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  1. Ching, Bih-Hwa, Khoo, Teik-Beng
    Neurology Asia, 2017;22(3):243-252.
    MyJurnal
    Objective: We aim to study the prevalence and predictive factors for hip displacement, in order to
    justify a hip surveillance programme for children with cerebral palsy (CP) in Malaysia.

    Methods:
    Children aged 2 to18 years old with CP were recruited from September 2013 till June 2014. The hip
    joint migration percentage (MP) and acetabular index (AI) were measured on all hip radiographs.
    The CP subtype was determined and gross motor function was classified according to the gross motor
    function classification system (GMFCS).

    Results: Seventy-five children were recruited. Fifty-five percent of them had marked hip displacement
    with MP > 30% and 15% developed hip dislocation (MP=100%). Marked hip displacement occurred
    as early as age of 2 years and most hip dislocations were detected by age of 10 years. The risk of
    marked hip displacement was directly related to the GMFCS level, from none in GMFCS I to 75% in
    GMFCS V. There was a moderate positive correlation between the initial AI and initial MP.

    Conclusions: One in every two children with CP was at risk of hip displacement, with GMFCS level
    and initial AI as significant predictive factors. We recommend a hip surveillance programme for
    Malaysian children with CP, based on the child’s age and GMFCS level, with both MP and AI as
    indicators for hip surveillance.
  2. Khoo TB
    J Child Neurol, 2013 Jan;28(1):56-9.
    PMID: 22532543 DOI: 10.1177/0883073812439623
    In its 2010 report, the International League Against Epilepsy Commission on Classification and Terminology had made a number of changes to the organization, terminology, and classification of seizures and epilepsies. This study aims to test the usefulness of this revised classification scheme on children with epilepsies aged between 0 and 18 years old. Of 527 patients, 75.1% only had 1 type of seizure and the commonest was focal seizure (61.9%). A specific electroclinical syndrome diagnosis could be made in 27.5%. Only 2.1% had a distinctive constellation. In this cohort, 46.9% had an underlying structural, metabolic, or genetic etiology. Among the important causes were pre-/perinatal insults, malformation of cortical development, intracranial infections, and neurocutaneous syndromes. However, 23.5% of the patients in our cohort were classified as having "epilepsies of unknown cause." The revised classification scheme is generally useful for pediatric patients. To make it more inclusive and clinically meaningful, some local customizations are required.

    Study site: The pediatric neurology clinic at the Institute of Pediatrics, Kuala
    Lumpur Hospital
  3. Anandakrishnan P, Khoo TB
    BMJ Case Rep, 2018 May 30;2018.
    PMID: 29848532 DOI: 10.1136/bcr-2018-224496
    Cerebral demyelination and optic neuritis are often seen in children with acute disseminated encephalomyelitis following various infections and immunisations. An eight month old girl presented with a left axillary lymph node swelling and an erythematous lace-like rash over her cheeks and trunk. She then developed acute encephalopathy, bilateral nystagmus, right hemiparesis and left facial nerve palsy. Her electroencephalogram showed an encephalopathic process and visual evoked response study were grossly abnormal. Her MRI brain showed hyperintensities in the midbrain, pons and bilateral cerebellar peduncles. She was treated as postinfectious cerebral demyelination with intravenous antibiotics, methylprednisolone and immunoglobulin. Left axillary lymph node excision biopsy and GeneXpert test detected Mycobacterium tuberculosis complex that prompted initiation of antituberculous therapy. Her chest X-ray and cerebrospinal fluid examinations for tuberculosis were normal. She showed significant recovery after 2 weeks. This case illustrates a rare presentation of cerebral demyelination and bilateral optic neuritis following suppurative BCG lymphadenitis.
  4. Gan KL, Khoo TB
    Cureus, 2024 Feb;16(2):e54693.
    PMID: 38524056 DOI: 10.7759/cureus.54693
    Despite having equal rights to education, children with neurodisability (CWND) continue to face a myriad of barriers to education. This cross-sectional survey aimed to investigate the barriers to education among CWND who attended follow-up at a Malaysian tertiary hospital. Sixty-two caregivers of CWND completed a questionnaire which included the 'Washington Group/United Nations Children's Fund (UNICEF) Child Functioning Module', type of schooling and open-ended questions for barriers to education. The out-of-school rate was 16/62 (26%). The level of schooling (i.e. not in school, special education or mainstream class) was strongly associated with difficulties in mobility, self-care, communication, controlling behaviour and getting along with other children. Five themes identified by caregivers as barriers to education were 'school infrastructure', 'inclusive opportunity, staffing and teacher's training', 'social environment', 'child's intrinsic factor' and 'practical limitations'. Practical solutions to overcome the significant barriers to education are required to address the unmet educational needs of CWND.
  5. Joseph CJ, Khoo TB, Lee KY
    BMJ Case Rep, 2017 Jan 06;2017.
    PMID: 28062435 DOI: 10.1136/bcr-2016-218044
    An infant, who was born preterm at 36 weeks, presented with fever and ulcer at umbilical region which progressed to necrotising fasciitis of anterior abdominal wall. He was treated with intravenous penicillin, intravenous cloxacillin and local application of medicated honey. Subsequently, he required wound debridement. Postoperatively, he required prolonged invasive ventilation due to poor respiratory effort which was associated with hypotonia and areflexia. Nerve conduction study revealed absent responses. The diagnosis of infant botulism was made based on the clinical presentation, nerve conduction study and his clinical progress. Botulinum immunoglobulin was not available. He was treated with intravenous immunoglobulin and oral pyridostigmine. He was successfully extubated after 37 days, and currently the patient is doing well.
  6. Siti Aishah Abdul Wahab, Yusnita Yakob, Khoo,Teik-Beng, Sangita Dharshini Terumalay, Vigneswari Ganesan, Teh,Chee-Ming, et al.
    Neurology Asia, 2017;22(2):99-111.
    MyJurnal
    Background & Objective: SCN1A gene which encodes for sodium channel alpha 1 subunit has been
    found to be the most common mutated gene in patients with epilepsy. This study aims to characterize the
    SCN1A mutations as well as to describe genotype and phenotype association in children with SCN1Arelated
    infantile-onset epileptic encephalopathies in Malaysia.

    Methods: Children with infantile-onset
    epileptic encephalopathy mostly suspected to have Dravet syndrome who had mutational analysis for
    SCN1A gene from hospitals all over Malaysia were included in the study. Their epilepsy syndrome
    diagnosis was classified into severe myoclonic epilepsy in infancy and its variants. Polymerase chain
    reaction and bidirectional sequencing were used to identify SCN1A mutations.

    Results: A total of 38
    children with heterozygous mutations were analysed, 22 (57.9%) of which were novel mutations.
    Truncated mutations were the most common mutation type (19, 50%). Other mutation types were
    missense mutations (14, 36.8%), splice site mutations (4, 10.5%) and in-frame deletion (1, 2.6%). The
    mean age of seizure onset was 4.7 months. Seizure following vaccination was observed in 26.3% of
    the children. All of them had drug resistant epilepsy. There was no significant association between
    the type of mutation with the syndromic diagnosis, age of seizure onset, tendency of the seizures to
    cluster or having status epilepticus, mean age when developmental delay was observed and response
    to various antiepileptic drugs.

    Conclusion: This study expands the spectrum of SCN1A mutations and proves the importance of
    SCN1A gene testing in diagnosing infantile-onset epileptic encephalopathies patients. Although, our
    study does not support any clinically meaningful genotype-phenotype association for SCN1A-related
    infantile-onset epileptic encephalopathies, the clinical characteristics of our cohort are similar to those
    that have been described in previous studies.
  7. Tan JW, Khoo TB, Burharudin NF, Mohamed Shah N
    Epilepsy Behav, 2020 10;111:107317.
    PMID: 32693382 DOI: 10.1016/j.yebeh.2020.107317
    PURPOSE: Self-management is crucial in the management of chronic diseases. However, information is limited on medication self-management among parents of children with epilepsy. This study aimed to assess medication self-management among parents of children with epilepsy and its association with sociodemographic data, clinical characteristics, antiepileptic drug (AED) regimen complexity, and parent self-reported AED adherence.

    METHOD: A cross-sectional survey was conducted at a tertiary care center in Malaysia from February 2019 to June 2019. Parents of children with epilepsy who were on AED for at least 3 months and aged ≤18 years old were recruited. Medication self-management was assessed using a validated Pediatric Epilepsy Medication Self-Management Questionnaire (PEMSQ). A higher total score reflects better medication self-management.

    RESULTS: A total of 166 patients were recruited. The mean ± standard deviation (SD) age of patients was 8.20 ± 5.21 years, and 51.8% and 36.7% of patients have generalized seizure and focal seizure, respectively. The mean ± SD PEMSQ score was 116.2 ± 11.28 from a total score of 135. Among the four domains of PEMSQ, the barriers to treatment contributed to the lowest mean scores. Univariate analysis showed that the following were significantly associated with poorer medication self-management: differences in ethnicity, religion; higher number of medications; presence of comorbidities; inability to swallow tablets; and a more complex AED regimen. Other variables were not significant. Multivariate analysis showed that only ethnicity and presence of comorbidity remained independently significant (R2 = 0.14; F [4, 161] = 6.28; p 

  8. Hadi DA, Mohamed AR, Rethanavelu K, Khoo TB
    Brain Dev, 2022 Jan;44(1):44-49.
    PMID: 34483011 DOI: 10.1016/j.braindev.2021.08.006
    BACKGROUND: Ferric chelate reductase 1 like (FRRS1L) encephalopathy is a rare cause of developmental and epileptic encephalopathy. Only a few cases have been reported thus far and seizures tend to be drug refractory. We report an additional case to highlight the good seizure response to sulthiame.

    CASE REPORT: A boy from non-consanguineous parents presented with history of 'abnormal movements' from 7 months of age. At one year of age, video electroencephalogram (EEG) monitoring demonstrated the 'abnormal movements' to be clonic seizures. Valproate, lamotrigine and clobazam combination were only partially effective at reducing the seizures. Repeat EEG at 1 year 8 months old revealed a continuous spikes-and-waves during slow sleep (CSWS) pattern, prompting a trial of sulthiame. After 2 weeks of sulthiame, seizures ceased completely. The clonic seizures recurred at age 4 years when sulthiame supply was interrupted, but the seizures promptly remitted following sulthiame's resumption. Subtle choreiform movements appeared from age one year and later became more prominent. Whole exome sequencing (WES) identified a homozygous novel variant (nonsense) in the FRRS1L gene (NM_014334.3: c.670C>T:p.Gln224*). He has been seizure free since 4 years of age but remained profoundly delayed.

    CONCLUSION: Sulthiame may have a role in the early treatment of seizures in children with refractory epilepsy due to FRRS1L mutation.

  9. Khoo TB, Kassim AB, Omar MA, Hasnan N, Amin RM, Omar Z, et al.
    Disabil Rehabil, 2009;31(21):1753-61.
    PMID: 19479558 DOI: 10.1080/09638280902751964
    To determine the magnitude and impact of physical disability on Malaysian school-aged children between 7 and less than 18 years old.
  10. Fong CY, Ong FN, Ong LC, Khoo TB, Lee ML
    Spinal Cord, 2020 Sep;58(9):1030-1036.
    PMID: 32060410 DOI: 10.1038/s41393-020-0441-7
    STUDY DESIGN: Cross-sectional study.

    OBJECTIVE: To determine the prevalence and potential risk factors of vitamin D deficiency and insufficiency among Malaysian children with spina bifida.

    SETTING: Four Malaysian tertiary hospitals.

    METHODS: Children with spina bifida were assessed for potential demographic, disease severity and lifestyle risk factors for vitamin D deficiency and insufficiency. Blood for 25-hydroxy vitamin D (25(OH)D) was taken. Vitamin D deficiency was defined as 25(OH)D levels ≤ 37.5 nmol/L and insufficiency as 37.6-50 nmol/L.

    RESULTS: Eighty children aged 2-18 years (42 males) participated in the study. Vitamin D levels ranged from 14 to 105 nmol/L (mean 52.8, SD 19.1). Vitamin D deficiency was identified in 18 (22.5%) and insufficiency in 26 (32.5%) children. Logistic regression analysis showed that skin exposure to sunlight ≤ 21% body surface area (OR: 6.2, CI 1.7-22.9) and duration of sun exposure ≤ 35 min/day (OR: 4.0, CI 1.2-14.1) were significant risk factors for vitamin D deficiency and insufficiency, respectively.

    CONCLUSIONS: Over half (55%) of Malaysian children with spina bifida seen in urban tertiary hospitals have vitamin D insufficiency and deficiency. Lifestyle sun exposure behaviours were risk factors for vitamin D deficiency and insufficiency.

  11. Khoo TB, Tan JW, Ng HP, Choo CM, Bt Abdul Shukor INC, Teh SH
    Int J Clin Pharm, 2017 Jun;39(3):551-559.
    PMID: 28417303 DOI: 10.1007/s11096-017-0463-1
    Background There is a lack of large comprehensive studies in developing countries on paediatric in-patient prescribing errors in different settings. Objectives To determine the characteristics of in-patient prescribing errors among paediatric patients. Setting General paediatric wards, neonatal intensive care units and paediatric intensive care units in government hospitals in Malaysia. Methods This is a cross-sectional multicentre study involving 17 participating hospitals. Drug charts were reviewed in each ward to identify the prescribing errors. All prescribing errors identified were further assessed for their potential clinical consequences, likely causes and contributing factors. Main outcome measures Incidence, types, potential clinical consequences, causes and contributing factors of the prescribing errors. Results The overall prescribing error rate was 9.2% out of 17,889 prescribed medications. There was no significant difference in the prescribing error rates between different types of hospitals or wards. The use of electronic prescribing had a higher prescribing error rate than manual prescribing (16.9 vs 8.2%, p 
  12. Fong CY, Chang WM, Kong AN, Rithauddin AM, Khoo TB, Ong LC
    Epilepsy Behav, 2018 03;80:15-20.
    PMID: 29396357 DOI: 10.1016/j.yebeh.2017.12.032
    BACKGROUND: Children with epilepsy (CWE) are at risk of impaired quality of life (QOL), and achieving a good QOL is an important treatment goal among CWE. To date, there are no published multiethnic QOL studies in Asia. Our study aimed to: i) investigate the QOL of multiethnic CWE in Malaysia as reported by both the child and parent; ii) determine the level of agreement between child-self report and parent-proxy report QOL; and iii) explore potential correlates of sociodemographic, epilepsy characteristics, and family functioning with QOL in CWE.

    METHODS: Cross-sectional study of all CWE aged 8-18years old with at least 6months' duration of epilepsy, minimum reading level of primary school education Year 1, and attending mainstream education. Quality of life was measured using the parent-proxy and child self-report of Quality of Life Measurement for Children with Epilepsy (CHEQOL-25) questionnaire. Total and subscale CHEQOL-25 scores were obtained. The levels of parent-child agreement were determined using intraclass correlation coefficients (ICC). Family functioning was assessed using the General functioning subscale (GF-12).

    RESULTS: A total of 115 CWE and their parents participated in the study. In general, Malaysian parents rated children's total CHEQOL-25 scores poorer than the children themselves [mean total parent score: 68.56 (SD: 10.86); mean total child score: 71.82 (SD: 9.55)]. Agreement between child and parent on the CHEQOL-25 was poor to moderate (ICC ranged from 0.31-0.54), with greatest discordance in the epilepsy secrecy domain (ICC=0.31, p=0.026). Parent and child were more likely to agree on more external domains: intrapersonal/social (ICC=0.54, p<0.001) and interpersonal/emotional (ICC=0.50, p<0.001). Malay ethnicity, focal seizure and high seizure frequency (≥1 seizure per month) were associated with lower CHEQOL-25 scores. There was a significant but weak correlation between GF-12 and parent-proxy CHEQOL-25 Total Scores (r=-0.186, p=0.046).

    CONCLUSION: Our results emphasize the importance to have the child's perspective of their QOL as the level of agreement between the parent and child reported scores were poor to moderate. Malaysian CWE of Malay ethnicity, those with focal seizures or high seizure frequency are at risk of poorer QOL.

  13. Mizuguchi T, Nakashima M, Moey LH, Ch'ng GS, Khoo TB, Mitsuhashi S, et al.
    J Hum Genet, 2019 Apr;64(4):347-350.
    PMID: 30626896 DOI: 10.1038/s10038-018-0556-2
    We report the second case of early infantile epileptic encephalopathy (EIEE) arising from a homozygous truncating variant of NECAP1. The boy developed infantile-onset tonic-clonic and tonic seizures, then spasms in clusters. His electroencephalogram (EEG) showed a burst suppression pattern, leading to the diagnosis of Ohtahara syndrome. Whole-exome sequencing revealed the canonical splice-site variant (c.301 + 1 G > A) in NECAP1. In rodents, Necap1 protein is enriched in neuronal clathrin-coated vesicles and modulates synaptic vesicle recycling. cDNA analysis confirmed abnormal splicing that produced early truncating mRNA. There has been only one previous report of a mutation in NECAP1 in a family with EIEE; this was a nonsense mutation (p.R48*) that was cited as EIEE21. Decreased mRNA levels and the loss of the WXXF motif in both the families suggests that loss of NECAP1 function is a common pathomechanism for EIEE21. This study provided additional support that synaptic vesicle recycling plays a key role in epileptogenesis.
  14. Kong AN, Fong CY, Ng CC, Mohamed AR, Khoo TB, Ng RL, et al.
    Seizure, 2020 Jul;79:103-111.
    PMID: 32464532 DOI: 10.1016/j.seizure.2020.05.009
    PURPOSE: Children with epilepsy (CWE) are at risk of vitamin D deficiency. Single nucleotide polymorphisms (SNPs) affecting the vitamin D pathway are potentially important risk factors for serum 25-hydroxyvitamin D [25(OH)D] concentration. The aims of our study were to evaluate the association of vitamin d-related SNPs to serum 25(OH)D concentrations in Malaysian CWE.

    METHODS: Cross-sectional study of Malaysian ambulant CWE on antiseizure medication for >1 year. Sixteen SNPs in 8 genes (GC, VDR, CYP2R1, CYP24A1, CYP27B1, CYP27A1, CYP3A4, NADSYN1/DHCR7) were genotyped. Linear and logistic regression models and co-variates adjusted analyses were used. SNPs with significant associations were further analysed in a group of ethnically-matched healthy Malaysian children.

    RESULTS: 239 CWE were recruited (52.7% Malay, 24.3% Chinese and 23.0% Indian) with mean serum 25(OH)D of 58.8 nmol/L (SD 25.7). Prevalence of vitamin D deficiency (≤37.5 nmol/L) was 23.0%. Minor allele of GC-rs4588-A was associated with lower serum 25(OH)D in the meta-analysis of both CWE (β -8.11, P = 0.002) and Malaysian healthy children (β -5.08, P < 0.001), while VDR-rs7975232-A was significantly associated with reduced odds of vitamin D deficiency in Malay subgroup of CWE (OR: 0.16; 95% CI: 0.06-0.49; P = 0.001) and this association was not found in the healthy children group.

    CONCLUSIONS: Our results suggest that GC-rs4588 is associated with lower serum 25(OH)D concentration in both Malaysian CWE and healthy children, while VDR-rs7975232A is associated with lower risk of vitamin D deficiency in Malaysian CWE of Malay ethnicity. Our findings may assist in the genetic risk stratification of low vitamin D status among CWE.

  15. Fong CY, Kong AN, Poh BK, Mohamed AR, Khoo TB, Ng RL, et al.
    Epilepsia, 2016 08;57(8):1271-9.
    PMID: 27378185 DOI: 10.1111/epi.13443
    OBJECTIVE: Long-term use of antiepileptic drugs (AEDs) is a significant risk factor for vitamin D deficiency in children with epilepsy. The aims of our study were to evaluate the prevalence and risk factors for vitamin D deficiency among Malaysian children with epilepsy.

    METHODS: Cross-sectional study of ambulant children with epilepsy on long-term AEDs for >1 year seen in three tertiary hospitals in Malaysia from April 2014 to April 2015. Detailed assessment of pubertal status, skin pigmentation, sunshine exposure behavior, physical activity, dietary vitamin D and calcium intake, anthropometric measurements and bone health blood tests (vitamin D, alkaline phosphatase, calcium, phosphate, and parathyroid hormone levels) were obtained on all patients. Vitamin D deficiency was defined as 25-hydroxy vitamin D [25(OH)D] levels ≤35 nmol/L and insufficiency as 25(OH)D levels of 36-50 nmol/L.

    RESULTS: A total of 244 children (146 male) participated in the study. Ages ranged between 3.7 and 18.8 years (mean 12.3 years). 25(OH)D levels ranged between 7.5 and 140.9 nmol/L (mean 53.9 nmol/L). Vitamin D deficiency was identified in 55 patients (22.5%), and a further 48 (19.7%) had vitamin D insufficiency. Multivariate logistic regression analysis identified polytherapy >1 AED (odds ratio [OR] 2.16, 95% confidence interval [CI] 1.07-4.36), age >12 years (OR 4.16, 95% CI 1.13-15.30), Indian ethnicity (OR 6.97, 95% CI 2.48-19.55), sun exposure time 30-60 min/day (OR 2.44, 95% CI 1.05-5.67), sun exposure time <30 min/day (OR 3.83, 95% CI 1.61-9.09), and female (OR 2.61, 95% CI 1.31-5.20) as statistically significant (p < 0.05) risk factors for vitamin D deficiency.

    SIGNIFICANCE: Despite living in the tropics, a high proportion of Malaysian children with epilepsy are at risk of vitamin D deficiency. Targeted strategies including vitamin D supplementation and lifestyle advice of healthy sunlight exposure behavior should be implemented among children with epilepsy, particularly for those at high risk of having vitamin D deficiency.

  16. Anuar MA, Lee JX, Musa H, Abd Hadi D, Majawit E, Anandakrishnan P, et al.
    Brain Dev, 2023 Nov;45(10):547-553.
    PMID: 37661525 DOI: 10.1016/j.braindev.2023.06.004
    INTRODUCTION: Since the emergence of COVID-19, we have experienced potent variants and sub-variants of the virus with non-specific neurological manifestations. We observed a surge of the Omicron variant of COVID-19 patients with neurological manifestations where less cases of multisystem inflammatory syndrome in children (MIS-C) were reported. This article describes our experience of children with severe and rare neurological manifestations following COVID-19 infection.

    METHODS: This is a retrospective observational case series of patients under 18 years old who fulfilled the WHO COVID-19 case definition and were referred to our paediatric neurology unit at Hospital Tunku Azizah Kuala Lumpur. Their demographic data, neurological symptoms, laboratory and supporting investigations, neuroimaging, treatment and outcomes were collected and analysed.

    RESULTS: There were eleven patients with neurological manifestations who fulfilled the WHO COVID-19 case definition. Nine patients presented with seizures and/or encephalopathy, one patient with eye opsoclonus and another patient with persistent limbs myokymia. Based on the history, clinical, electrophysiological and radiological findings, two of them had febrile infection-related epilepsy syndrome, two had acute disseminated encephalomyelitis, two had acute necrotising encephalopathy of childhood, one each had hemiconvulsion-hemiplegia-epilepsy syndrome, acute encephalopathy with bilateral striatal necrosis, hemi-acute encephalopathy with biphasic seizures and reduced diffusion, infection-associated opsoclonus and myokymia.

    CONCLUSIONS: This case series highlighted a wide spectrum of neurological manifestations of COVID-19 infection. Early recognition and prompt investigations are important to provide appropriate interventions. It is essential that these investigations should take place in a timely fashion and COVID-19 quarantine period should not hinder the confirmation of various presenting clinical syndromes.

  17. Lee VWM, Khoo TB, Teh CM, Heng HS, Li L, Yusof YLM, et al.
    Dev Med Child Neurol, 2023 Sep;65(9):1256-1263.
    PMID: 36748407 DOI: 10.1111/dmcn.15536
    This case series compared clinical variables and various combinations of immunotherapy received with outcomes of patients with severe acute necrotizing encephalopathy (ANE). We performed a retrospective review of clinical variables, immunotherapy received, and outcomes (based on the modified Rankin Scale) in Malaysia between February 2019 and January 2020. Twenty-seven children (12 male), aged 7 months to 14 years (mean 4 years) at diagnosis were included. Of these, 23 had an ANE severity score of 5 to 9 out of 9 (high risk). Eleven patients received tocilizumab (four in combination with methylprednisolone [MTP], seven with MTP + intravenous immunoglobulin [IVIG]) and 16 did not (two received MTP alone, 14 received MTP + IVIG). Nine died. Among the survivors, six had good outcomes (modified Rankin Score 0-2) at 6 months follow-up. All patients who received tocilizumab in combination with MTP + IVIG survived. Twenty children received first immunotherapy within 48 hours of admission. No significant association was found between the timing of first immunotherapy with outcomes. Those with brainstem dysfunction (p = 0.016) were observed to have poorer outcomes. This study showed a trend towards better survival when those with severe ANE were treated with tocilizumab in combination with MTP + IVIG. However, larger studies will be needed to determine the effect of this regime on the long-term outcomes.
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