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Article ; Online: Circuit imaging biomarkers in preclinical and prodromal Parkinson's disease.

Meles, Sanne K / Oertel, Wolfgang H / Leenders, Klaus L

2021 Volume 27, Issue 1, Page(s) 111

Abstract: Parkinson's disease (PD) commences several years before the onset of motor features. Pathophysiological understanding of the pre-clinical or early prodromal stages of PD are essential for the development of new therapeutic strategies. Two categories of ... ...

Abstract	Parkinson's disease (PD) commences several years before the onset of motor features. Pathophysiological understanding of the pre-clinical or early prodromal stages of PD are essential for the development of new therapeutic strategies. Two categories of patients are ideal to study the early disease stages. Idiopathic rapid eye movement sleep behavior disorder (iRBD) represents a well-known prodromal stage of PD in which pathology is presumed to have reached the lower brainstem. The majority of patients with iRBD will develop manifest PD within years to decades. Another category encompasses non-manifest mutation carriers, i.e. subjects without symptoms, but with a known mutation or genetic variant which gives an increased risk of developing PD. The speed of progression from preclinical or prodromal to full clinical stages varies among patients and cannot be reliably predicted on the individual level. Clinical trials will require inclusion of patients with a predictable conversion within a limited time window. Biomarkers are necessary that can confirm pre-motor PD status and can provide information regarding lead time and speed of progression. Neuroimaging changes occur early in the disease process and may provide such a biomarker. Studies have focused on radiotracer imaging of the dopaminergic nigrostriatal system, which can be assessed with dopamine transporter (DAT) single photon emission computed tomography (SPECT). Loss of DAT binding represents an effect of irreversible structural damage to the nigrostriatal system. This marker can be used to monitor disease progression and identify individuals at specific risk for phenoconversion. However, it is known that changes in neuronal activity precede structural changes. Functional neuro-imaging techniques, such as
MeSH term(s)	Animals ; Biomarkers/blood ; Brain/metabolism ; Brain/pathology ; Cerebrovascular Circulation ; Diagnostic Imaging/methods ; Disease Management ; Disease Susceptibility ; Dopamine/metabolism ; Energy Metabolism ; Fluorodeoxyglucose F18 ; Gene Expression Regulation ; Genetic Predisposition to Disease ; Humans ; Magnetic Resonance Imaging ; Multimodal Imaging ; Neurotransmitter Agents/metabolism ; Parkinson Disease/blood ; Parkinson Disease/diagnosis ; Parkinson Disease/etiology ; Parkinson Disease/metabolism ; Positron-Emission Tomography ; Prodromal Symptoms ; Severity of Illness Index
Chemical Substances	Biomarkers ; Neurotransmitter Agents ; Fluorodeoxyglucose F18 (0Z5B2CJX4D) ; Dopamine (VTD58H1Z2X)
Language	English
Publishing date	2021-09-16
Publishing country	England
Document type	Journal Article ; Review
ZDB-ID	1283676-x
ISSN	1528-3658 ; 1076-1551
ISSN (online)	1528-3658
ISSN	1076-1551
DOI	10.1186/s10020-021-00327-x
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Article: Investigating the aspect of asymmetry in brain-first versus body-first Parkinson's disease.

Lövdal, S S / Carli, G / Orso, B / Biehl, M / Arnaldi, D / Mattioli, P / Janzen, A / Sittig, E / Morbelli, S / Booij, J / Oertel, W H / Leenders, K L / Meles, S K

NPJ Parkinson's disease

2024 Volume 10, Issue 1, Page(s) 74

Abstract: Parkinson's disease (PD) is characterized by a progressive loss of dopaminergic neurons in the substantia nigra. Recent literature has proposed two subgroups of PD. The "body-first subtype" is associated with a prodrome of isolated REM-sleep Behavior ... ...

Abstract	Parkinson's disease (PD) is characterized by a progressive loss of dopaminergic neurons in the substantia nigra. Recent literature has proposed two subgroups of PD. The "body-first subtype" is associated with a prodrome of isolated REM-sleep Behavior Disorder (iRBD) and a relatively symmetric brain degeneration. The "brain-first subtype" is suggested to have a more asymmetric degeneration and a prodromal stage without RBD. This study aims to investigate the proposed difference in symmetry of the degeneration pattern in the presumed body and brain-first PD subtypes. We analyzed
Language	English
Publishing date	2024-03-30
Publishing country	United States
Document type	Journal Article
ZDB-ID	2819218-7
ISSN	2373-8057
ISSN	2373-8057
DOI	10.1038/s41531-024-00685-3
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Article ; Online: Occipital hypometabolism is a risk factor for conversion to Parkinson's disease in isolated REM sleep behaviour disorder.

Carli, Giulia / Meles, Sanne K / Janzen, Annette / Sittig, Elisabeth / Kogan, Rosalie V / Perani, Daniela / Oertel, Wolfgang H / Leenders, Klaus L

European journal of nuclear medicine and molecular imaging

2023 Volume 50, Issue 11, Page(s) 3290–3301

Abstract: Purpose: Isolated REM sleep behaviour disorder (iRBD) patients are at high risk of developing clinical syndromes of the α-synuclein spectrum. Progression markers are needed to determine the neurodegenerative changes and to predict their conversion. ... ...

Abstract	Purpose: Isolated REM sleep behaviour disorder (iRBD) patients are at high risk of developing clinical syndromes of the α-synuclein spectrum. Progression markers are needed to determine the neurodegenerative changes and to predict their conversion. Brain imaging with Methods: Twenty iRBD patients underwent two consecutive Results: Individual hypometabolism t-maps revealed three scenarios: (1) normal Conclusions: Our results suggest that occipital hypometabolism at baseline in iRBD implies a short-term conversion to PD. This might help in stratification strategies for disease-modifying trials.
MeSH term(s)	Humans ; REM Sleep Behavior Disorder/diagnostic imaging ; REM Sleep Behavior Disorder/metabolism ; Parkinson Disease/diagnostic imaging ; Parkinson Disease/metabolism ; Fluorodeoxyglucose F18 ; 3-Iodobenzylguanidine ; Positron-Emission Tomography/methods ; Risk Factors
Chemical Substances	2-carbomethoxy-8-(3-fluoropropyl)-3-(4-iodophenyl)tropane (155797-99-2) ; Iodine-123 (8YWR746RPQ) ; Fluorodeoxyglucose F18 (0Z5B2CJX4D) ; 3-Iodobenzylguanidine (35MRW7B4AD)
Language	English
Publishing date	2023-06-13
Publishing country	Germany
Document type	Journal Article
ZDB-ID	8236-3
ISSN	1619-7089 ; 0340-6997 ; 1619-7070
ISSN (online)	1619-7089
ISSN	0340-6997 ; 1619-7070
DOI	10.1007/s00259-023-06289-y
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Zs.A 1227: Show issues

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Article ; Online: The impact of social network change due to spousal loss: A qualitative study on experiences of older adults who are aging in place.

Vos den Ouden, Willeke / Janssen, Meriam / van Boekel, Leonieke / Leenders, Roger / Luijkx, Katrien

Death studies

2022 Volume 47, Issue 5, Page(s) 559–573

Abstract: Spousal loss due to nursing home admission or death is challenging for the well-being of the remaining partner and for aging in place. We explored: "How does social network change due to spousal loss impact older adults who are aging in place?." In-depth ...

Abstract	Spousal loss due to nursing home admission or death is challenging for the well-being of the remaining partner and for aging in place. We explored: "How does social network change due to spousal loss impact older adults who are aging in place?." In-depth interviews were held with six older women who were aging in place and who lost their spouses in the past two years. Narrative analysis was conducted. Results indicate that the impact varies in three dimensions and that variations within dimensions follow three themes. The results emphasize the complexity of impact and the urgent need for a person-centred approach toward older adults after spousal loss.
MeSH term(s)	Humans ; Female ; Aged ; Independent Living ; Spouses ; Qualitative Research ; Nursing Homes ; Social Networking
Language	English
Publishing date	2022-08-17
Publishing country	United States
Document type	Journal Article
ZDB-ID	632596-8
ISSN	1091-7683 ; 0748-1187
ISSN (online)	1091-7683
ISSN	0748-1187
DOI	10.1080/07481187.2022.2108942
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Article ; Online: Subspace corrected relevance learning with application in neuroimaging.

van Veen, Rick / Tamboli, Neha Rajendra Bari / Lövdal, Sofie / Meles, Sanne K / Renken, Remco J / de Vries, Gert-Jan / Arnaldi, Dario / Morbelli, Silvia / Clavero, Pedro / Obeso, José A / Oroz, Maria C Rodriguez / Leenders, Klaus L / Villmann, Thomas / Biehl, Michael

Artificial intelligence in medicine

2024 Volume 149, Page(s) 102786

Abstract: In machine learning, data often comes from different sources, but combining them can introduce extraneous variation that affects both generalization and interpretability. For example, we investigate the classification of neurodegenerative diseases using ... ...

Abstract	In machine learning, data often comes from different sources, but combining them can introduce extraneous variation that affects both generalization and interpretability. For example, we investigate the classification of neurodegenerative diseases using FDG-PET data collected from multiple neuroimaging centers. However, data collected at different centers introduces unwanted variation due to differences in scanners, scanning protocols, and processing methods. To address this issue, we propose a two-step approach to limit the influence of center-dependent variation on the classification of healthy controls and early vs. late-stage Parkinson's disease patients. First, we train a Generalized Matrix Learning Vector Quantization (GMLVQ) model on healthy control data to identify a "relevance space" that distinguishes between centers. Second, we use this space to construct a correction matrix that restricts a second GMLVQ system's training on the diagnostic problem. We evaluate the effectiveness of this approach on the real-world multi-center datasets and simulated artificial dataset. Our results demonstrate that the approach produces machine learning systems with reduced bias - being more specific due to eliminating information related to center differences during the training process - and more informative relevance profiles that can be interpreted by medical experts. This method can be adapted to similar problems outside the neuroimaging domain, as long as an appropriate "relevance space" can be identified to construct the correction matrix.
MeSH term(s)	Humans ; Neuroimaging ; Positron-Emission Tomography ; Machine Learning ; Parkinson Disease/diagnostic imaging
Language	English
Publishing date	2024-01-24
Publishing country	Netherlands
Document type	Journal Article
ZDB-ID	645179-2
ISSN	1873-2860 ; 0933-3657
ISSN (online)	1873-2860
ISSN	0933-3657
DOI	10.1016/j.artmed.2024.102786
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Zs.A 2547: Show issues

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Article ; Online: Comparison of univariate and multivariate analyses for brain [18F]FDG PET data in α-synucleinopathies.

Carli, Giulia / Meles, Sanne K / Reesink, Fransje E / de Jong, Bauke M / Pilotto, Andrea / Padovani, Alessandro / Galbiati, Andrea / Ferini-Strambi, Luigi / Leenders, Klaus L / Perani, Daniela

NeuroImage. Clinical

2023 Volume 39, Page(s) 103475

Abstract: Background: Brain imaging with [18F]FDG-PET can support the diagnostic work-up of patients with α-synucleinopathies. Validated data analysis approaches are necessary to evaluate disease-specific brain metabolism patterns in neurodegenerative disorders. ... ...

Abstract	Background: Brain imaging with [18F]FDG-PET can support the diagnostic work-up of patients with α-synucleinopathies. Validated data analysis approaches are necessary to evaluate disease-specific brain metabolism patterns in neurodegenerative disorders. This study compared the univariate Statistical Parametric Mapping (SPM) single-subject procedure and the multivariate Scaled Subprofile Model/Principal Component Analysis (SSM/PCA) in a cohort of patients with α-synucleinopathies. Methods: We included [18F]FDG-PET scans of 122 subjects within the α-synucleinopathy spectrum: Parkinson's Disease (PD) normal cognition on long-term follow-up (PD - low risk to dementia (LDR); n = 28), PD who developed dementia on clinical follow-up (PD - high risk of dementia (HDR); n = 16), Dementia with Lewy Bodies (DLB; n = 67), and Multiple System Atrophy (MSA; n = 11). We also included [18F]FDG-PET scans of isolated REM sleep behaviour disorder (iRBD; n = 51) subjects with a high risk of developing a manifest α-synucleinopathy. Each [18F]FDG-PET scan was compared with 112 healthy controls using SPM procedures. In the SSM/PCA approach, we computed the individual scores of previously identified patterns for PD, DLB, and MSA: PD-related patterns (PDRP), DLBRP, and MSARP. We used ROC curves to compare the diagnostic performances of SPM t-maps (visual rating) and SSM/PCA individual pattern scores in identifying each clinical condition across the spectrum. Specifically, we used the clinical diagnoses ("gold standard") as our reference in ROC curves to evaluate the accuracy of the two methods. Experts in movement disorders and dementia made all the diagnoses according to the current clinical criteria of each disease (PD, DLB and MSA). Results: The visual rating of SPM t-maps showed higher performance (AUC: 0.995, specificity: 0.989, sensitivity 1.000) than PDRP z-scores (AUC: 0.818, specificity: 0.734, sensitivity 1.000) in differentiating PD-LDR from other α-synucleinopathies (PD-HDR, DLB and MSA). This result was mainly driven by the ability of SPM t-maps to reveal the limited or absent brain hypometabolism characteristics of PD-LDR. Both SPM t-maps visual rating and SSM/PCA z-scores showed high performance in identifying DLB (DLBRP = AUC: 0.909, specificity: 0.873, sensitivity 0.866; SPM t-maps = AUC: 0.892, specificity: 0.872, sensitivity 0.910) and MSA (MSARP: AUC: 0.921, specificity: 0.811, sensitivity 1.000; SPM t-maps: AUC: 1.000, specificity: 1.000, sensitivity 1.000) from other α-synucleinopathies. PD-HDR and DLB were comparable for the brain hypo and hypermetabolism patterns, thus not allowing differentiation by SPM t-maps or SSM/PCA. Of note, we found a gradual increase of PDRP and DLBRP expression in the continuum from iRBD to PD-HDR and DLB, where the DLB patients had the highest scores. SSM/PCA could differentiate iRBD from DLB, reflecting specifically the differences in disease staging and severity (AUC: 0.938, specificity: 0.821, sensitivity 0.941). Conclusions: SPM-single subject maps and SSM/PCA are both valid methods in supporting diagnosis within the α-synucleinopathy spectrum, with different strengths and pitfalls. The former reveals dysfunctional brain topographies at the individual level with high accuracy for all the specific subtype patterns, and particularly also the normal maps; the latter provides a reliable quantification, independent from the rater experience, particularly in tracking the disease severity and staging. Thus, our findings suggest that differences in data analysis approaches exist and should be considered in clinical settings. However, combining both methods might offer the best diagnostic performance.
MeSH term(s)	Humans ; Synucleinopathies/diagnostic imaging ; Synucleinopathies/metabolism ; Fluorodeoxyglucose F18/metabolism ; Brain/diagnostic imaging ; Brain/metabolism ; Parkinson Disease/metabolism ; Alzheimer Disease/metabolism ; Multiple System Atrophy/diagnostic imaging ; Positron-Emission Tomography/methods ; Multivariate Analysis ; Lewy Body Disease/diagnostic imaging ; Lewy Body Disease/metabolism
Chemical Substances	Fluorodeoxyglucose F18 (0Z5B2CJX4D)
Language	English
Publishing date	2023-07-13
Publishing country	Netherlands
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	2701571-3
ISSN	2213-1582 ; 2213-1582
ISSN (online)	2213-1582
ISSN	2213-1582
DOI	10.1016/j.nicl.2023.103475
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Article ; Online: The impact of social network change and health decline: a qualitative study on experiences of older adults who are ageing in place.

Ouden, Willeke Vos-den / van Boekel, Leonieke / Janssen, Meriam / Leenders, Roger / Luijkx, Katrien

BMC geriatrics

2021 Volume 21, Issue 1, Page(s) 480

Abstract: Background: Older adults prefer to age in place. Social network change and health decline challenge ageing in place, as stressors that make age-related advantages disappear. The aim of this study was to explore social network change and health decline ... ...

Abstract	Background: Older adults prefer to age in place. Social network change and health decline challenge ageing in place, as stressors that make age-related advantages disappear. The aim of this study was to explore social network change and health decline and its impact on older adults who are ageing in place. Method: In-depth interviews (n = 16) were conducted with older adults who were ageing in place and who were experiencing health decline and social network change. Procedures for grounded theory building were followed to analyse the interviews with respondents who were discharged from the hospital less than 4 months ago (n = 7). Narrative analysis was conducted to reach a deeper understanding of the expected complexity of experiences of this targeted sample. Results: Results encompass a typology with four types of impact: A. Sneak preview of old age, B. Disruptive transition into old age, C. Drastically ageing, and D. Steadily ageing. Additionally, indications were found that older adults should be able to move along the four types of impact and ideally could end up in quartile D, experiencing little or no impact at all (anymore). Conclusion: The results present an optimistic view on the possibilities of older adults to continue ageing in place despite experiencing unavoidable and uncontrollable stressors in life. Also, the results provide leads for practice, to develop an action perspective for home care nurses and gerontological social workers to determine and reduce the impact of social network change and health decline on older adults who are ageing in place. Suggestions for further research would be to unravel how to detect temporal setbacks in successful ageing in place.
MeSH term(s)	Aged ; Aging ; Home Care Services ; Humans ; Independent Living ; Qualitative Research ; Social Networking
Language	English
Publishing date	2021-09-04
Publishing country	England
Document type	Journal Article
ZDB-ID	2059865-8
ISSN	1471-2318 ; 1471-2318
ISSN (online)	1471-2318
ISSN	1471-2318
DOI	10.1186/s12877-021-02385-6
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Article: Feeding Problems in Patients with Noonan Syndrome: A Narrative Review.

Tiemens, Dagmar K / van Haaften, Leenke / Leenders, Erika / van Wegberg, Annemiek M J / Gunther Moor, Bregtje / Geelen, Joyce / Draaisma, Jos M T

Journal of clinical medicine

2022 Volume 11, Issue 3

Abstract: Noonan syndrome (NS) belongs to the group of Noonan syndrome spectrum disorders (NSSD), which is a group of phenotypically related conditions. Feeding problems are often present not only in infancy but also in childhood, and even beyond that period. We ... ...

Abstract	Noonan syndrome (NS) belongs to the group of Noonan syndrome spectrum disorders (NSSD), which is a group of phenotypically related conditions. Feeding problems are often present not only in infancy but also in childhood, and even beyond that period. We describe the different aspects of feeding problems using a (theoretical) concept proposed in 2019. More than 50% of infants with NS develop feeding problems, and up to half of these infants will be tube-dependent for some time. Although, in general, there is a major improvement between the age of 1 and 2 years, with only a minority still having feeding problems after the age of 2 years, as long as the feeding problems continue, the impact on the quality of life of both NS infants and their caregivers may be significant. Feeding problems in general improve faster in children with a pathogenic
Language	English
Publishing date	2022-01-30
Publishing country	Switzerland
Document type	Journal Article ; Review
ZDB-ID	2662592-1
ISSN	2077-0383
ISSN	2077-0383
DOI	10.3390/jcm11030754
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Article ; Online: Hippocampus activation related to 'real-time' processing of visuospatial change.

Beudel, M / Leenders, K L / de Jong, B M

Brain research

2016 Volume 1652, Page(s) 204–211

Abstract: The delay associated with cerebral processing time implies a lack of real-time representation of changes in the observed environment. To bridge this gap for motor actions in a dynamical environment, the brain uses predictions of the most plausible future ...

Abstract	The delay associated with cerebral processing time implies a lack of real-time representation of changes in the observed environment. To bridge this gap for motor actions in a dynamical environment, the brain uses predictions of the most plausible future reality based on previously provided information. To optimise these predictions, adjustments to actual experiences are necessary. This requires a perceptual memory buffer. In our study we gained more insight how the brain treats (real-time) information by comparing cerebral activations related to judging past-, present- and future locations of a moving ball, respectively. Eighteen healthy subjects made these estimations while fMRI data was obtained. All three conditions evoked bilateral dorsal-parietal and premotor activations, while judgment of the location of the ball at the moment of judgment showed increased bilateral posterior hippocampus activation relative to making both future and past judgments at the one-second time-sale. Since the condition of such 'real-time' judgments implied undistracted observation of the ball's actual movements, the associated hippocampal activation is consistent with the concept that the hippocampus participates in a top-down exerted sensory gating mechanism. In this way, it may play a role in novelty (saliency) detection.
MeSH term(s)	Adult ; Brain Mapping ; Female ; Hippocampus/physiology ; Humans ; Imagination/physiology ; Judgment/physiology ; Magnetic Resonance Imaging ; Male ; Motion Perception/physiology ; Neuropsychological Tests ; Photic Stimulation ; Reaction Time ; Space Perception/physiology ; Spatial Memory/physiology
Language	English
Publishing date	2016-12-01
Publishing country	Netherlands
Document type	Journal Article
ZDB-ID	1200-2
ISSN	1872-6240 ; 0006-8993
ISSN (online)	1872-6240
ISSN	0006-8993
DOI	10.1016/j.brainres.2016.10.010
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Article ; Online: The most important problems and needs of rasopathy patients with a noonan syndrome spectrum disorder.

Tiemens, Dagmar K / Kleimeier, Lotte / Leenders, Erika / Wingbermühle, Ellen / Roelofs, Renee L / Sibbles, Barbara / Oostwegel, Floor S M / Vroonland, Eva / van Leeuwen, Conny / Niessen, Hanneke / Sonnega, Paul / Duursma, Anniek / Willemsen, Michel A A P / Draaisma, Jos M T / Pittens, Carina A C M

Orphanet journal of rare diseases

2023 Volume 18, Issue 1, Page(s) 198

Abstract: Background: Noonan syndrome spectrum disorders (NSSDs) constitute a group within the Rasopathies, and are one of the largest groups of syndromes with impact on multi-organ involvement known. The extreme variability of the clinical phenotype is, among ... ...

Abstract	Background: Noonan syndrome spectrum disorders (NSSDs) constitute a group within the Rasopathies, and are one of the largest groups of syndromes with impact on multi-organ involvement known. The extreme variability of the clinical phenotype is, among others, due to the numerous different genes that are involved, and the differences in clinical presentation over the life span. We have studied the needs of patients and their relatives aiming to develop, evaluate and choose focus in research, medical care and policy to better meet their perspectives. Methods: Using the participatory and interactive Dialogue method, 80 patients and relatives mentioned 53 different problems or needs (topics) that were categorized into eight themes. These themes and the topics within each theme, were subsequently prioritized by putting them in order of importance methodologically. Results: The four highest prioritized themes were: (1) Physical problems (non-musculoskeletal related); (2) Social, emotional and behavioral problems; (3) Cognitive functioning and information processing; and (4) Problems related to the musculoskeletal system. Nineteen out of the 53 topics were physical problems. According to the total group of respondents, the top 3 prioritized topics within theme 1 were coagulation problems, heart problems, and feeding problems. Also data stratified by age groups, phenotype (NS and other NSSDs) and gender showed some remarkable results. For instance, feeding problems were prioritized as the most important topic of the highest prioritized theme, according to patients aged 0-12 years. Also feeding problems show a significant difference in its prioritization according to female patients (2) compared to male patients (7). On the other hand, heart problems were not mentioned in the top three prioritized topics in the youngest age groups, although heart problems are generally considered most important for patients with NSSD. Conclusions: With our results we underline the importance of methodologically inventorying the needs of NSSD patients, not only at the group level, but to also focus on specific needs according to e.g. age, phenotype and gender. For instance, it is remarkable that both the current Clinical Guidelines and the Noonan Syndrome diagnostic criteria give little to no attention to feeding problems, though our results indicate that, to the youngest patients, these problems have top priority. A similar situation appears to apply to the clinical management of e.g. coagulation, neuropsychological and musculoskeletal problems (like physiotherapy or occupational therapy) and to a need for (educational) tools to support patients at school or at work. Our study may help to shape targeted (clinical) management, research and policy inside and outside medical (research) institutes and shed light on the complex phenotypes of NSSDs, the families' and patients' perspectives on the everyday consequences of the many different problems, as well as their needs.
MeSH term(s)	Humans ; Male ; Female ; Noonan Syndrome/genetics ; Noonan Syndrome/diagnosis ; Cognition ; Phenotype
Language	English
Publishing date	2023-07-21
Publishing country	England
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	2225857-7
ISSN	1750-1172 ; 1750-1172
ISSN (online)	1750-1172
ISSN	1750-1172
DOI	10.1186/s13023-023-02818-y
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