Neuroleptic malignant syndrome with prolonged catatonia in a dopa-responsive dystonia patient

Ihara, Masafumi; Kohara, N.; Urano, Fumihiro; Ichinose, Hiroshi; Takao, Seiji; Nishida, Takuji; Saiki, Hidemoto; Kawamoto, Yoshitaka; Ikeda, Aya; Takagi, Sho; Shibasaki, Hiroshi

doi:10.1212/wnl.59.7.1102

Cited by 13 publications

(5 citation statements)

References 10 publications

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“…It is presumed that at least some patients with psychiatric disturbances received psychiatric pharmacothera py; however, the literature is scarce. There are 2 published cases of adult patients with AD-GTPCHD who received electroconvulsive therapy (ECT) [150,151]. One of the patients with psychosis [151] developed a neuroleptic malignant syndrome due to haloperidol treatment followed by a prolonged catatonic state, which required ECT.…”

Section: Other Supportive Therapiesmentioning

confidence: 99%

“…There are 2 published cases of adult patients with AD-GTPCHD who received electroconvulsive therapy (ECT) [150,151]. One of the patients with psychosis [151] developed a neuroleptic malignant syndrome due to haloperidol treatment followed by a prolonged catatonic state, which required ECT. In the other patient, a combination of SSRI and 5-HTP did not prevent the emergence of a delusional depression; he was therefore treated with ECT.…”

Section: Other Supportive Therapiesmentioning

confidence: 99%

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Consensus guideline for the diagnosis and treatment of tetrahydrobiopterin (BH4) deficiencies

Opladen

López‐Laso

Cortès‐Saladelafont

et al. 2020

Orphanet J Rare Dis

124

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Background: Tetrahydrobiopterin (BH 4) deficiencies comprise a group of six rare neurometabolic disorders characterized by insufficient synthesis of the monoamine neurotransmitters dopamine and serotonin due to a disturbance of BH 4 biosynthesis or recycling. Hyperphenylalaninemia (HPA) is the first diagnostic hallmark for most BH 4 deficiencies, apart from autosomal dominant guanosine triphosphate cyclohydrolase I deficiency and sepiapterin reductase deficiency. Early supplementation of neurotransmitter precursors and where appropriate, treatment of HPA results in significant improvement of motor and cognitive function. Management approaches differ across the world and therefore these guidelines have been developed aiming to harmonize and optimize patient care. Representatives of the International Working Group on Neurotransmitter related Disorders (iNTD) developed the guidelines according to the SIGN (Scottish Intercollegiate Guidelines Network) methodology by evaluating all available evidence for the diagnosis and treatment of BH 4 deficiencies. Conclusion: Although the total body of evidence in the literature was mainly rated as low or very low, these consensus guidelines will help to harmonize clinical practice and to standardize and improve care for BH 4 deficient patients.

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Section: Other Supportive Therapiesmentioning

confidence: 99%

Section: Other Supportive Therapiesmentioning

confidence: 99%

Consensus guideline for the diagnosis and treatment of tetrahydrobiopterin (BH4) deficiencies

Opladen

López‐Laso

Cortès‐Saladelafont

et al. 2020

Orphanet J Rare Dis

124

View full text Add to dashboard Cite

show abstract

“…Usually, catatonic stupor is associated with minor EEG changes or low amplitude background activity. However, we reported relatively high amplitude slow activity in a patient with catatonic stupor with a diagnosis of dopa‐responsive dystonia (Ihara et al 2002). It has been reported that some cases of catatonic stupor are accompanied by relatively high amplitude slow activity (Suzuki et al 2006).…”

Section: Discussionmentioning

confidence: 93%

“…The patient later developed catatonic episodes, and the EEG showed continuous rhythmic, high amplitude slow activity diffusely more anteriorly on the right; this disappeared promptly after diazepam injection. Ihara et al reported a patient with dopa‐responsive dystonia manifesting a malignant syndrome with prolonged catatonia, whose EEG activities were markedly diminished after the diazepam injection (Ihara et al 2002). The quick disappearance of continuous EEG slowing immediately after diazepam infusion supports the diagnosis of catatonia.…”

Section: Discussionmentioning

confidence: 99%

Impairment of the cortical GABAergic inhibitory system in catatonic stupor: a case report with neuroimaging

Iseki

Ikeda²,

Kihara³

et al. 2009

Epileptic Disorders

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We report the case of a 32-year-old patient who presented with catatonic stupor during the course of acute aseptic encephalitis involving the right frontotemporal area. Flumazenil-PET performed during the stupor indicated decreased benzodiazepine receptor binding in the right frontotemporal area where glucose metabolism was preserved as revealed by FDG-PET. An injection of diazepam immediately ameliorated catatonic symptoms and reduced widespread high amplitude slow EEG activities with right frontotemporal predominance. Compared with a SPECT study performed a week earlier, there was no abnormal right-sided anteriorly predominant cerebral hyperperfusion after injection of diazepam. While neither flumazenil-nor FDG-PET could be repeated, and with the caveat that generalized convulsions occurred initially and epilepsia partialis continua was present for two weeks starting on the 23 rd day after illness onset, these findings suggest that in our case the presentation with catatonic stupor may be related to impairment of the cortical GABAergic inhibitory system.

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“…15 Under circumstances in which ECT must be withheld or postponed, the use of amantadine in addition to lorazepam can be effective and safe. 29 Bromocriptine 30 and levodopa 31 may also be considered as adjunctive third-line treatments in patients with refractory catatonia.…”

Section: Discussionmentioning

confidence: 99%

Catatonia in Neurologic and Psychiatric Patients at a Tertiary Neurological Center

Espínola-Nadurille¹,

Ramı́rez-Bermúdez²,

Fricchione³

et al. 2016

JNP

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This study describes the prevalence, phenomenology, treatment, and outcome of neurological patients and psychiatric patients with catatonia at a tertiary neurological center. Clinical variables included nosological diagnoses and complications. Admission length and days with catatonia were used as outcome measures. Of 2,044 patients who were evaluated prospectively, 68 (3.32%) had catatonia, 42 (61.7%) were neurological patients, 19 (27.9%) were psychiatric patients, and 7 (10.2%) had drug-related diagnoses. Of all patients, the ratio of neurological to psychiatric patients was 3:1. Encephalitis was the most common diagnosis (N=26 [38.2%]), followed by schizophrenia (N=12 [17.6%]). Psychiatric patients exhibited a stuporous type of catatonia (15 [83.3%] versus 14 [33.3%], p>0.001), whereas neurological patients exhibited a mixed form of catatonia (25 [59.5%] versus 1 [5.6], p<0.001). Neurological patients had more complications, longer hospitalizations, and more days with catatonia. A total of 56 patients (82.3%) received lorazepam, and 14 patients (20.5%) underwent ECT. Second- and third-line treatments included amantadine, bromocriptine, and levodopa. Catatonia is a prevalent syndrome that can remit with proper and opportune treatment.

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Neuroleptic malignant syndrome with prolonged catatonia in a dopa-responsive dystonia patient

Cited by 13 publications

References 10 publications

Consensus guideline for the diagnosis and treatment of tetrahydrobiopterin (BH4) deficiencies

Consensus guideline for the diagnosis and treatment of tetrahydrobiopterin (BH4) deficiencies

Impairment of the cortical GABAergic inhibitory system in catatonic stupor: a case report with neuroimaging

Catatonia in Neurologic and Psychiatric Patients at a Tertiary Neurological Center

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