June 2022
Volume 63, Issue 7
Open Access
ARVO Annual Meeting Abstract  |   June 2022
The Incidence and Characteristics of Charles Bonnet Syndrome in a Large Cohort of Leber’s Hereditary Optic Neuropathy Patients
Author Affiliations & Notes
  • Hong-An Nguyen
    Ophthalmology, University of Ottawa Faculty of Medicine, Ottawa, Ontario, Canada
    Ottawa Hospital Research Institute, Ottawa, Ontario, Canada
  • Mohammad Kreimei
    Ophthalmology, University of Ottawa Faculty of Medicine, Ottawa, Ontario, Canada
  • Eisi Mollanji
    Ophthalmology, University of Ottawa Faculty of Medicine, Ottawa, Ontario, Canada
  • Lissa Poincenot
    LHON.org, California, United States
  • Rustum Karanjia
    Ophthalmology, University of Ottawa Faculty of Medicine, Ottawa, Ontario, Canada
    Dohney Eye Institute, California, United States
  • Footnotes
    Commercial Relationships   Hong-An Nguyen None; Mohammad Kreimei None; Eisi Mollanji None; Lissa Poincenot None; Rustum Karanjia None
  • Footnotes
    Support  International Foundation for Optic Nerve Disease (IFOND), LHON.org, LHON Canada
Investigative Ophthalmology & Visual Science June 2022, Vol.63, 432. doi:
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      Hong-An Nguyen, Mohammad Kreimei, Eisi Mollanji, Lissa Poincenot, Rustum Karanjia; The Incidence and Characteristics of Charles Bonnet Syndrome in a Large Cohort of Leber’s Hereditary Optic Neuropathy Patients. Invest. Ophthalmol. Vis. Sci. 2022;63(7):432.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Purpose : Charles Bonnet Syndrome (CBS) is a condition involving visual hallucinations as a result of vision loss, but it is not well characterized nor routinely assessed for. Some patients with Leber’s Hereditary Optic Neuropathy (LHON) experience CBS, but there is limited published data. This is the first study screening for and characterizing CBS in a large cohort of the LHON patient population.

Methods : A recently validated French-Canadian CBS screening questionnaire was adapted to an online bilingual (English-French) format, tailored to LHON patients (Cantin et al. CJO, 2019;54:323–7). The 59-item questionnaire was distributed to numerous mailing lists, online communities and social media platforms consisting of LHON patients. Quantitative and qualitative response data was used for cross-sectional analysis.

Results : A total of 155 affected LHON patients, 59% male and 41% female with over 85% of participants between the ages of 18 to 65, completed the questionnaire – with 54% screening positive for CBS. Of the participants who screened positive for CBS, 55% were affected by the 11778G>A mutation, 13.3% by 14484T>C, 14.5% by 3460G>A, 8.4% by other mutations and 8.4% by an unknown mutation. In addition, 41% of these participants have been diagnosed with LHON for more than 10 years while 38.5% have been diagnosed for less than 5 years.

Among CBS-positive patients, 90% of patients reported experiencing the hallucinations within the past year. Furthermore, 39% of patients reported that the images disturb their sleep, while 53% reported that the images negatively affect their mood. Additionally, only 55% had heard of CBS before and only 12% (10 participants) had been diagnosed with CBS by a health-care professional.

Conclusions : Our results yielded a high proportion of LHON patients screening positive for CBS (54%), indicating the condition may be a lot more common than previously suspected, and only a minority of LHON patients are being adequately assessed and managed for the condition. There is also an indication that their CBS tends to be long-lasting and can be associated with various negative health outcomes. Overall, this study provides a better understanding of the prevalence of CBS in LHON and the need for proactive discussions by health care providers.

This abstract was presented at the 2022 ARVO Annual Meeting, held in Denver, CO, May 1-4, 2022, and virtually.

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