Long-Term Follow-Up on Bilateral Posterior Hypothalamic Deep Brain Stimulation for Treating Refractory Aggressive Behavior in a Patient with Cri du Chat Syndrome: Analysis of Clinical Data, Intraoperative Microdialysis, and Imaging Connectomics

Adriana Lucia López Ríos; Jürgen Germann; William D Hutchison; Luis Fernando Botero Posada; Luisa Fernanda Ahunca Velasquez; Francisco Aureliano Garcia Jimenez; José Miguel Gloria Escobar; Raquel Chacon Ruiz Martinez; Clement Hamani; Ivo Lebrun; Aline V V Auada; Carlos Anibal Restrepo Bravo; Flavia Venetucci Gouveia

doi:10.1159/000526871

Long-Term Follow-Up on Bilateral Posterior Hypothalamic Deep Brain Stimulation for Treating Refractory Aggressive Behavior in a Patient with Cri du Chat Syndrome: Analysis of Clinical Data, Intraoperative Microdialysis, and Imaging Connectomics

Stereotact Funct Neurosurg. 2022;100(5-6):275-281. doi: 10.1159/000526871. Epub 2022 Nov 29.

Authors

Adriana Lucia López Ríos^{1

2}, Jürgen Germann³, William D Hutchison^{2

4

5}, Luis Fernando Botero Posada^{6

7}, Luisa Fernanda Ahunca Velasquez⁸, Francisco Aureliano Garcia Jimenez⁹, José Miguel Gloria Escobar^{10

11}, Raquel Chacon Ruiz Martinez^{12

13}, Clement Hamani⁴, Ivo Lebrun¹⁴, Aline V V Auada¹⁴, Carlos Anibal Restrepo Bravo^{10

11}, Flavia Venetucci Gouveia¹⁵

Affiliations

¹ Department of Functional and Stereotactic Neurosurgery, University Hospital San Vicente Fundación, Medellín, Colombia.
² Department of Functional and Stereotactic Neurosurgery, San Vicente Fundación, Rionegro, Colombia.
³ Division of Neurosurgery, Department of Surgery, University Health Network and University of Toronto, Toronto, Ontario, Canada.
⁴ Division of Neurosurgery, Department of Surgery, University of Toronto, Toronto, Ontario, Canada.
⁵ Department of Physiology, University of Toronto, Toronto, Ontario, Canada.
⁶ Department of Neuroanesthesiology, San Vicente Fundación, Rionegro, Colombia.
⁷ Department of Physiology, CES University, Medellín, Colombia.
⁸ Department of Neuropsychiatry, San Vicente Fundación, Rionegro, Colombia.
⁹ Department of Neurology, University Hospital San Vicente Fundacion, Medellin, Colombia.
¹⁰ Department of Basic Sciences, Medical School, CES University, Medellín, Colombia.
¹¹ Department of Basic Sciences, San Vicente Fundación, Rionegro, Colombia.
¹² Laboratory of Neuroscience, Hospital Sirio Libanes, São Paulo, Brazil.
¹³ LIM 23, Institute of Psychiatry, School of Medicine, University of São Paulo, São Paulo, Brazil.
¹⁴ Biochemistry and Biophysics Laboratory, Butantan Institute, São Paulo, Brazil.
¹⁵ Neuroscience and Mental Health, Hospital for Sick Children Research Institute, Toronto, Ontario, Canada.

PMID: 36446334
DOI: 10.1159/000526871

Abstract

Posterior hypothalamic-deep brain stimulation (pHyp-DBS) has been reported as a successful treatment for reducing refractory aggressive behaviors in patients with distinct primary diagnoses. Here, we report on a patient with cri du chat syndrome presenting severe self-injury and aggressive behaviors toward others, who was treated with pHyp-DBS. Positive results were observed at long-term follow-up in aggressive behavior and quality of life. Intraoperative microdialysis and imaging connectomics analysis were performed to investigate possible mechanisms of action. Our results suggest the involvement of limbic and motor areas and alterations in main neurotransmitter levels in the targeted area that are associated with positive results following treatment.

Keywords: Cri du chat syndrome; Deep brain stimulation; Imaging connectomics; Intraoperative microdialysis; Posterior hypothalamus.

Publication types

Case Reports
Research Support, Non-U.S. Gov't

MeSH terms

Connectome*
Cri-du-Chat Syndrome* / complications
Deep Brain Stimulation* / methods
Follow-Up Studies
Humans
Microdialysis
Quality of Life

Abstract

Publication types

MeSH terms

Grants and funding