35 years of research in Amsterdam: a revolution in the field of leukodystrophies
Prof. dr. Marjo van der Knaap and her team: Dr. Marianna Bugiani, Dr. Marc Engelen, Dr. Niek van Til and Prof. dr. Nicole Wolf
Together with her team, Marjo van der Knaap will provide a presentation on 35 years of research: a revolution in the field of leukodystrophies. She will speak about in-depth phenotyping and genotyping for a revolution in diagnoses. Then her colleague Marianna Bugiani will continue the presentation, providing her insight into leukodystrophy disease mechanisms. Marc Engelen will speak about revolutions in therapy, followed by Niek van Til, who will speak about including gene therapy. Nicole Wolf, together with a patient and their family, will provide a valuable family perspective on the revolution in this field.
Marjo van der Knaap started in the nineteen eighties with the leukodystrophy research, at a time that most patients remained without a specific diagnosis and very little could be done for patients. Together with prof. Jaap Valk, neuroradiologist at the VUmc, she developed an MRI-based pattern recognition system for in-depth phenotyping and classification of patients. This system turned out to be extremely successful in defining new disease entities. Subsequently, the genetic defects of numerous novel disorders were found in Amsterdam. Currently, almost all leukodystrophy patients receive a DNA-based diagnosis.
Marianna Bugiani, a former child neurologist, turned to neuropathology about 20 years ago to accelerate research towards elucidating disease mechanisms based on the pathological findings in the patients’ brain. Approaching leukodystrophies from a cellular functional pathology perspective turned out to be successful to this aim. Recently, she developed a method to culture post-mortem patient brain tissue in 3D to complement the other ALC preclinical data and further investigate diseases at their onset in the human brain.
Marc van Engelen worked on natural history studies and on developing surrogate clinical outcome measures for leukodystrophies for the past decade. Now that many leukodystrophies are “clinical trial ready”, the last couple of years have seen an increase in clinical trials being initiated, including several gene therapy trials. The field of leukodystrophies is moving from the diagnostic revolution into a treatment revolution.
Niek van Til focusses on preclinical efficacy and safety assessment of gene therapies for leukodystrophies. Gene therapy technologies are transforming the landscape of available treatment options, such as gene augmentation using viral delivery systems and more recently developed groundbreaking gene editing approaches. Leukodystrophies are diverse and tailor-made therapies are required for successful treatment.
Nicole Wolf works on treatment mechanisms and outcomes for leukodystrophies, including accessibility of novel treatments and expertise within Europe. Gene therapy for metachromatic leukodystrophy, one of the most frequent leukodystrophies, is now registered treatment and reimbursed in the Netherlands. What this means, is illustrated with a family as example.
Marjo van der Knaap
Young Amsterdam Neuroscientists take the stage
Boosting endogenous dopamine production: a novel therapeutic approach for Parkinson's disease?
Dr. Lars van der Heide
Parkinson’s disease is characterized by a spectrum of motor symptoms primarily stemming from striatal dopamine deficiency, a consequence of progressive degeneration of midbrain dopamine neurons of the nigrostriatal pathway. The cornerstone of managing these symptoms is dopamine replacement therapy with exogenous levodopa (L-DOPA), which has been the gold standard for over half a century. Unfortunately, L-DOPA therapy is burdened by enduring and inescapable challenges, primarily the lack of long-term efficacy and the development of L-DOPA-induced dyskinesias. Consequently, there is an urgent demand for alternatives to L-DOPA based therapies.
With this in mind, we focus on tyrosine hydroxylase (TH), the rate-limiting enzyme in the endogenous dopamine biosynthesis pathway, as a promising therapeutic target. Key to activating TH as a therapeutic approach is utilizing unique and highly specific targets expressed in the substantia nigra midbrain dopamine neurons, thus activating TH where it is needed, eliminating off-target effects.
Lars van der Heide
Identifying therapeutic mechanisms of deep-brain stimulation in a mouse model for OCD
Prof. dr. Ingo Willuhn
Deep-brain stimulation (DBS) effectively treats patients suffering from otherwise therapy-resistant obsessive-compulsive disorder, presumably via the modulation of cortico-striatal circuits. To gain mechanistic insight, we monitored neuronal activity in cortico-striatal regions in a mouse model for compulsive behavior, while systematically varying clinically relevant DBS parameters. Increasing DBS intensity dose-dependently decreased compulsive behavior, as well as recruited an increasing, yet balanced, number of excited and inhibited neurons throughout cortico-striatal regions. Such neuronal recruitment did not alter basic brain function such as resting-state activity. In addition to these widespread effects, we observed specific involvement of the medial orbitofrontal cortex in therapeutic outcomes, which was corroborated by optogenetic stimulation. Together, our findings provide mechanistic insight into how DBS exerts its therapeutic effects on compulsive behaviors.
Ingo Willuhn
Keynote Lecture 'Harnessing my daughter's diagnosis to drive novel treatments for neurodevelopmental disorders'
Dr. Madeleine Oudin
In 2021, Madeleine's daughter Margot was diagnosed with two de novo mutations in the SCN8A gene, causing her to develop seizures at 3 months old and infantile spams at 5 months old. Realizing the limited information available on the effects of these mutations and the lack of effective treatment options, Madeleine decided to start doing research on SCN8A in my own lab, and work with clinicians, epilepsy researchers and biotech to increase our understanding of SCN8A and develop a novel splice-switching ASO. She will discuss my journey as a scientist and mother learning about epilepsy and collaborating to develop a new treatment.