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15. A Case of a Male Patient with Frontotemporal Dementia with Behavioral and Psychological Symptoms Responding to a Low Dose of Escitalopram

Author

Atsuko Ikenouchi, Okamoto N, Murakami S, Takaoka K, Reiji Yoshimura, and Issei Seki

Subjects
Pediatrics, medicine.medical_specialty, Text mining, business.industry, Male patient, mental disorders, Low dose, medicine, Escitalopram, medicine.disease, business, Frontotemporal dementia, medicine.drug
Abstract

Background: Frontotemporal dementia (FTD) is a common type of early-onset dementia. It is characterized by insidious onset and gradual progression, early decline in social interpersonal conduct and regulation of personal conduct, early emotional blunting and loss of insight. Behavioral disorder, speech and language are supportive features in FTD. Here, we present a case of a patient with FTD with behavioral and psychological symptoms of dementia (BPSD) responding to escitalopram treatment.Case presentation: A 60-year-old Japanese man had been treated for early-onset Alzheimer-type dementia with galantamine (24 mg/day) for 5 years. He developed behavioral, executive and semantic deficits. He had stereotypical, compulsive behavior, eating habit changes, disinhibition, excitement, irritability and anxiety. His diagnosis changed to FTD based on clinical features and MRI and SPECT. Galantamine was tapered off, and escitalopram 5 mg/day was started and increased to 10 mg/day. The symptoms of FTD, including behavioral and psychological symptoms, were decreased.Conclusion: This case is the first report demonstrating that escitalopram might be effective for BPSD that accompany FTD.

Published
2020

16. CFAP45 deficiency causes situs abnormalities and asthenospermia by disrupting an axonemal adenine nucleotide homeostasis module

Author

Dougherty, G.W., Mizuno, K., Nöthe-Menchen, T., Ikawa, Y., Boldt, K., Ta-Shma, A., Aprea, I., Minegishi, K., Pang, Y.P., Pennekamp, P., Loges, N.T., Raidt, J., Hjeij, R., Wallmeier, J., Mussaffi, H., Perles, Z., Elpeleg, O., Rabert, F., Shiratori, H., Letteboer, S.J.F., Horn, N., Young, S., Strünker, T., Stumme, F., Werner, C., Olbrich, H., Takaoka, K., Ide, T., Twan, W.K., Biebach, L., Große-Onnebrink, J., Klinkenbusch, J.A., Praveen, K., Bracht, D.C., Höben, I.M., Junger, K., Gützlaff, J., Cindrić, S., Aviram, M., Kaiser, T., Memari, Y., Dzeja, P.P., Dworniczak, B., Ueffing, M., Roepman, R., Bartscherer, K., Katsanis, N., Davis, E.E., Amirav, I., Hamada, H., Omran, H., Dougherty, G.W., Mizuno, K., Nöthe-Menchen, T., Ikawa, Y., Boldt, K., Ta-Shma, A., Aprea, I., Minegishi, K., Pang, Y.P., Pennekamp, P., Loges, N.T., Raidt, J., Hjeij, R., Wallmeier, J., Mussaffi, H., Perles, Z., Elpeleg, O., Rabert, F., Shiratori, H., Letteboer, S.J.F., Horn, N., Young, S., Strünker, T., Stumme, F., Werner, C., Olbrich, H., Takaoka, K., Ide, T., Twan, W.K., Biebach, L., Große-Onnebrink, J., Klinkenbusch, J.A., Praveen, K., Bracht, D.C., Höben, I.M., Junger, K., Gützlaff, J., Cindrić, S., Aviram, M., Kaiser, T., Memari, Y., Dzeja, P.P., Dworniczak, B., Ueffing, M., Roepman, R., Bartscherer, K., Katsanis, N., Davis, E.E., Amirav, I., Hamada, H., and Omran, H.

Abstract

Contains fulltext : 229376.pdf (publisher's version ) (Open Access), Axonemal dynein ATPases direct ciliary and flagellar beating via adenosine triphosphate (ATP) hydrolysis. The modulatory effect of adenosine monophosphate (AMP) and adenosine diphosphate (ADP) on flagellar beating is not fully understood. Here, we describe a deficiency of cilia and flagella associated protein 45 (CFAP45) in humans and mice that presents a motile ciliopathy featuring situs inversus totalis and asthenospermia. CFAP45-deficient cilia and flagella show normal morphology and axonemal ultrastructure. Proteomic profiling links CFAP45 to an axonemal module including dynein ATPases and adenylate kinase as well as CFAP52, whose mutations cause a similar ciliopathy. CFAP45 binds AMP in vitro, consistent with structural modelling that identifies an AMP-binding interface between CFAP45 and AK8. Microtubule sliding of dyskinetic sperm from Cfap45(-/-) mice is rescued with the addition of either AMP or ADP with ATP, compared to ATP alone. We propose that CFAP45 supports mammalian ciliary and flagellar beating via an adenine nucleotide homeostasis module.

Published
2020

45. Time to onset of bisphosphonate-related osteonecrosis of the jaws : a multicentre retrospective cohort study

Author

Fung, P. P. L., Bedogni, G., Bedogni, A., Petrie, A., Porter, S., Campisi, G., Bagan, J., Fusco, V., Saia, G., Acham, S., Musto, P., Petrucci, M. T., Diz, P., Colella, G., Mignogna, M. D., Pentenero, M., Arduino, P., Lodi, G., Maiorana, C., Manfredi, M., Hallberg, Pär, Wadelius, Mia, Takaoka, K., Leung, Y. Y., Bonacina, R., Schiodt, M., Lakatos, P., Taylor, T., De Riu, G., Favini, G., Rogers, S. N., Pirmohamed, M., Nicoletti, P., Fedele, S., Fung, P. P. L., Bedogni, G., Bedogni, A., Petrie, A., Porter, S., Campisi, G., Bagan, J., Fusco, V., Saia, G., Acham, S., Musto, P., Petrucci, M. T., Diz, P., Colella, G., Mignogna, M. D., Pentenero, M., Arduino, P., Lodi, G., Maiorana, C., Manfredi, M., Hallberg, Pär, Wadelius, Mia, Takaoka, K., Leung, Y. Y., Bonacina, R., Schiodt, M., Lakatos, P., Taylor, T., De Riu, G., Favini, G., Rogers, S. N., Pirmohamed, M., Nicoletti, P., and Fedele, S.

Abstract

Objectives: Osteonecrosis of the jaw (ONJ) is a potentially severe adverse effect of bisphosphonates (BP). Although the risk of ONJ increases with increasing duration of BP treatment, there are currently no reliable estimates of the ONJ time to onset (TTO). The objective of this study was to estimate the TTO and associated risk factors in BP-treated patients. Subjects and Methods: Retrospective analysis of data from 22 secondary care centres in seven countries relevant to 349 patients who developed BP-related ONJ between 2004 and 2012. Results: The median (95%CI) TTO was 6.0 years in patients treated with alendronate (n=88) and 2.2years in those treated with zoledronate (n=218). Multivariable Cox regression showed that dentoalveolar surgery was inversely associated, and the use of antiangiogenics directly associated, with the TTO in patients with cancer treated with zoledronate. Conclusions: The incidence of ONJ increases with the duration of BP therapy, with notable differences observed with respect to BP type and potency, route of administration and underlying disease. When data are stratified by BP type, a time of 6.0 and 2.2years of oral alendronate and intravenous zoledronate therapy, respectively, is required for 50% of patients to develop ONJ. After stratification by disease, a time of 5.3 and 2.2years of BP therapy is required for 50% of patients with osteoporosis and cancer, respectively, to develop ONJ. These findings have significant implications for the design of future clinical studies and the development of risk-reduction strategies aimed at either assessing or modulating the risk of ONJ associated with BP.

Published
2017
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