Document Detail


Aicardi syndrome accompanied by auditory disturbance and multiple brain tumors.
MedLine Citation:
PMID:  1667249     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We described a 5-month-old girl with Aicardi syndrome accompanied by auditory disturbance and multiple brain tumors. She was admitted to our hospital because she suffered from intractable flexor spasms. Physical examination revealed craniofacial asymmetry, left auricular deformity, scoliosis, and remarkable hypotonia with psychomotor retardation. Abnormal ophthalmological findings included chorioretinopathy with pale and round-shaped peripapillary lacunae, and there was modified hypsarrhythmia in her EEG. MRI revealed multiple brain tumors in the 3rd and the lateral ventricles which are considered to be choroid plexus papilloma with agenesis of the corpus callosum. ACTH therapy was administered because of the intractable seizures. After ACTH therapy, the thresholds of waves I and V were much improved. The interpeak latency of waves I-V of the left ear and the peak latency of wave I of the right ear had been lengthened. Acoustic reflex with contralateral stimulation showed no response in the left ear. These findings indicate that the auditory system is also involved in the Aicardi syndrome and that ACTH is effective for its dysfunction.
Authors:
K Hamano; T Matsubara; S Shibata; C Hirano; Z Ito; Y Ase; J Kusakari; H Takita
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Brain & development     Volume:  13     ISSN:  0387-7604     ISO Abbreviation:  Brain Dev.     Publication Date:  1991 Nov 
Date Detail:
Created Date:  1992-05-26     Completed Date:  1992-05-26     Revised Date:  2006-11-15    
Medline Journal Info:
Nlm Unique ID:  7909235     Medline TA:  Brain Dev     Country:  JAPAN    
Other Details:
Languages:  eng     Pagination:  438-41     Citation Subset:  IM    
Affiliation:
Department of Pediatrics, University of Tsukuba, Ibaraki, Japan.
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MeSH Terms
Descriptor/Qualifier:
Acoustic Impedance Tests
Adrenocorticotropic Hormone / pharmacology
Audiometry
Brain Neoplasms / congenital*,  pathology
Corpus Callosum / abnormalities*,  pathology
Female
Hearing Disorders / congenital*,  pathology
Humans
Infant
Magnetic Resonance Imaging
Reflex / physiology
Syndrome
Chemical
Reg. No./Substance:
9002-60-2/Adrenocorticotropic Hormone

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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