HONG Meng-di,YU Si-han,KANG Shuo-shuo,et al.Perrault Syndrome Disease Characterization and Cochlear Implantation[J].Chinese Scientific Journal of Hearing and Speech Rehabilitation,2025,23(01):5-7.
HONG Meng-di,YU Si-han,KANG Shuo-shuo,et al.Perrault Syndrome Disease Characterization and Cochlear Implantation[J].Chinese Scientific Journal of Hearing and Speech Rehabilitation,2025,23(01):5-7.DOI: 10.3969/j.issn.1672-4933.2025.01.002.
Perrault Syndrome Disease Characterization and Cochlear Implantation
This article describes the diagnosis of Perrault syndrome in a male child with profound sensorineural deafness in both ears and gait instability who was successfully implanted with a cochlear implant.
Methods
2
Analyze and organize the preoperative evaluations
surgical procedures
postoperative adjustment parameters
and efficacy assessments of a patient with Perrault Syndrome. Observe the patient's auditory outcomes with a cochlear implant and speech discrimination ability.
Results
2
The patient was diagnosed with Perrault
Syndrome based on the results of genetic testing that confirmed compound heterozygous
TWNK
missense variants (c.1172G
>
A: p.Arg391His and c.1844G
>
C: p.Gly615Ala). The audiological manifestations
in conjunction with clinical findings
are indicative of Auditory Neuropathy Spectrum Disorder (ANSD). The patient
five years after cochlear implant
has an average aided hearing threshold of 30 dB HL and can distinguish and imitate the Ling's Sounds. The Categories of Auditory Performance is at level 6. The Speech Intelligibility Rating is at level 4. The patient can communicate daily with family members
but has slightly poor speech expression ability.
Conclusion
2
Patients with Perrault syndrome who have hearing loss can restore auditory and verbal communication ability after cochlear implantation. The early genetic diagnosis of hearing loss patients plays an important role in the treatment plan
intervention methods and intervention efficacy of patients.
Geethalakshmi S , Narendrakumar V . Perrault Syndrome - A Rare Case Report [J ] . J Clin Diagn Res , 2015 , 9 ( 3 ): 1 - 2 .
Gottschalk ME , Coker SB , Fox LA . Neurologic anomalies of Perrault syndrome [J ] . American Journal of Medical Genetics , 2010 , 65 ( 4 ): 274 - 276 .
Ideura M , Nishio SY , Moteki H , et al . Comprehensive analysis of syndromic hearing loss patients in Japan [J ] . Scientific Reports , 2019 , 9 ( 1 ): 11976 .
Faridi R , Rea A , Fenollar-Ferrer C , et al . New insights into Perrault syndrome, a clinically and genetically heterogeneous disorder [J ] . Human Genetics , 2021 , 8 ( 2 ): 721 - 737 .
Morino H , Pierce SB , Matsuda Y , et al . Mutations in Twinkle primase-helicase cause Perrault syndrome with neurologic features [J ] . Neurology , 2014 , 83 ( 22 ): 2054 - 2061 .
Gotta F , Lamp M , Geroldi A , et al . A novel mutation of Twinkle in Perrault syndrome: A not rare diagnosis? [J ] . Annals of human genetics , 2020 , 84 ( 5 ): 417 - 422 .
Quality of Life Assessment of Children with Cochlear Implants in Shanghai: A Retrospective Analysis Based on the Nijmegen Scale
Preoperative Multidisciplinary Assessment for Cochlear Implantation
Preliminary Observation of Electrically Evoked Cortical Auditory Potential P1-N1 in Patients with No Residual Hearing
A Preliminary Study to Investigate the Correlation Between Position-Specific Effect of Interphase Gap and Competing Acoustic Environments Speech Recognition in Cochlear Implant Receivers
Integrated Intracochlear Electrocochleography during Cochlear Implantation: Clinical Application and Proposal for a Standardized Protocol