Mice deficient in microtubule-associated protein MAP1B show a distinct behavioral phenotype and altered retina function

Pangratz-Fuehrer, Susanne; Bubna-Littitz, Hermann; Propst, Friedrich; Reitsamer, Herbert A.

doi:10.1016/j.bbr.2005.06.018

Cited by 12 publications

(14 citation statements)

References 46 publications

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“…The frequency of MAP1B IgG is equivalent to that of amphiphysin IgG as a marker of paraneoplastic autoimmunity (the latter predicts either SCLC or breast adenocarcinoma) and exceeds that of ANNA-2 IgG (also known as anti-Ri; also a predictor of either lung or breast carcinoma) or PCA-Tr IgG (marker of immune response to Hodgkin lymphoma). As Spinal 5 [11] ANNA-1 (n 5 1) Sensory neuropathy (n 5 2), cerebellar (n 5 2), limbic encephalitis (n 5 1), myelopathy (n 5 2)…”

Section: Discussionmentioning

confidence: 99%

“…9 Nevertheless, for cases lacking CRMP5 IgG, it is noteworthy that the MAP1B antigen is expressed in the retina and that mice genetically lacking MAP1B have retinal dysfunction as a phenotypic manifestation. [10][11][12] Neurological manifestations noted in 5 MAP1B-IgG-seropositive patients (chorea, 3; dystonia, 1; dyskinesia, 1) were plausibly attritutable in 4 to coexisting autoimmunity to other SCLC onconeural antigens. 13 Three patients who were CRMP5 IgG positive had chorea; a fourth patient with coexisting amphiphysin IgG had dystonia.…”

Section: Discussionmentioning

confidence: 99%

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Microtubule‐associated protein 1B: Novel paraneoplastic biomarker

Gadoth¹,

Kryzer²,

Fryer³

et al. 2017

Annals of Neurology

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Objective: To report the identification of microtubule-associated protein (MAP) 1B as the antigen of the previously described Purkinje cell cytoplasmic antibody type 2 (PCA-2) antibody, its frequency, and clinical, oncological, and serological associations. Methods: Archival serum or cerebrospinal fluid (CSF) specimens were available from 96 of 118 consecutive PCA-2-IgG-seropositive patients identified during 1993-2016. The autoantigen, defined in mouse brain lysate by Western blot and mass spectrometry, was confirmed by dual immunohistochemical staining using commercial antibodies. The major antigenic region was defined by Western blot using recombinant protein fragments. Results: IgG in 95 of 96 patients' serum or CSF (but in none of 98 healthy or disease control subjects' serum specimens) bound to recombinant MAP1B. A minority (17.5%) of patients' IgG also bound to MAP1A. PCA-2 was often accompanied by additional neural autoantibody markers of small-cell carcinoma, including collapsin responsemediated protein 5 (CRMP5) IgG (26%) or antineuronal nuclear antibody type 1 (ANNA-1) IgG (also known as antiHu; 13%). Neurological manifestations in 95 patients were (in decreasing frequency): peripheral neuropathy, 53%; cerebellar ataxia, dysmetria, or dysarthria, 38%; and encephalopathy, 27%. Cancer (majority small-cell lung carcinoma [SCLC]) was detected in 66 of 84 evaluated patients (79%). The MAP1B (PCA-2) autoantibody detection rate, among approximately 70,000 patients undergoing service neural autoantibody evaluation in 2015, was 0.024%, equaling amphiphysin IgG (0.026%) and more common than ANNA-2 (also known as anti-Ri; 0.016%) and PCA-Tr (also known as delta/notch-like epidermal growth factor-related receptor [DNER]; 0.006%). Interpretation: MAP1B, the PCA-2 autoantigen, represents a novel target in paraneoplastic neurological disorders and has high predictive value for SCLC. Its relatively high prevalence, compared with other recognized paraneoplastic neural autoantibodies, justifies its testing in comprehensive paraneoplastic neural autoantibody evaluation. ANN NEUROL 2017;81:266-277 I n 2000, Vernino and Lennon 1 reported 10 patients with neurological autoimmunity, 9 of whom had imaging or histopathologic evidence of lung cancer (8 biopsyproven small-cell lung carcinoma [SCLC]). Their serum IgG bound to a protein of 280kDa mass in both rodent brain and a SCLC line and yielded a characteristic staining pattern on rodent neural tissues. The autoantibody was named Purkinje cell cytoplasmic antibody type 2 (PCA-2), to distinguish it from a biomarker of ovarian and breast cancer-related cerebellar degeneration, PCA-1 (also known as anti-Yo). 2 The neurological associations initially reported for PCA-2 were mixed, of subacute onset and predominantly central (brainstem and limbic encephalitis, cerebellar ataxia), but some patients had peripheral manifestations (Lambert-Eaton myasthenic syndrome [LEMS] or autonomic or motor neuropathy). 1 Our study extends the immunochemical characteristization of PCA-2 IgG an...

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Microtubule‐associated protein 1B: Novel paraneoplastic biomarker

Gadoth¹,

Kryzer²,

Fryer³

et al. 2017

Annals of Neurology

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“…MAP1B deficiency is shown to be accompanied by abnormal actin microfilament polymerization and dramatic changes in the activity of small GTPases controlling the actin cytoskeleton [ 23 ]. Mice deficient in Map1b showed impaired long-term potentiation [ 24 ] and also a distinct behavioral phenotype and altered retinal function [ 25 ].…”

Section: Discussionmentioning

confidence: 99%

Autism and Intellectual Disability-Associated KIRREL3 Interacts with Neuronal Proteins MAP1B and MYO16 with Potential Roles in Neurodevelopment

et al. 2015

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Cell-adhesion molecules of the immunoglobulin superfamily play critical roles in brain development, as well as in maintaining synaptic plasticity, the dysfunction of which is known to cause cognitive impairment. Recently dysfunction of KIRREL3, a synaptic molecule of the immunoglobulin superfamily, has been implicated in several neurodevelopmental conditions including intellectual disability, autism spectrum disorder, and in the neurocognitive delay associated with Jacobsen syndrome. However, the molecular mechanisms of its physiological actions remain largely unknown. Using a yeast two-hybrid screen, we found that the KIRREL3 extracellular domain interacts with brain expressed proteins MAP1B and MYO16 and its intracellular domain can potentially interact with ATP1B1, UFC1, and SHMT2. The interactions were confirmed by co-immunoprecipitation and colocalization analyses of proteins expressed in human embryonic kidney cells, mouse neuronal cells, and rat primary neuronal cells. Furthermore, we show KIRREL3 colocalization with the marker for the Golgi apparatus and synaptic vesicles. Previously, we have shown that KIRREL3 interacts with the X-linked intellectual disability associated synaptic scaffolding protein CASK through its cytoplasmic domain. In addition, we found a genomic deletion encompassing MAP1B in one patient with intellectual disability, microcephaly and seizures and deletions encompassing MYO16 in two unrelated patients with intellectual disability, autism and microcephaly. MAP1B has been previously implicated in synaptogenesis and is involved in the development of the actin-based membrane skeleton. MYO16 is expressed in hippocampal neurons and also indirectly affects actin cytoskeleton through its interaction with WAVE1 complex. We speculate KIRREL3 interacting proteins are potential candidates for intellectual disability and autism spectrum disorder. Moreover, our findings provide further insight into understanding the molecular mechanisms underlying the physiological action of KIRREL3 and its role in neurodevelopment.

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“…By placing them in the start box and being offered food pellets in the goal box, the time to reach the goal box and the number of wrong decisions were recorded. Then 14 days later the memory recall/ retention was tested (Lohninger et al, 2001;Pangratz-Fuhrer et al, 2005;Pollak et al, 2005).…”

Section: Cognitive Testsmentioning

confidence: 99%

The neuropsychopharmacological effects of Catha edulis in mice offspring born to mothers exposed during pregnancy and lactation

Bedada

Engidawork

2009

Phytotherapy Research

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Chewing fresh leaves of the khat plant (Catha edulis Forsk) is a deep rooted and widespread habit in East Africa and the Middle East. Although a body of knowledge exists about the adverse effects of khat during pregnancy, data are sparse with regard to the consequences of long-term exposure during pregnancy and lactation. The present work, therefore, was initiated to evaluate the neuropsychopharmacological effects of Catha edulis exposure during pregnancy and lactation in mice at postnatal day 28. To this effect, a lyophilized extract of khat (100 mg/kg, K100 and 200 mg/kg, K200), amphetamine (1 mg/kg, positive control, AMP), and a similar volume of 2% v/v Tween-80 in distilled water (negative control, CONT) were administered daily to pregnant mice from gestational day 6 until weaning. Neuropsychopharmacological measurements were done by making use of a battery of neurobehavioural and cognitive tests. Moreover, toxicity to liver and kidney was also evaluated by determining biochemical markers for possible tissue damage. K200 produced significant motor in-coordination and emotional instability; as revealed by impairment in both cliff avoidance (p < 0.01) and forelimb grip strength (p < 0.001), as well as by an increase in stereotyped behaviour such as grooming (p < 0.05), and in the percent of time spent in open arms (p < 0.05). On the other hand, K100 had an effect only on grip strength where a decrement was noted (p < 0.01). A different pattern emerged with AMP whilst it increased duration of sniffing (p < 0.05), neither grip strength nor the time spent in the open arm was affected compared with CONT. Increased latency to reach the goal box and the number of wrong decisions (p < 0.05) in both the learning and the recall tests was observed with K200 and AMP. By contrast, both doses of khat and AMP equally affected performance in the Y-maze (p < 0.05). Alterations in the biochemical indices of liver and kidney function were also noted with AMP and K200. These findings indicate that khat exposure produces dose-related central and peripheral effects during pregnancy and lactation which might pose a serious impediment to the physical and mental development of the offspring.

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Mice deficient in microtubule-associated protein MAP1B show a distinct behavioral phenotype and altered retina function

Cited by 12 publications

References 46 publications

Microtubule‐associated protein 1B: Novel paraneoplastic biomarker

Microtubule‐associated protein 1B: Novel paraneoplastic biomarker

Autism and Intellectual Disability-Associated KIRREL3 Interacts with Neuronal Proteins MAP1B and MYO16 with Potential Roles in Neurodevelopment

The neuropsychopharmacological effects of Catha edulis in mice offspring born to mothers exposed during pregnancy and lactation

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