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Prazosin use in a patient with rare Neurobeachin gene deletion shows improvement in paranoid behavior: a case report

BACKGROUND: Disruption of the Neurobeachin gene is a rare genetic mutation that has been implicated in the development of autism and enhanced long-term potentiation of the hippocampal CA1 region, causing a heightened conditioned fear response and impaired fear extinction. Prazosin, an alpha-1 recept...

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Autores principales: Cantwell, Christina Y., Fortman, Jamie, Seegan, Alexis
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8705137/
https://www.ncbi.nlm.nih.gov/pubmed/34949210
http://dx.doi.org/10.1186/s13256-021-03209-2
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author Cantwell, Christina Y.
Fortman, Jamie
Seegan, Alexis
author_facet Cantwell, Christina Y.
Fortman, Jamie
Seegan, Alexis
author_sort Cantwell, Christina Y.
collection PubMed
description BACKGROUND: Disruption of the Neurobeachin gene is a rare genetic mutation that has been implicated in the development of autism and enhanced long-term potentiation of the hippocampal CA1 region, causing a heightened conditioned fear response and impaired fear extinction. Prazosin, an alpha-1 receptor antagonist, has been used in patients with posttraumatic stress disorder to mitigate the increased alpha-1 activity involved in fear and startle responses. Here we report a case of a patient with a rare Neurobeachin gene deletion, who demonstrated marked and sustained improvement in paranoid behavior within days of prazosin initiation. CASE PRESENTATION: The patient is a 27-year-old White male with autism spectrum disorder, obsessive–compulsive disorder, and schizophrenia, with a chromosome 13q12 deletion including deletion of the Neurobeachin gene, who presented to the emergency department due to worsening functional status and profound weight loss as a result of only eating prepackaged foods. He had not showered or changed clothes in several months prior to presentation. He was hospitalized in the inpatient psychiatric unit for 2 months before prazosin was initiated. During that time, he demonstrated paranoia as evidenced by heightened sensitivity to doors opening, guarded interactions, and limited communication with providers and other patients. He also exhibited poor grooming habits, with aversion to showering, shaving, and changing clothes. Since initiating prazosin, he has demonstrated a brighter affect, initiates and maintains conversations, showers and changes clothes on a regular basis, and eats a variety of foods. At the time of this report, the patient was discharged to live in an apartment with a caregiver after a 7-month inpatient hospitalization. CONCLUSIONS: Low-dose prazosin shows rapid and sustained improvement in paranoid behavior in a patient with a rare Neurobeachin gene deletion. Prazosin has a relatively favorable side effect profile with once-daily dosing and low cost. Prazosin may provide clinical improvement in patients with Neurobeachin gene deletions due to its theoretical attenuation in fear response through alpha-1 antagonism.
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spelling pubmed-87051372022-01-05 Prazosin use in a patient with rare Neurobeachin gene deletion shows improvement in paranoid behavior: a case report Cantwell, Christina Y. Fortman, Jamie Seegan, Alexis J Med Case Rep Case Report BACKGROUND: Disruption of the Neurobeachin gene is a rare genetic mutation that has been implicated in the development of autism and enhanced long-term potentiation of the hippocampal CA1 region, causing a heightened conditioned fear response and impaired fear extinction. Prazosin, an alpha-1 receptor antagonist, has been used in patients with posttraumatic stress disorder to mitigate the increased alpha-1 activity involved in fear and startle responses. Here we report a case of a patient with a rare Neurobeachin gene deletion, who demonstrated marked and sustained improvement in paranoid behavior within days of prazosin initiation. CASE PRESENTATION: The patient is a 27-year-old White male with autism spectrum disorder, obsessive–compulsive disorder, and schizophrenia, with a chromosome 13q12 deletion including deletion of the Neurobeachin gene, who presented to the emergency department due to worsening functional status and profound weight loss as a result of only eating prepackaged foods. He had not showered or changed clothes in several months prior to presentation. He was hospitalized in the inpatient psychiatric unit for 2 months before prazosin was initiated. During that time, he demonstrated paranoia as evidenced by heightened sensitivity to doors opening, guarded interactions, and limited communication with providers and other patients. He also exhibited poor grooming habits, with aversion to showering, shaving, and changing clothes. Since initiating prazosin, he has demonstrated a brighter affect, initiates and maintains conversations, showers and changes clothes on a regular basis, and eats a variety of foods. At the time of this report, the patient was discharged to live in an apartment with a caregiver after a 7-month inpatient hospitalization. CONCLUSIONS: Low-dose prazosin shows rapid and sustained improvement in paranoid behavior in a patient with a rare Neurobeachin gene deletion. Prazosin has a relatively favorable side effect profile with once-daily dosing and low cost. Prazosin may provide clinical improvement in patients with Neurobeachin gene deletions due to its theoretical attenuation in fear response through alpha-1 antagonism. BioMed Central 2021-12-24 /pmc/articles/PMC8705137/ /pubmed/34949210 http://dx.doi.org/10.1186/s13256-021-03209-2 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Cantwell, Christina Y.
Fortman, Jamie
Seegan, Alexis
Prazosin use in a patient with rare Neurobeachin gene deletion shows improvement in paranoid behavior: a case report
title Prazosin use in a patient with rare Neurobeachin gene deletion shows improvement in paranoid behavior: a case report
title_full Prazosin use in a patient with rare Neurobeachin gene deletion shows improvement in paranoid behavior: a case report
title_fullStr Prazosin use in a patient with rare Neurobeachin gene deletion shows improvement in paranoid behavior: a case report
title_full_unstemmed Prazosin use in a patient with rare Neurobeachin gene deletion shows improvement in paranoid behavior: a case report
title_short Prazosin use in a patient with rare Neurobeachin gene deletion shows improvement in paranoid behavior: a case report
title_sort prazosin use in a patient with rare neurobeachin gene deletion shows improvement in paranoid behavior: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8705137/
https://www.ncbi.nlm.nih.gov/pubmed/34949210
http://dx.doi.org/10.1186/s13256-021-03209-2
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