The truth about being a scientist, an investigator trying to uncover or discover something, is that if you see something, other people probably see it, too. So, you should attempt to conduct studies and get them published as soon as you can if you have an original idea. The following is one of my research journeys, which clearly demonstrates this.
I have always had divergent thinking, thinking outside the box. For instance, I had a theory that ADHD could predispose to narcolepsy. I wrote about this in my senior/undergraduate thesis at Harvard in 1997, which was purely theoretical. It was titled “Attention-Deficit Disorder and Narcolepsy: Different Stages of the Same Disorder?”
Next, I wrote a master's thesis at PENN about this concept, which included case studies and a survey study (using a validated and reliable measure for childhood history of ADHD) collected from the now-closed National Narcolepsy Registry (NNR). This thesis was titled: “Attention-Deficit Disorder and Narcolepsy/Hypersomnia: Different Stages of the Same Disorder?” Distinguished Paper.
I published a book chapter about this concept with a psychologist who was faculty at Yale at the time, which included much of the original text from my undergraduate thesis (Brown & Modestino, 2000). This was then updated and published as a new chapter in another book, with some of the original text from undergraduate and master's theses (Brown, Owens, & Modestino, 2009).
Based on the survey data (using a validated and reliable measure for childhood ADHD) from my master's thesis and other data within the (NNR) which also included objective physiological measures, and subsequently collecting the same survey data from a different cohort, a control group without ADHD, for comparison, I published this as a study (Modestino & Winchester, 2013).
I experienced multiple neurologists, sleep specialists, and psychiatrists refusing to believe that it was possible that there was a connection between the childhood-onset psychiatric disorder ADHD and the neurological/sleep disorder narcolepsy. I was putting together a book for a publisher (for which I did not follow through because of time constraints in my postdoc and lack of follow-through with potential authors), recruiting researchers based on their research to write chapters related to the comorbidity of sleep disorders and ADHD. Two research groups told me that this relationship was not possible and that their research showed something else, such as shared symptoms, despite the fact that there was no overlap between the diagnostic criteria or core symptoms of these two pathologies. Additionally, their research did provide evidence in support of my theory. One of these researchers, whom I shall not name, vehemently denied any such connection and was hostile in many of our communications. Within less than a year, this same researcher published a study about this connection in support of my already published theory. It was clear this person was in the process of writing up this study during communications with me. This person clearly lied for reasons of which I can only speculate.
At my second postdoc, in the dept. of Neurology at BU, I was invited to give a talk about my research on this theory. I had sleep experts insist I was mistaken for thinking there could be an authentic relationship between ADHD and narcolepsy, that it was random chance and coincidence. I explained the joint prevalence calculation that I had done on my survey participants, which divulged that this was 8-15 times greater than if it was purely a random chance that an individual might have ADHD and comorbid narcolepsy (Modestino & Winchester, 2013). Here is a link to the slides for that talk, which included my first published study about this and future planned research to explore this.
Medicine Lecture Series, Sleep Disorders Center, Department of Neurology, Boston University School of Medicine, Boston Medical Center, Boston, MA Wednesday, January 15, 2014
Many of the narcolepsy information and support groups/forums online had countless people claiming to have both diagnoses officially. I continued with my theory and proposed another study to include testing genetics in a publication with people from the previous book project and even a member of the Sleep Disorders Center from BU who invited me to give the talk. I published this proposed study; please see Modestino et al., 2015. In that publication, I referenced a large-scale epidemiological study with narcoleptics showing the comorbidity with ADHD to be significant (Ohayon, 2013). In fact, it was five times greater than chance for the two disorders to co-occur randomly, based on a joint-prevalence calculation I was able to perform from the study's statistics.
I worked over the last decade attempting to secure funding to do this study, which included screening for shared genes, as both ADHD and narcolepsy are not single-gene disorders. They are polygenic. As I was no longer working at a medical school, a large university, or a hospital, I worked with colleagues attempting to find a way to do this study. Time after time, things fell through, and it was put on the back burner as working and earning a living was more important.
Guess what? Someone else has published a study showing shared genetic variants common to ADHD and narcolepsy. Both narcolepsy and ADHD are polygenic, and the multiple genes that contribute to one also contribute to the other (Takahashi et al., 2020). I was right all along, despite the fact several people told me I was crazy and illogical! And the fact that I did not have the funding or opportunity to conduct such a study yet meant someone else did and published it. What makes this even more disappointing to me than not being the first one to publish a genetic study about this was that neither my published study about this nor my publication about my proposed future research were referenced in this study despite my publications being indexed on PubMed.
Currently, I am working with Drs. Blum and Thanos. We are planning to submit a research grant for a genetic study, which will be far different but, in essence, a conceptual replication of the genetic study by Takahashi et al. (2020). I cannot predict what will happen with securing funding. Again, in the world of science, it is important to get your original ideas published and, hopefully, secure funding to conduct studies before someone else comes up with the same hypothesis, theory, concept, or idea and conducts a study before you do. As this was my hypothesis for decades, it is highly likely that I had this idea before the Takahashi group. So, it is a bit disappointing to me they did it first. However, the fact that their study was clear, objective evidence in support of my original hypothesis from decades ago, does make me feel vindicated in that my idea was neither merely anecdotal nor pure coincidence.
References
Brown, T.E. & Modestino, E.J. (2000) Ch.10: Attention-Deficit Disorders with Sleep/Arousal Disturbances. In: Attention-Deficit Disorders and Comorbidities in Children, Adolescents, and Adults. Edited by Brown, T. E., American Psychiatric Press, Inc., Washington, DC.: 341-362.
Brown, T.E., Owens, J.A., & Modestino, E.J. (2009) Ch.15: ADHD with Sleep/Arousal Disturbances. In: ADHD Comorbidities: Handbook for ADHD Complications in Children and Adults. Edited by Brown, T. E., American Psychiatric Press, Inc., Washington, DC.: 279-291.
Modestino, E.J. (unpublished undergraduate thesis 1997) Attention-Deficit Disorder and Narcolepsy: Different Stages of the Same Disorder? Harvard University
Modestino, E.J. (unpublished master's thesis 1999) Attention-Deficit Disorder and Narcolepsy/Hypersomnia: Different Stages of the Same Disorder? Distinguished Paper. University of Pennsylvania
Modestino, E. J., Blum, K., Oscar-Berman, M., Gold, M. S., Duane, D. D., Sultan, S. G. S., & Auerbach, S. H. (2015). Reward deficiency syndrome: Attentional/arousal subtypes, limitations of current diagnostic nosology, and future research. Journal of Reward Deficiency Syndrome, 01(01). https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4545661/pdf/nihms-692690.pdf
Modestino, E.J., & Winchester, J. (2013). A retrospective survey of childhood ADHD symptomatology among Adult Narcoleptics. Journal of Attention Disorders, 17(7), 574-582. https://doi.org/10.1177/1087054713480033
Ohayon, M. M. (2013). Narcolepsy is complicated by high medical and psychiatric comorbidities: A comparison with the general population. Sleep Medicine, 14(6), 488–492. https://doi.org/10.1016/j.sleep.2013.03.002
Takahashi, N., Nishimura, T., Harada, T., Okumura, A., Choi, D., Iwabuchi, T., Kuwabara, H., Takagai, S., Nomura, Y., Newcorn, Jeffrey. H., Takei, N., & Tsuchiya, K. J. (2020). Polygenic risk score analysis revealed shared genetic background in attention deficit hyperactivity disorder and narcolepsy. Translational Psychiatry, 10(1). https://doi.org/10.1038/s41398-020-00971-7
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