More Than a Decade of Misdiagnosis of Alternating Hemiplegia of Childhood with Catastrophic Outcome

Alternating hemiplegia of childhood (AHC) is a distinct clinical disorder characterized by recurrent episodes of hemiplegia, abnormal ocular movement, and progressive developmental delay. It is an extremely rare genetic disorder related to ATP1A3 gene mutations. In this paper, we present a case of A...

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Main Authors: Hussein Algahtani, Bashair Ibrahim, Bader Shirah, Ahmad Aldarmahi, Ahad Abdullah
Format: Article
Language:English
Published: Wiley 2017-01-01
Series:Case Reports in Medicine
Online Access:http://dx.doi.org/10.1155/2017/5769837
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author Hussein Algahtani
Bashair Ibrahim
Bader Shirah
Ahmad Aldarmahi
Ahad Abdullah
author_facet Hussein Algahtani
Bashair Ibrahim
Bader Shirah
Ahmad Aldarmahi
Ahad Abdullah
author_sort Hussein Algahtani
collection DOAJ
description Alternating hemiplegia of childhood (AHC) is a distinct clinical disorder characterized by recurrent episodes of hemiplegia, abnormal ocular movement, and progressive developmental delay. It is an extremely rare genetic disorder related to ATP1A3 gene mutations. In this paper, we present a case of AHC in which the diagnosis was missed for many years until severe hypoxic brain insult occurred from prolonged status epilepticus. Not only we are presenting an interesting clinical entity and radiological images, but also we are shedding the light on a rare genetic disease with catastrophic sequelae. The challenges in diagnosis and treatment lead to a poor outcome as seen in our case. Although early recognition and accurate diagnosis and treatment of the disease may not change the outcome, counseling of the family may change their expectation and reduce their frustration. Referral to a center with expertise in genetic disorders and access to genetic laboratories is of paramount importance in the diagnosis of this disease. Due to the rarity of this disease in Saudi Arabia, a genotype-phenotype correlation is not feasible.
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spelling doaj-art-e1c7bf275bd1430595236ad8a3c954aa2025-08-20T02:21:38ZengWileyCase Reports in Medicine1687-96271687-96352017-01-01201710.1155/2017/57698375769837More Than a Decade of Misdiagnosis of Alternating Hemiplegia of Childhood with Catastrophic OutcomeHussein Algahtani0Bashair Ibrahim1Bader Shirah2Ahmad Aldarmahi3Ahad Abdullah4King Abdulaziz Medical City, King Saud bin Abdulaziz University for Health Sciences, Jeddah, Saudi ArabiaKing Abdulaziz University, Jeddah, Saudi ArabiaKing Abdullah International Medical Research Center, King Saud bin Abdulaziz University for Health Sciences, Jeddah, Saudi ArabiaKing Saud bin Abdulaziz University for Health Sciences, Jeddah, Saudi ArabiaBatterjee Medical College, Jeddah, Saudi ArabiaAlternating hemiplegia of childhood (AHC) is a distinct clinical disorder characterized by recurrent episodes of hemiplegia, abnormal ocular movement, and progressive developmental delay. It is an extremely rare genetic disorder related to ATP1A3 gene mutations. In this paper, we present a case of AHC in which the diagnosis was missed for many years until severe hypoxic brain insult occurred from prolonged status epilepticus. Not only we are presenting an interesting clinical entity and radiological images, but also we are shedding the light on a rare genetic disease with catastrophic sequelae. The challenges in diagnosis and treatment lead to a poor outcome as seen in our case. Although early recognition and accurate diagnosis and treatment of the disease may not change the outcome, counseling of the family may change their expectation and reduce their frustration. Referral to a center with expertise in genetic disorders and access to genetic laboratories is of paramount importance in the diagnosis of this disease. Due to the rarity of this disease in Saudi Arabia, a genotype-phenotype correlation is not feasible.http://dx.doi.org/10.1155/2017/5769837
spellingShingle Hussein Algahtani
Bashair Ibrahim
Bader Shirah
Ahmad Aldarmahi
Ahad Abdullah
More Than a Decade of Misdiagnosis of Alternating Hemiplegia of Childhood with Catastrophic Outcome
Case Reports in Medicine
title More Than a Decade of Misdiagnosis of Alternating Hemiplegia of Childhood with Catastrophic Outcome
title_full More Than a Decade of Misdiagnosis of Alternating Hemiplegia of Childhood with Catastrophic Outcome
title_fullStr More Than a Decade of Misdiagnosis of Alternating Hemiplegia of Childhood with Catastrophic Outcome
title_full_unstemmed More Than a Decade of Misdiagnosis of Alternating Hemiplegia of Childhood with Catastrophic Outcome
title_short More Than a Decade of Misdiagnosis of Alternating Hemiplegia of Childhood with Catastrophic Outcome
title_sort more than a decade of misdiagnosis of alternating hemiplegia of childhood with catastrophic outcome
url http://dx.doi.org/10.1155/2017/5769837
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