Monozygotic twin cases of the agenesis of the corpus callosum with schizophrenic disorder

Motomura, Naoyasu; Satani, Seiji; Inaba, Masaaki

doi:10.1046/j.1440-1819.2002.00944.x

Cited by 20 publications

(18 citation statements)

References 14 publications

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“…However, the background strains (C57BL/6J and CBA/Ca) of the Disc1 tr transgenic mice reported in this study are known to have an intact corpus callosum. Consistent with our findings, a complete agenesis of the corpus callosum is rare in schizophrenia (Motomura et al, 2002;Chinnasamy et al, 2006;Paul et al, 2007). However, mild alterations in interhemispheric callosal connections may be relatively common in schizophrenia and in autism (Innocenti et al, 2003;Miyata et al, 2007).…”

Section: Discussionsupporting

confidence: 91%

Schizophrenia-Related Neural and Behavioral Phenotypes in Transgenic Mice Expressing TruncatedDisc1

Shen¹,

Luo²,

Nakamoto³

et al. 2008

J. Neurosci.

238

204

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Disrupted-in-Schizophrenia-1 (DISC1), identified by positional cloning of a balanced translocation (1;11) with the breakpoint in intron 8 of a large Scottish pedigree, is associated with a range of neuropsychiatric disorders including schizophrenia. To model this mutation in mice, we have generated Disc1 tr transgenic mice expressing 2 copies of truncated Disc1 encoding the first 8 exons using a bacterial artificial chromosome (BAC). With this partial simulation of the human situation, we have discovered a range of phenotypes including a series of novel features not previously reported. Disc1 tr transgenic mice display enlarged lateral ventricles, reduced cerebral cortex, partial agenesis of the corpus callosum, and thinning of layers II/III with reduced neural proliferation at midneurogenesis. Parvalbumin GABAergic neurons are reduced in the hippocampus and medial prefrontal cortex, and displaced in the dorsolateral frontal cortex. In culture, transgenic neurons grow fewer and shorter neurites. Behaviorally, transgenic mice exhibit increased immobility and reduced vocalization in depression-related tests, and impairment in conditioning of latent inhibition. These abnormalities in Disc1 tr transgenic mice are consistent with findings in severe schizophrenia.

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Section: Discussionsupporting

confidence: 91%

Schizophrenia-Related Neural and Behavioral Phenotypes in Transgenic Mice Expressing TruncatedDisc1

Shen¹,

Luo²,

Nakamoto³

et al. 2008

J. Neurosci.

238

204

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“…Although clinically similar to later onset schizophrenia, this population exhibits greater premorbid impairments and a more chronic course of illness, possibly due to a more severe genetic and/or environmentally determined disturbance of brain development 14 . Impaired intelligence disturbances are very common in cases of callosal agenesis, probably because of this neurodevelopmental insult 3 . Several cases with some degree of CCA combined with psychotic symptoms have been reported, but this is the fi rst report of early onset schizophrenia.…”

Section: Discussionmentioning

confidence: 99%

“…The development of the CC is associated with the formation of the hippocampus and cingulate cortex, who are implicated in the pathophysiology of schizophrenia. However, whether the CC is abnormal in patients with schizophrenia is unclear 3,4 . The incidence of agenesis is diffi cult to estimate because is rare [5][6][7] and not routinely detected.…”

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confidence: 99%

“…In most cases accomplished by schizophrenia the agenesis is partial, involving the anterior part of the CC 3,10,11 . Schizophrenia has been described in association with pure complete agenesis in 5 case reports, usually accompanied by another brain malformations 3,5,10,11 . Its symptoms occurred in adulthood and there were no particular psychopathological features.…”

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confidence: 99%

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Total agenesis of the corpus callosum in a patient with childhood-onset schizophrenia

Hallak

Crippa

Pinto

et al. 2007

Arq. Neuro-Psiquiatr.

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-The hypothesis that schizophrenia involves aberrant inter-hemispheric communication has a long pedigree, however its precise role remains unclear. We therefore report the case of a total agenesis of the corpus callosum in a 21-year-old man with childhood-onset schizophrenia. The presence of schizophrenia with very early onset on absence of corpus callosum offers an opportunity to examine neurodevelopmental model and theories regarding to interhemispheric communication in the pathogenesis of psychosis.KEY WORDS: schizophrenia, corpus callosum, agenesis. Agenesia completa de corpo caloso em um paciente com esquizofrenia de início na infânciaRESUMO -A hipótese que a esquizofrenia envolve comunicação inter-hemisférica aberrante possui longa tradição, entretanto seu papel permanece incerto. Nós relatamos um caso de agenesia total do corpo caloso em um homem de 21 anos portador de esquizofrenia de início na infância. A associação de esquizofrenia de início precoce na ausência de corpo caloso oferece uma oportunidade para exame do modelo neurodesenvolvimental e de teorias que envolvem a comunicação interemisférica na patogênese da psicose. PALAVRAS-CHAVE: esquizofrenia, corpo caloso, agenesia.

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“…Prevalence estimates of ACC in the general population are low, and range from 0.073 [5] to 2.2% [6] . ACC has been reported to be found in patients with alien hand syndrome (AHS), a phenomenon that is characterized by the subjective feeling that the hand does not belong to the patient [7] .A growing number of cases have described ACC in patients with schizophrenia [8][9][10] . We report a case of a young woman with the coenesthetic subtype of schizophrenia presenting partial ACC and AHS.…”

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confidence: 99%

First-Episode ‘Coenesthetic’ Schizophrenia Presenting with Alien Hand Syndrome and Partial Agenesis of the Corpus Callosum

et al. 2008

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We describe the case of a 23-year-old Caucasian woman who presented with alien hand syndrome and a first episode of the coenesthetic subtype of schizophrenia. ‘Alienness’ of her non-dominant hand was intimately phenomenologically associated with the onset of first-psychosis. Cerebral MRI revealed a partial agenesis of the corpus callosum with a complete absence of the rostrum, hypoplastic anterior and inferior genu, and a hypoplastic splenium. This case suggests that this syndrome can occur with the development of a functional disconnection syndrome involving the anterior callosum, and in this case the ‘second hit’ proposed to occur in early adulthood in schizophrenia may have interacted with her earlier neurodevelopmental lesion to result in a combination of psychosis and alien hand syndrome.

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Monozygotic twin cases of the agenesis of the corpus callosum with schizophrenic disorder

Cited by 20 publications

References 14 publications

Schizophrenia-Related Neural and Behavioral Phenotypes in Transgenic Mice Expressing TruncatedDisc1

Schizophrenia-Related Neural and Behavioral Phenotypes in Transgenic Mice Expressing TruncatedDisc1

Total agenesis of the corpus callosum in a patient with childhood-onset schizophrenia

First-Episode ‘Coenesthetic’ Schizophrenia Presenting with Alien Hand Syndrome and Partial Agenesis of the Corpus Callosum

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