 15.3 years (range, 15–68 years). The final diagnosis of neoplasia was confirmed by biopsy in 4 patients (2 after 13 failure of a previous therapeutic test), surgical removal in 7 (all except 1 after unsuccessful therapeutic test), autopsy in 1, biopsy of a muscle lesion in 1, and after failure of therapeutic test with cysticidal drug but without histological confirmation in 1. Two articles (see Table 3) reported brain neurocysticercosis that mimicked tuberculoma, in a woman (age not specified) and a 20year-old man, respectively. In both cases, diagnosis was confirmed after resection of the lesion. Four articles (3 full-texts retrieved) reported tuberculoma that mimicked neurocysticercosis (see Table 4) in 4 patients. Mean age was 22.3 ± 13.4 years (range, 4–41 years). Diagnosis was made after failure of therapeutic test and through MR spectroscopy (2 cases), or polymerase chain reaction on cerebrospinal fluid (1 case). When the provisional diagnosis was neurocysticercosis, clinicians were more likely to be working in regions endemic for cysticercosis (9 of the 17 cases with final diagnosis of tumor, and 3 of the 4 cases of tuberculoma), while only 9 of the 32 cases with initial diagnosis of tumor or tuberculoma occurred in endemic regions. When the provisional diagnosis was neurocysticercosis and the final diagnosis was tumor, lesions were most often multiple (13 of the 16 cases). Conversely, when the provisional diagnosis was tumor, the number of lesions was 1 or 2 in 20 of the 33 cases (unspecified number in 9 of the 33 cases). Given the frequent misdiagnosis of parenchymal brain neurocysticercosis, diagnostic criteria have been proposed to help clinicians avoid delayed or inappropriate management [52]. However, often neither brain imaging nor serum and cerebrospinal fluid cysticercosis tests are sufficient for definitive diagnosis. Moreover, positive cysticercosis tests have been reported even when performed on a tumor biopsy sample [41]. For all these reasons, current management of neurocysticercosis remains contradictory and an individual approach is highly recommended [53]. Antiparasitic treatment may be used for viable cysticerci, but is generally not recommended for degenerative cysts. Because of the risk of additional brain damage, surgery is classically not considered, except for neurocysticercosis-induced hydrocephalus, with or without intraventricular or cisternal cysts. However, in case of large parenchymal cyst or uncertain diagnosis, surgical resection can also be indicated [54]. Bulky lesions can not only have major functional impact Fig. 4. PRISMA chart for literature review. Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 G Model NEUCHI-1008; No. of Pages 15 ARTICLE IN PRESS A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx 14 but also be life threatening, and removal can resolve the patient’s symptoms. If surgery is planned, conserving brain functions is of paramount importance, notably for lesions in eloquent areas. Direct electrical stimulation during awake surgery is the gold standard for detecting functional brain areas [55] and can be of great interest here. Traditionally used in the management of diffuse brain tumors and epilepsy, this technique is increasingly proving useful for other types of brain condition. In the present case, diagnosis could not be confirmed preoperatively despite a large set of laboratory and imaging investigations. In the absence of a lesion for which antiparasitic drugs could be indicated (cysticerci with viable appearance), no therapeutic test was performed. A wait-and-see attitude was rejected, given the large volume of the lesion, its unknown growth potential, and the existing neurological deficit and epilepsy. Despite the location of the lesion in a highly eloquent brain area, resection was safely performed using intraoperative mapping, demonstrating the usefulness of awake surgery in the management of degenerating parenchymal neurocysticercosis. Informed consent Informed consent was obtained from all participants included in the study. Disclosure of interest The authors declare that they have no competing interest. Acknowledgments We thank Stéphane Lecoules and Jean-Marie Cournac for their help with clinical evaluation. References [1] Bajpayee A, Gulati S, Lodha R, Kabra SK. Tuberculosis clinical grand round. Indian J Pediatr 2003;70:57–61. [2] Garg RK. Single enhancing computerized tomography-detected lesion in immunocompetent patients. Neurosurg Focus 2002;12:e4. [3] García HH, Gonzalez AE, Evans CAW, Gilman RH. Cysticercosis Working Group in Peru. Taenia solium cysticercosis. Lancet Lond Engl 2003;362:547–56, http://dx.doi.org/10.1016/S0140-6736(03)14117-7. [4] Arismendi Morillo GJ, Fernández Abreu MC, Cardozo Sosa DP, Cardozo JJ. Non neoplastic space occupying lesions mimicking central nervous system tumors. Rev Neurol 2004;38:427–30. [5] Bouillot S, Monteil P, Dautheribes M, Rougier A, Guerin J, Vital A. Two cases of neurocysticercosis mimicking brain tumor. Ann Pathol 2003;23:355–7. [6] Bousquet CF, Dufour TF, Derome PC. Retrobulbar optic nerve cysticercosis. Case report. J Neurosurg 1996;84:293–6, http://dx.doi.org/10. 3171/jns.1996.84.2.0293. [7] Bruschi F, Giangaspero F, Castagna M, Becherini F, Filice ME, Vannozzi R, et al. Neurocysticercosis: surgical treatment of an autochthonous case in a nonendemic region. Pathologica 2006;98:229–31. [8] Colli BO, Martelli N, Assirati Júnior JA, Machado HR, Guerreiro NE, Belluci A. Tumor form of neurocysticercosis: surgical removal of a cysticercus measuring 70X77 mm and treatment with praziquantel. Arq Neuropsiquiatr 1984;42:158–65. [9] Colli BO, Martelli N, Assirati Júnior JA, Machado HR. [Multiple nonneoplastic lesions in computerized cranial tomography]. Arq Neuropsiquiatr 1986;44:155–64. [10] Coulibaly B, Gautier G, Fuentes S, Ranque S, Bouvier C. [Degenerating neurocysticercosis cysts: differential diagnosis with cerebral metastasis]. Rev Neurol (Paris) 2008;164:948–52, http://dx.doi.org/10.1016/j.neurol.2008.03.019. [11] Dao A, Benchakroun N, Ahomadegbé C, Karkouri M, Benider A. Neurocysticercosis discovered incidentally during an asynchronous single brain metastasis of breast adenocarcinoma. Rev Neurol (Paris) 2015;171:84–7, http://dx.doi.org/10.1016/j.neurol.2014.07.014. [12] Kaw YT. Cytologic diagnosis of neurocysticercosis. A case report. Acta Cytol 1994;38:87–9. [13] Kim J-H, Suh S-I, Kim J-H, Kwon T-H, Chung H-S. Giant neurocysticercosis cyst in the cerebellar hemisphere. Neurol Med Chir (Tokyo) 2006;46:412–4. [14] L’Ollivier C, González LM, Gárate T, Martin L, Martha B, Duong M, et al. Histological and molecular biology diagnosis of neurocysticercosis in a patient without history of travel to endemic areas: case report. Parasite Paris Fr 2012;19:441–4, http://dx.doi.org/10.1051/parasite/2012194441. [15] Matyja E, Schmidt-Sidor B, Zabek M, Jagielski J, Królicki L. Multilocular cysticeral and hydatid cysts of the brain: a report of three cases. Folia Neuropathol 1998;36:239–43. [16] McDowell D, Harper CG. Neurocysticercosis – two Australian cases. Med J Aust 1990;152:217–8. [17] Michael AS, Levy JM, Paige ML. Cysticercosis mimicking brain neoplasm: MR and CT appearance. J Comput Assist Tomogr 1990;14:708–11. [18] Mittal A, Sehgal R, Sureka B, Kumar A, Aggarwal KC. Giant cerebral cysticercosis in an infant confused with a thalamic glioma. J Child Neurol 2014;29:NP154–6, http://dx.doi.org/10.1177/0883073813513332. [19] Nandhagopal R. Coexistence of cerebral tubers with neurocysticercosis. South Med J 2010;103:953–5, http://dx.doi.org/10.1097/SMJ.0b013e3181eb345f. [20] Ohsaki Y, Matsumoto A, Miyamoto K, Kondoh N, Araki K, Ito A, et al. Neurocysticercosis without detectable specific antibody. Intern Med Tokyo Jpn 1999;38:67–70. [21] Sabel M, Neuen-Jacob E, Vogt C, Weber F. Intracerebral neurocysticercosis mimicking glioblastoma multiforme: a rare differential diagnosis in Central Europe. Neuroradiology 2001;43:227–30. [22] Silver SA, Erozan YS, Hruban RH. Cerebral cysticercosis mimicking malignant glioma: a case report. Acta Cytol 1996;40:351–7, http://dx.doi.org/10. 1159/000333767. [23] Song L, Guo F, Ma S, Song Z, Wu L, Sun H. A rare presentation of intracranial cysticercosis involving the cerebellopontine angle. J Clin Neurosci 2013;20:892–4, http://dx.doi.org/10.1016/j.jocn.2012.05.046. [24] Umredkar A, Singla N, Mohindra S, Bal A, Gupta SK. Giant intraparenchymal neurocysticercosis: report of surgical aspects two cases. Neurol India 2009;57:800–2, http://dx.doi.org/10.4103/0028-3886.59483. [25] Vasiljević-Vučković V, Milošević Medenica S, Grujičić D. Neurocysticercosis mimicking brain tumor. Neuroradiol J 2011;24:419–23, http://dx.doi.org/10. 1177/197140091102400311. [26] Wada D, Morita M, Hardman JM. January 2004: elderly Filipino man with frontal lobe tumor. Brain Pathol Zurich Switz 2004;14:337–8 [343]. [27] Yong JL, Warren BA. Neurocysticercosis: a report of four cases. Pathology (Phila) 1994;26:244–9. [28] Agapejev S, Alves A, Zanini MA, Ueda AK, Pereira EM. Cystic oligodendroglioma and positivity of reactions for cysticercosis: report of a case. Arq Neuropsiquiatr 1992;50:234–8. [29] Bago-Rožanković P, Bašić S, Vavro H, Rožanković M, Nikolić I, Muller D, et al. Multiple cystic brain metastases from papillary thyroid carcinoma mimicking neurocysticercosis. Int J Surg Pathol 2013;21:390–3, http://dx.doi.org/10. 1177/1066896912473353. [30] Bick BL, Haji S, Laughlin RS, Watson RE, Kumar N. Metastatic breast cancer suggesting parasitic disease. Neurology 2012;79:2366, http://dx.doi.org/10. 1212/WNL.0b013e318278b696. [31] Choi HJ, Choi SK. Multiple cystic brain metastases from adenocarcinoma mimicking cysticercosis. Case report. Clin Neuroradiol 2012;22:105–7, http://dx.doi.org/10.1007/s00062-011-0074-5. [32] Costa R, Costa RB, Bacchi C, Sarinho F. Adenocarcinoma of the lung presenting with atypical cystic brain lesions. BMJ Case Rep 2014;2014, http://dx.doi. org/10.1136/bcr-2013-203506 [3 pages]. [33] Fantini J, Sartori A, Manganotti P. Avoiding misdiagnosis: cystic calcified brain metastases of uterine cervical cancer mimicking neurocysticercosis. BMJ Case Rep 2017;2017, http://dx.doi.org/10.1136/bcr-2016-217952 [3 pages]. [34] Graber JJ, Nayar A, Zagzag D. Metastatic cerebral malignant fibrous histiocytoma masquerading as neurocysticercosis. J Neurooncol 2011;105:437–9, http://dx.doi.org/10.1007/s11060-011-0583-8. [35] Hoang A, Khine KT, Knight OJ, Tesser RA, Chesnutt DA. A rare ocular presentation of metastatic lung cancer: unilateral anterior chamber angle, bilateral choroidal, and multiple intracranial metastases. J Glaucoma 2017;26:e93–5, http://dx.doi.org/10.1097/IJG.0000000000000518. [36] Kim S-H, Hyun J-W, Kim HJ, Gwak H-S, Lee SH, Hong E-K, et al. De novo cystic brain lesions mimicking neurocysticercosis in ALKpositive lung cancer. Lung Cancer Amst Neth 2017;110:53–5, http://dx.doi. org/10.1016/j.lungcan.2017.06.003. [37] Lam JC, Robinson SR, Schell A, Vaughan S. Pulmonary neuroendocrine carcinoma mimicking neurocysticercosis: a case report. J Med Case Rep 2016;10:144, http://dx.doi.org/10.1186/s13256-016-0910-y. [38] Li H-J, Han H-X, Feng D-F. Rapid increase in cystic volume of an anaplastic astrocytoma misdiagnosed as neurocysticercosis: a case report. Oncol Lett 2016;12:2825–7, http://dx.doi.org/10.3892/ol.2016.4986. [39] Mota PC, Reis C, Pires NF, Sousa G, Chamadoira C, Guimarães M, et al. Lung cancer: atypical brain metastases mimicking neurocysticercosis. Int J Clin Oncol 2011;16:746–50, http://dx.doi.org/10.1007/s10147-011-0221-7. [40] Ruppert A-M, Stankoff B, Lavolé A, Gounant V, Milleron B, Seilhean D. Miliary brain metastases in lung cancer. J Clin Oncol 2010;28:e714–6, http://dx.doi.org/10.1200/JCO.2009.27.0140. [41] Salomão JF, Pone MV, da Silva ARA, Leibinger RD, Bellas AR, Campos JMS, et al. Positive reaction for cysticercosis and multicentric anaplastic oligoastrocytoma. Childs Nerv Syst ChNS 2006;22:182–5, http://dx.doi.org/ 10.1007/s00381-004-1099-8. [42] Troiani C, Lopes CCB, Scardovelli CA, Nai GA. Cystic brain metastases radiologically simulating neurocysticercosis. Sao Paulo Med J Rev Paul Med 2011;129:352–6. [43] Tuchman A, Kalhorn SP, Mikolaenko I, Wisoff JH. Cystic choroid plexus papilloma in the cavum septum pellucidum. J Neurosurg Pediatr 2009;4:580–3, http://dx.doi.org/10.3171/2009.7.PEDS08484. Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002 G Model NEUCHI-1008; No. of Pages 15 ARTICLE IN PRESS A. Vienne et al. / Neurochirurgie xxx (2019) xxx–xxx [44] Metz M, Metz-Lutz MN, Kremin H, Deloche G, Hannequin D, Ferrand RA, et al. Standardisation d’un test de dénomination orale : contrôle des effets de l’âge, du sexe et du niveau de scolarité chez les sujets adultes normaux. Rev Neuropsychol 1991;1:73–95. [45] DeGiorgio CM, Medina MT, Durón R, Zee C, Escueta SP. Neurocysticercosis. Epilepsy Curr 2004;4:107–11, http://dx.doi.org/10.1111/j. 1535-7597.2004.43008.x. [46] Dayananda L, Kesavadas C, Thomas B, Neelima R, Radhakrishnan VV. Atypical clinical and imaging manifestation in neurocysticercosis. Ann Indian Acad Neurol 2011;14:295–7, http://dx.doi.org/10.4103/0972-2327.91955. [47] Maeda T, Ito A, Sako Y, Yamasaki H, Oyaizu N, Odawara T, et al. Neurocysticercosis case with tuberculoma-like epithelioid granuloma strongly suspected by serology and confirmed by mitochondrial DNA. BMJ Case Rep 2011;2011, http://dx.doi.org/10.1136/bcr.04.2011.4125 [4 pages]. [48] Lu Z, Zhang B, Qiu W, Hu X. Disseminated intracranial tuberculoma mimicking neurocysticercosis. Intern Med Tokyo Jpn 2011;50:2031–4. [49] Seth R, Kalra V, Sharma U, Jagannathan N. Magnetic resonance spectroscopy in ring enhancing lesions. Indian Pediatr 2010;47:803–4. [50] Tosomeen AH, Berbari EF, Levy NT, McClure RF, Krecke KN, Osmon DR. Instructive case report. A 26-year-old Indian woman with seizures and multiple intracranial mass lesions. J Med Liban 1998;46:349–52. 15 [51] Verma R, Gupta R. Multiple ring-enhancing lesions: diagnostic dilemma between neurocysticercosis and tuberculoma. BMJ Case Rep 2014;2014, http://dx.doi.org/10.1136/bcr-2013-202528 [3 pages]. [52] Del Brutto OH. Diagnostic criteria for neurocysticercosis, revisited. Glob Health 2012;106:299–304, http://dx.doi.org/10.1179/ Pathog 2047773212Y.0000000025. [53] García HH, Evans CAW, Nash TE, Takayanagui OM, White AC, Botero D, et al. Current consensus guidelines for treatment of neurocysticercosis. Clin Microbiol Rev 2002;15:747–56. [54] Singh G, Prabhakar S. Taenia solium cysticercosis: from basic to clinical science. CABI; 2002. [55] Duffau H, Lopes M, Arthuis F, Bitar A, Sichez J-P, Van Effenterre R, et al. Contribution of intraoperative electrical stimulations in surgery of low grade gliomas: a comparative study between two series without (1985-96) and with (1996-2003) functional mapping in the same institution. J Neurol Neurosurg Psychiatry 2005;76:845–51, http://dx.doi.org/10.1136/jnnp.2004.048520. Please cite this article in press as: Vienne A, et al. Awake surgery for isolated parenchymal degenerating neurocysticercosis – Case report and focused review of misdiagnosis of neurocysticercosis. Neurochirurgie (2019), https://doi.org/10.1016/j.neuchi.2019.07.002