Anti-NMDA-receptor encephalitis presenting with catatonia and neuroleptic malignant syndrome in patients with intellectual disability and autism

Kiani, Reza; Lawden, Mark; Eames, Penelope J.; Critchley, Peter; Bhaumik, Sabyasachi; Odedra, Sunita; Gumber, Rohit

doi:10.1192/pb.bp.112.041954

Cited by 36 publications

(33 citation statements)

References 27 publications

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“…Furthermore, loss of skills beyond infancy—such as catatonic symptoms in adolescence or adulthood—may occur at higher rates than previously thought [Breen & Hare, ]. Although research is limited, further research into the similarities and differences between loss of skills in infancy and adolescence or adulthood is warranted and may suggest directions for investigation of etiology such as autoimmunity [Kiani et al, ] and genetic factors [Breckpot et al, ], as well as possible treatments [Dhossche, ]. Importantly, attempts to explore etiology should move beyond retrospective parent‐report of regression, utilizing prospective designs and considering dimensional definitions of regression alongside categorical approaches.…”

Section: Evidence For Regression and Prevalence Ratesmentioning

confidence: 99%

Regression in autism spectrum disorder: Reconciling findings from retrospective and prospective research

et al. 2018

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The way in which the behavioral manifestations of autism spectrum disorder (ASD) emerge in infancy is variable. Regression-loss of previously acquired skills-occurs in a subset of children. However, the etiology and significance of regression remains unclear. Until recently, investigation of regression relied on retrospective report by parents or examination of home videos from early in life. However, home videos and retrospective report of the nature and timing of regression, and association with factors such as illness or immunization, is potentially subject to bias. The advent of prospective studies of infant siblings at familial high-risk of ASD has the potential to document regression as it occurs. Recent research has suggested that subtle loss of skills occurs in a larger proportion of children with ASD than previously assumed; however, there are few reports of clear-cut regressions, such as that involving dramatic loss of language and other established skills, in the prospective literature. This could be because of the following: clear-cut regression occurs less commonly than parent report suggests, study design limits the potential to detect regression, or there are differences between multiplex and simplex families in the rate of de novo genetic mutations and therefore regression risk. This review will bring together literature from retrospective and prospective research and attempt to reconcile diverging findings, with a specific focus on methodological issues. Changing conceptualizations of regression will be discussed, as well as etiological factors that may be associated with regression. The main challenges that need to be addressed to measure regression in prospective studies will be set out.Lay Summary: Regression-a loss of previously established skills-occurs in a subset of children with ASD. Parental recall is not always accurate but studying younger siblings of children with ASD, 10-20% of whom will develop ASD, should make it possible to measure regression as it occurs. Clear-cut regression, like loss of language, has not often been reported in infant sibling studies, but recent research suggests that gradual loss of social engagement might be more common. This review looks at the evidence for regression from infant sibling studies and asks how study design affects the likelihood of capturing regression.

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Section: Evidence For Regression and Prevalence Ratesmentioning

confidence: 99%

Regression in autism spectrum disorder: Reconciling findings from retrospective and prospective research

et al. 2018

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“…Other differentials which could be kept in this case are tardive stereotypies, malignant catatonia, and anti‐NMDA (N‐methyl‐D‐aspartate) receptor encephalitis . Stereotypies reported as part of tardive dyskinesia are mainly orofacial and include tongue protrusion, chewing, grimacing, and lip pursing .…”

Section: Discussionmentioning

confidence: 91%

Stereotypy in Neuroleptic Malignant Syndrome

Pandey

2019

Movement Disord Clin Pract

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Various types of movement disorders are described in patients of neuroleptic malignant syndrome (NMS), like tremors, dystonia, opisthotonos, trismus, and chorea, but none of the reports in our knowledge have reported stereotypic movements in these patients. 1 Here, we report on a case of NMS presenting with stereotypic movements of the right hand, which rapidly disappeared following treatment of the underlying condition. CaseAn 80-year-old woman presented in emergency with complaints of fever, stiffness of all four limbs causing difficulty in walking, altered sensorium, and repeated tapping movement of the right upper limb for 3 days. She also had episodes of palpitations, urinary incontinence, respiratory distress, and postural dizziness suggestive of autonomic dysfunction. For the last 1 month, she was taking a tablet of risperidone (2 mg/day) for agitated behavior and excessive talking. At the time of admission, she was febrile, in altered sensorium, and dehydrated. Her blood pressure was labile (130/80-180/110 mm Hg), she had tachycardia (110 beats/minute), and tachypnea (30 breaths/minute). There was generalized rigidity (both axial and appendicular). We also observed a prolonged repetitive, rhythmic, and purposeless tapping of her right upper limb in a stereotypic manner (see Video, Segment 1). On hematological investigations, her total leukocyte counts (TLC; 18,000/mm 3 ) and creatine phosphokinase (CPK; 1,285 U/L) were very high. MRI of the brain showed agerelated atrophy with chronic ischemic changes. Her EEG and cerebrospinal fluid examination were normal. She was diagnosed as NMS, the offending drug risperidone was stopped, and she was treated with antibiotics (injection ceftriaxone 1 g, intravenous three times daily for 7 days) and a tablet of bromocriptine (2.5 mg three times daily, which was increased to 5 mg three times daily after 3 days). Her sensorium improved over the next 7 days, and her CPK levels came down to 235 U/L and TLC decreased to 9,700/mm 3 . Stereotypic movements slowly decreased in severity, but the pattern remained the same. These movements completely disappeared by day 15, when she was discharged (see Video, Segment 2). DiscussionStereotypies are involuntary, patterned, coordinated, repetitive, rhythmic, seemingly purposeless movements or utterances. 2 The right-hand stereotypic movements in our patient started and disappeared with NMS symptoms. So, these stereotypic movements were most likely the part of a clinical spectrum of NMS. Stereotypies have been reported secondary to developmental delay, drugs, basal ganglia, and thalamic lesions, including stroke, and heredo-degenerative causes, including dementia. 2-4 But the presence of stereotypies in NMS has not been reported to date to the best of our knowledge.Other differentials which could be kept in this case are tardive stereotypies, malignant catatonia, and anti-NMDA (N-methyl-D-aspartate) receptor encephalitis. 1,[5][6][7] Stereotypies reported as part of tardive dyskinesia are mainly orofacial and include tongue protrusion...

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“…It is then difficult to discern whether subsequent abnormal movements and autonomic instability are a result of neuroleptic induced extrapyramidal effects, NMS, or are a part of the natural disease progression. Several case reports have described concurrent NMS and anti-NMDA receptor encephalitis in adults (Kiani et al 2014;Chmayssani et al 2011). One case report describes malignant catatonia and increased creatine kinase in a 17-year-old girl who was treated with antipsychotics for anti-NMDA receptor encephalitis (Consoli et al 2011).…”

Section: Discussionmentioning

confidence: 97%

Neuroleptic Malignant Syndrome in a Boy with NMDA Receptor Encephalitis

Berg

Byrne

Coffey

2015

Journal of Child and Adolescent Psychopharmacology

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Chief Complaint and Presenting ProblemM . is an 11-year-old African American boy who was referred to the psychiatry consult liaison service eight days after admission to an epilepsy monitoring unit for behavioral disruption and inability to sleep. History of Present IllnessParents reported that M. had been a normally developing fifth grader who did well in school. M. developed the sudden onset of seizures six weeks prior to admission; his family found him curled in a fetal position with irregular jerking movements. Parents transported him to the emergency department, and an EEG demonstrated absence seizures. M. was hospitalized for several days, treated with valproic acid, and then discharged to outpatient followup. During this admission, M. was evaluated by the psychiatry team for anxiety, as he had expressed fear that he was going to die as a result of his illness. He also endorsed auditory hallucinations of hearing voices, at one point telling him to have more seizures. Outpatient psychotherapy was recommended.In the following six weeks, M. had no identified seizures but experienced many new symptoms. M.'s attention, concentration, and academic performance declined dramatically, according to his teachers. He had multiple crying fits and mood swings each day. M. became increasingly somnolent, but had difficulty sleeping each night and began to slur his speech. Teachers recommended an Individualized Education Plan one month prior to referral due to his significant academic decline.One day prior to admission, M. presented to the emergency department with four absence seizures, and was loaded with valproic acid and discharged. On the day of admission, M. again presented to the emergency department with two brief absence seizures culminating in a generalized tonic-clonic seizure that lasted 30 minutes and required intravenous midazolam. M.'s serum valproic acid level was 126 (therapeutic range 50-100 ug/mL) and ammonia was 102 (normal range 11-35 uM/L). His presumptive diagnosis was hyperammonemia due to valproic acid toxicity, and he was admitted.Following admission, M. experienced episodes of dystonic posturing and nystagmus, only some of which were associated with epileptiform activity on long-term electroencephalogram (EEG) monitoring. His EEG consistently showed diffuse delta wave slowing. At the time of psychiatry consultation, M. was in full upper-extremity restraints due to agitation, and his verbal capacity had regressed to repeating the same word or phrase, often speaking in unintelligible sounds entirely. He followed commands inconsistently, and had not slept more than three hours a night in over a week. M. was given 0.3 mg of risperidone for agitation the night before psychiatry consultation. Psychiatry consultation was requested to further advise on medication management.After extensive assessment, psychiatry concluded that M. had suffered a grand mal seizure of unknown etiology and had recent cognitive decline consistent with delirium. In addition to continuing the extensive workup already underway, t...

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Anti-NMDA-receptor encephalitis presenting with catatonia and neuroleptic malignant syndrome in patients with intellectual disability and autism

Cited by 36 publications

References 27 publications

Regression in autism spectrum disorder: Reconciling findings from retrospective and prospective research

Regression in autism spectrum disorder: Reconciling findings from retrospective and prospective research

Stereotypy in Neuroleptic Malignant Syndrome

Neuroleptic Malignant Syndrome in a Boy with NMDA Receptor Encephalitis

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