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Fulltext Response to: "Before blaming a COVID vaccine for cytotoxic lesions of the corpus callosum all other differentials must be ruled out"

Ohara H, Shimizu H, Kasamatsu T, Kajita A, Uno K, Lai KW, et al.

Neuroradiology, 2022 Nov;64(11):2101-2102.
PMID: 36138239 DOI: 10.1007/s00234-022-03057-x
Matched MeSH terms: Corpus Callosum/pathology
Depression preceding Marchiafava-Bignami disease

Zhen NB, Gaillard F, Walterfang M, Mohd Zain NR, Yoon CK

Aust N Z J Psychiatry, 2021 02;55(2):224-225.
PMID: 32720512 DOI: 10.1177/0004867420945786
Matched MeSH terms: Corpus Callosum
Fulltext Cytotoxic lesions of the corpus callosum after COVID-19 vaccination

Ohara H, Shimizu H, Kasamatsu T, Kajita A, Uno K, Lai KW, et al.

Neuroradiology, 2022 Oct;64(10):2085-2089.
PMID: 35809100 DOI: 10.1007/s00234-022-03010-y

A 23-year-old previously healthy man (Patient 1) and a 33-year-old woman with a past history of depression (Patient 2) developed neurological symptoms approximately 1 week after receipt of the first COVID-19 mRNA vaccination and deteriorated over the next week. Patient 1 reported nausea, headache, a high fever, and retrograde amnesia. Patient 2 reported visual disturbance, headache, dysarthria, a left forearm tremor, dysesthesia of the mouth and distal limbs, and visual agnosia. PCR test results for SARS-CoV-2 were negative. Complete blood cell count, biochemistry, and antibody test and cerebrospinal fluid test findings were unremarkable. Diffusion-weighted and fluid-attenuated inversion recovery MRI of the brain showed a high signal intensity lesion at the midline of the splenium of the corpus callosum compatible with cytotoxic lesions of the corpus callosum (CLOCCs). High-dose intravenous methylprednisolone improved their symptoms and imaging findings. CLOCCs should be considered in patients with neurological manifestation after COVID-19 vaccination.

Matched MeSH terms: Corpus Callosum/pathology
Frontoethmoidal encephalomeningocoele with colpocephaly: case report and clinical review

Bhattacharjee A, Chakraborty A, Purkaystha P

J Laryngol Otol, 2008 Mar;122(3):321-3.
PMID: 17666141

Frontoethmoidal encephalomeningocoele is a rare congenital disease in which an intracranial mass protrudes through a midline defect from the anterior cranial fossa into the facial skeleton. The condition affects patients in South East Asian countries, such as Thailand, Burma, Malaysia and Indonesia, with frequency of 1 in 5000. The pathogenesis of encephalocoeles may be regarded as a 'late' neurulation defect during the fourth gestational week. We present a case of frontoethmoidal encephalomeningocoele with corpus callosal agenesis and colpocephaly; this may well be the first report of this combination. The patient had a bulging mass in the middle frontonasal area, with broadening of the nasal bridge and hypertelorism. Computed tomography scans delineated the skull defect and associated brain anomalies. A one-stage, combined transfacial-transcranial approach, correctional procedure was performed. We present here a discussion of the findings, with special reference to the condition's pathogenesis, morphological classification and evolving surgical treatments. Early diagnosis and referral, involving multidisciplinary teamwork, are of paramount importance because of the distorting influence of the extruding mass on facial growth.

Matched MeSH terms: Agenesis of Corpus Callosum
Marchiafava-Bignami disease in a non-alcoholic Indian male

Leong AS

Pathology, 1979 Apr;11(2):241-9.
PMID: 460949

Marchiafava-Bignami disease, a rare affliction of alcoholic males, is described in a severely malnourished Malaysian Indian male who took no alcohol. It is the second report of the disease in an Asian and represents one of the few cases which have occurred in non-alcoholics. Besides the pathognomonic demyelination of the central portion of the corpus callosum, there were striking demyelinative plaques in the subcortical white matter. In addition, neuropathological features of Wernicke's disease were found suggesting that severe malnutrition with thiamine deficiency was probably the cause of his demise.

Matched MeSH terms: Corpus Callosum/pathology
Fulltext Evaluation of neuroprotective effects of alpha-tocopherol in cuprizone-induced demyelination model of multiple sclerosis

Mitra NK, Xuan KY, Teo CC, Xian-Zhuang N, Singh A, Chellian J

Res Pharm Sci, 2020 Dec;15(6):602-611.
PMID: 33828603 DOI: 10.4103/1735-5362.301345

Background and Purpose: Multiple sclerosis (MS) is an autoimmune disorder characterized by demyelination and axonal loss. Quantitative estimation of behavioral, locomotor, and histological changes following the use of alpha-tocopherol (AT) in the animal model of MS have not been reported. The present study was planned to evaluate whether AT can improve sensorimotor dysfunction and reduce demyelination in the cuprizone (CPZ)-induced rat model of MS.
Experimental approach: Female Sprague-Dawley rats (8 weeks) were fed with cuprizone diet for 5 weeks followed by intraperitoneal injections of alpha-tocopherol (100 mg/Kg) or PBS for 2 weeks (groups E1 and E2, n = 8). Group C (n = 8) was fed with normal pellets followed by intraperitoneal doses of PBS. Open-field test and beam walking were carried out on every 10th day. The mean area of demyelination in the corpus callosum was quantified in Luxol® fast blue (LFB) stained histological sections of the forebrain. Qualitative grading for relative changes in the stains of myelinated fibers was also done.
Findings/Results: During withdrawal of CPZ, AT treatment increased the average speed by 22% in group E1, compared to group E2 (P < 0.05). The mean time to walk the beam was reduced in group E1 by 2.6% compared to group E2 (P < 0.05). The rearing frequency was increased in group E1 during week 6-7 compared to that in the period of CPZ treatment. The mean area of demyelination in the corpus callosum showed a 12% reduction in group E1 compared to group E2 (P < 0.05).
Conclusion and implications: Short-term AT therapy showed improvement in motor dysfunction and reduction of demyelination in the animal model of MS.

Matched MeSH terms: Corpus Callosum
'Pai Syndrome' with anterior alveolar polyp: A variant of a rare clinical entity

Debnath KK, Kanapaty Y, Yong DJ, Chandran S, Bakri A

Med J Malaysia, 2019 Jun;74(3):229-230.
PMID: 31256179

'Pai syndrome' (PS) is a rare congenital syndrome. Presented here, a new-born baby-girl who exhibited the characteristic features of having a midline nasal (septal) polyp, an anterior alveolar process polyp, and a pericallosal lipoma associated with corpus callosum dysgenesis of the brain. Both polyps were lined with stratified-squamous epithelium. The overall features were largely consistent with those described by Pai et al., in 1987. A midline cleft-lip (with or without cleft-alveolus) is one of the most common features of the syndrome which was however absent in this case. Instead, an anterior alveolar polyp is present, which is relatively rare.

Matched MeSH terms: Agenesis of Corpus Callosum
Fulltext Challenging lymphoid malignancy of primary central nervous system lymphoma: A case report

Kasinathan G, Kori AN, Hassan N

Ann Med Surg (Lond), 2020 Sep;57:307-310.
PMID: 32874561 DOI: 10.1016/j.amsu.2020.08.011

Introduction: Primary central nervous lymphoma is an aggressive disease without evidence of systemic spread with an annual incidence of 7 cases per 1,000,000 people in the United States.
Case presentation: A 68-year-old gentleman of Malay ethnicity presented with left sided weakness associated with reduced sensation for one month. The patient was healthy and denied any constitutional symptoms, joint pains, rash or seizures. There was no recent trauma. Physical examination revealed left upper and lower limb motor grade power of 3/5 with upper motor neurone weakness of the left facial nerve. He had brisk reflexes and an upgoing extensor plantar response. Brain imaging (Magnetic Resonance Imaging) showed two lesions: one occupying the right head of the caudate nucleus and the other seen at the right side of the body of the corpus callosum. Histomorphology and immunohistochemistry confirmed Diffuse Large B-Cell Lymphoma (DLBCL) of non-germinal center type. He was treated with De Angelis protocol which involves chemoradiotherapy consisting of high dose methotrexate and whole brain radiotherapy (WBRT), followed by high dose cytarabine. Brain imaging post chemoradiation showed complete remission.
Conclusion: Prompt detection with appropriate therapeutic protocol could significantly minimise the permanent neurological deficits in patients with this rare and challenging lymphoid malignancy.

Matched MeSH terms: Corpus Callosum
Fulltext Changes in white matter fiber density and morphology across the adult lifespan: A cross-sectional fixel-based analysis

Choy SW, Bagarinao E, Watanabe H, Ho ETW, Maesawa S, Mori D, et al.

Hum Brain Mapp, 2020 08 15;41(12):3198-3211.
PMID: 32304267 DOI: 10.1002/hbm.25008

White matter (WM) fiber bundles change dynamically with age. These changes could be driven by alterations in axonal diameter, axonal density, and myelin content. In this study, we applied a novel fixel-based analysis (FBA) framework to examine these changes throughout the adult lifespan. Using diffusion-weighted images from a cohort of 293 healthy volunteers (89 males/204 females) from ages 21 to 86 years old, we performed FBA to analyze age-related changes in microscopic fiber density (FD) and macroscopic fiber morphology (fiber cross section [FC]). Our results showed significant and widespread age-related alterations in FD and FC across the whole brain. Interestingly, some fiber bundles such as the anterior thalamic radiation, corpus callosum, and superior longitudinal fasciculus only showed significant negative relationship with age in FD values, but not in FC. On the other hand, some segments of the cerebello-thalamo-cortical pathway only showed significant negative relationship with age in FC, but not in FD. Analysis at the tract-level also showed that major fiber tract groups predominantly distributed in the frontal lobe (cingulum, forceps minor) exhibited greater vulnerability to the aging process than the others. Differences in FC and the combined measure of FD and cross section values observed between sexes were mostly driven by differences in brain sizes although male participants tended to exhibit steeper negative linear relationship with age in FD as compared to female participants. Overall, these findings provide further insights into the structural changes the brain's WM undergoes due to the aging process.

Matched MeSH terms: Corpus Callosum
Fulltext Homonymous Hemianopia: A Rare Presentation of Secondary Central Nervous System Neurolymphomatosis

Awis Qarni F, Tai E, Wh WH, Husin A

Cureus, 2018 May 29;10(5):e2708.
PMID: 30062082 DOI: 10.7759/cureus.2708

Neurolymphomatosis is an atypical complication of non-Hodgkin lymphoma and leukaemia involving infiltration of neurotropic neoplastic cells in the central or peripheral nervous system. A 28-year-old Malay lady with background diffuse large B-cell lymphoma stage IV presented with left homonymous hemianopia associated with cognitive function deterioration. Her best corrected visual acuity was 6/9 in both eyes. Magnetic resonance imaging (MRI) of the brain showed a lesion suggestive of secondary lymphomatous infiltration of the splenium of corpus callosum. The patient underwent chemotherapy, after which repeated MRI showed a reduction in the lesion size. Homonymous hemianopia is a rare presentation of secondary central nervous system neurolymphomatosis. A comprehensive history, physical examination, and radiological imaging are essential to establish the diagnosis in patients presenting with visual field defects.

Matched MeSH terms: Corpus Callosum
A novel ITPA variant causes epileptic encephalopathy with multiple-organ dysfunction

Sakamoto M, Kouhei D, Haniffa M, Silva S, Troncoso M, Santander P, et al.

J Hum Genet, 2020 Sep;65(9):751-757.
PMID: 32405030 DOI: 10.1038/s10038-020-0765-3

Inborn errors of metabolism can cause epileptic encephalopathies. Biallelic loss-of-function variants in the ITPA gene, encoding inosine triphosphate pyrophosphatase (ITPase), have been reported in epileptic encephalopathies with lack of myelination of the posterior limb of the internal capsule, brainstem tracts, and tracts to the primary visual and motor cortices (MIM:616647). ITPase plays an important role in purine metabolism. In this study, we identified two novel homozygous ITPA variants, c.264-1 G > A and c.489-1 G > A, in two unrelated consanguineous families. The probands had epilepsy, microcephaly with characteristic magnetic resonance imaging findings (T2 hyperintensity signals in the pyramidal tracts of the internal capsule, delayed myelination, and thin corpus callosum), hypotonia, and developmental delay; both died in early infancy. Our report expands the knowledge of clinical consequences of biallelic ITPA variants.

Matched MeSH terms: Corpus Callosum/pathology