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[Central sleep apnea syndrome as the first sign of type I Chiari malformation].
An Pediatr (Barc). 2008 Mar; 68(3):277-81.AP

Abstract

INTRODUCTION

Type I Chiari malformation consists on the caudal displacement of cerebellar tonsils through the foramen magnum. It is often asymptomatic, although it may display symptoms as a result of cerebellum, brainstem, high cervical spinal cord or the lower cranial nerve, involvement.

OBJECTIVE

We report our experience over the last 16 years. We have identified 16 patients with type I Chiari malformation. Only 2 cases showed common type I Chiari symptoms and just one had respiratory disorder as the first clinical sign.

CLINICAL CASE

A 15 year old girl presented with a 5 years' history of chronic daily cough aggravated by exercise. Snoring and sleep apnea had been noted by her mother for 1 year. The girl eventually suffered from migraine and diurnal hypersomnolence. The physical and neurological examination was normal with the only exception being the absence of bilateral nauseous reflex. A nocturnal polysomnography study demonstrated a pseudoperiodic pattern with apnea pauses associated to cycles of deep breathing, resulting in severe gasometric repercussion and bradycardia. Magnetic resonance imaging of the brain showed Chiari I malformation. Non-invasive mechanical ventilation treatment significantly improved the clinical symptoms and gasometric analysis.

DISCUSSION

Surgical posterior fossa decompression is discussed. Early decompression before appearance of irreversible neurological damage is recommended. It is associated with a significant reduction in the number of central apneas and sleep arousals. Surgical intervention is recommended in symptomatic patients and in cases of radiographic Chiari malformation or syrinx progression.

Authors+Show Affiliations

Sección de Neuropediatría, Hospital Infantil Universitario Miguel Servet, Po. Isabel la Católica 1 y 3, Zaragoza, Spain.No affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
English Abstract
Journal Article

Language

spa

PubMed ID

18358141

Citation

Miralbés Terraza, S, et al. "[Central Sleep Apnea Syndrome as the First Sign of Type I Chiari Malformation]." Anales De Pediatria (Barcelona, Spain : 2003), vol. 68, no. 3, 2008, pp. 277-81.
Miralbés Terraza S, García Oguiza A, López Pisón J, et al. [Central sleep apnea syndrome as the first sign of type I Chiari malformation]. An Pediatr (Barc). 2008;68(3):277-81.
Miralbés Terraza, S., García Oguiza, A., López Pisón, J., Sáenz Moreno, I., Jiménez Escobar, V., Peña Segura, J. L., & Marco Rived, A. (2008). [Central sleep apnea syndrome as the first sign of type I Chiari malformation]. Anales De Pediatria (Barcelona, Spain : 2003), 68(3), 277-81.
Miralbés Terraza S, et al. [Central Sleep Apnea Syndrome as the First Sign of Type I Chiari Malformation]. An Pediatr (Barc). 2008;68(3):277-81. PubMed PMID: 18358141.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - [Central sleep apnea syndrome as the first sign of type I Chiari malformation]. AU - Miralbés Terraza,S, AU - García Oguiza,A, AU - López Pisón,J, AU - Sáenz Moreno,I, AU - Jiménez Escobar,V, AU - Peña Segura,J L, AU - Marco Rived,A, PY - 2008/3/25/pubmed PY - 2008/8/21/medline PY - 2008/3/25/entrez SP - 277 EP - 81 JF - Anales de pediatria (Barcelona, Spain : 2003) JO - An Pediatr (Barc) VL - 68 IS - 3 N2 - INTRODUCTION: Type I Chiari malformation consists on the caudal displacement of cerebellar tonsils through the foramen magnum. It is often asymptomatic, although it may display symptoms as a result of cerebellum, brainstem, high cervical spinal cord or the lower cranial nerve, involvement. OBJECTIVE: We report our experience over the last 16 years. We have identified 16 patients with type I Chiari malformation. Only 2 cases showed common type I Chiari symptoms and just one had respiratory disorder as the first clinical sign. CLINICAL CASE: A 15 year old girl presented with a 5 years' history of chronic daily cough aggravated by exercise. Snoring and sleep apnea had been noted by her mother for 1 year. The girl eventually suffered from migraine and diurnal hypersomnolence. The physical and neurological examination was normal with the only exception being the absence of bilateral nauseous reflex. A nocturnal polysomnography study demonstrated a pseudoperiodic pattern with apnea pauses associated to cycles of deep breathing, resulting in severe gasometric repercussion and bradycardia. Magnetic resonance imaging of the brain showed Chiari I malformation. Non-invasive mechanical ventilation treatment significantly improved the clinical symptoms and gasometric analysis. DISCUSSION: Surgical posterior fossa decompression is discussed. Early decompression before appearance of irreversible neurological damage is recommended. It is associated with a significant reduction in the number of central apneas and sleep arousals. Surgical intervention is recommended in symptomatic patients and in cases of radiographic Chiari malformation or syrinx progression. SN - 1695-4033 UR - https://www.unboundmedicine.com/medline/citation/18358141/[Central_sleep_apnea_syndrome_as_the_first_sign_of_type_I_Chiari_malformation]_ L2 - http://www.elsevier.es/en/linksolver/ft/ivp/1695-4033/68/277 DB - PRIME DP - Unbound Medicine ER -