Benign paroxysmal torticollis in infancy – diagnostic error possibility

Background/Aim. Benign paroxysmal torticollis (BPT) is a rare paroxysmal dyskinesia characterized by attacks of head tilt alone or tilt accompanied by vomiting and ataxia, which may last hours to days. It is claimed that BPT disappears completely in childhood, but that it can evolve into other condi...

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Main Authors: Zlatanović Dragan, Dimitrijević Lidija, Stanković Anita, Balov Biljana
Format: Article
Language:English
Published: Ministry of Defence of the Republic of Serbia, University of Defence, Belgrade 2017-01-01
Series:Vojnosanitetski Pregled
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Online Access:http://www.doiserbia.nb.rs/img/doi/0042-8450/2017/0042-84501600223Z.pdf
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author Zlatanović Dragan
Dimitrijević Lidija
Stanković Anita
Balov Biljana
author_facet Zlatanović Dragan
Dimitrijević Lidija
Stanković Anita
Balov Biljana
author_sort Zlatanović Dragan
collection DOAJ
description Background/Aim. Benign paroxysmal torticollis (BPT) is a rare paroxysmal dyskinesia characterized by attacks of head tilt alone or tilt accompanied by vomiting and ataxia, which may last hours to days. It is claimed that BPT disappears completely in childhood, but that it can evolve into other conditions, such as benign paroxysmal vertigo, cyclical vomiting syndrome, abdominal migraine, hemiplegic migraine, motion sickness and/or migraine with aura. The aim of this manuscript was to renew focus on benign paroxysmal torticollis because the disorder is almost always under-recognized by pediatric practitioners, who often order extensive and unrewarding testing and physiotherapy treatment. Methods. Twelve BPT cases observed during a 5-year period (2009–2014) at the Clinical Centre Niš, Niš, Serbia were reviewed. Data were collected on the features of torticollis, the age of onset, the duration of episodes, associated symptoms, the frequency of episodes, the persistence of symptoms over time, the age when the disorder finally disappeared, sequelae appearing after the 5th birthday, and family history of BPT, migraine or kinetosis. All the children were followed for periods ranging from 48 to 72 months. Results. The series included 6 females and 6 males. The age at onset of BPT was less than 8 months in 84% of the cases. Episodes of torticollis occurred suddenly on waking in all the cases without any trigger factors. The duration of torticollis ranged from a few hours to a few weeks. In 58% of cases, the condition persisted for more than one week. The frequency of the episodes ranged from once every 3 days to once every 25 days. The episodes were more frequent and lasted longer in the early months and tended to cease as the child became older. The age when episodes ended ranged from 11 months to 62 months. In 11 (91.66%) cases, the disorder disappeared before the patient's 5th birthday. No patient had a family history of BPT. In 6 cases, family members had kinetosis. In 5 cases, family members were positive for both migraine and kinetosis. All the children had normal motor development and normal speech and language development. After the disappearance of BPT, two children developed other forms of periodic syndromes: one boy had migraine with aura, and one girl experienced cyclic vomiting. Conclusion. BPT is probably an age-sensitive and migraine-related disorder that is benign in nature. The disorder is often misinterpreted, and children may pointlessly undergo numerous tests. Therefore, it is very important to recognize and observe this condition in order to avoid extensive, unnecessary and unpleasant procedures on the child.
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spelling doaj-art-95b631e67fdf49e7be6a635b3f84f69d2025-08-20T02:10:06ZengMinistry of Defence of the Republic of Serbia, University of Defence, BelgradeVojnosanitetski Pregled0042-84502406-07202017-01-0174546346610.2298/VSP150504223Z0042-84501600223ZBenign paroxysmal torticollis in infancy – diagnostic error possibilityZlatanović Dragan0Dimitrijević Lidija1Stanković Anita2Balov Biljana3Clinical Center Niš, Physical Medicine and Rehabilitation Clinic, Pediatric Rehabilitation Department, NišClinical Center Niš, Physical Medicine and Rehabilitation Clinic, Pediatric Rehabilitation Department, Niš + Faculty of Medicine, NišClinical Center Niš, Physical Medicine and Rehabilitation Clinic, Pediatric Rehabilitation Department, NišHigh Medical School, Physiotherapy Department, ĆuprijaBackground/Aim. Benign paroxysmal torticollis (BPT) is a rare paroxysmal dyskinesia characterized by attacks of head tilt alone or tilt accompanied by vomiting and ataxia, which may last hours to days. It is claimed that BPT disappears completely in childhood, but that it can evolve into other conditions, such as benign paroxysmal vertigo, cyclical vomiting syndrome, abdominal migraine, hemiplegic migraine, motion sickness and/or migraine with aura. The aim of this manuscript was to renew focus on benign paroxysmal torticollis because the disorder is almost always under-recognized by pediatric practitioners, who often order extensive and unrewarding testing and physiotherapy treatment. Methods. Twelve BPT cases observed during a 5-year period (2009–2014) at the Clinical Centre Niš, Niš, Serbia were reviewed. Data were collected on the features of torticollis, the age of onset, the duration of episodes, associated symptoms, the frequency of episodes, the persistence of symptoms over time, the age when the disorder finally disappeared, sequelae appearing after the 5th birthday, and family history of BPT, migraine or kinetosis. All the children were followed for periods ranging from 48 to 72 months. Results. The series included 6 females and 6 males. The age at onset of BPT was less than 8 months in 84% of the cases. Episodes of torticollis occurred suddenly on waking in all the cases without any trigger factors. The duration of torticollis ranged from a few hours to a few weeks. In 58% of cases, the condition persisted for more than one week. The frequency of the episodes ranged from once every 3 days to once every 25 days. The episodes were more frequent and lasted longer in the early months and tended to cease as the child became older. The age when episodes ended ranged from 11 months to 62 months. In 11 (91.66%) cases, the disorder disappeared before the patient's 5th birthday. No patient had a family history of BPT. In 6 cases, family members had kinetosis. In 5 cases, family members were positive for both migraine and kinetosis. All the children had normal motor development and normal speech and language development. After the disappearance of BPT, two children developed other forms of periodic syndromes: one boy had migraine with aura, and one girl experienced cyclic vomiting. Conclusion. BPT is probably an age-sensitive and migraine-related disorder that is benign in nature. The disorder is often misinterpreted, and children may pointlessly undergo numerous tests. Therefore, it is very important to recognize and observe this condition in order to avoid extensive, unnecessary and unpleasant procedures on the child.http://www.doiserbia.nb.rs/img/doi/0042-8450/2017/0042-84501600223Z.pdftorticollisdiagnosisdiagnostic errorschild
spellingShingle Zlatanović Dragan
Dimitrijević Lidija
Stanković Anita
Balov Biljana
Benign paroxysmal torticollis in infancy – diagnostic error possibility
Vojnosanitetski Pregled
torticollis
diagnosis
diagnostic errors
child
title Benign paroxysmal torticollis in infancy – diagnostic error possibility
title_full Benign paroxysmal torticollis in infancy – diagnostic error possibility
title_fullStr Benign paroxysmal torticollis in infancy – diagnostic error possibility
title_full_unstemmed Benign paroxysmal torticollis in infancy – diagnostic error possibility
title_short Benign paroxysmal torticollis in infancy – diagnostic error possibility
title_sort benign paroxysmal torticollis in infancy diagnostic error possibility
topic torticollis
diagnosis
diagnostic errors
child
url http://www.doiserbia.nb.rs/img/doi/0042-8450/2017/0042-84501600223Z.pdf
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AT dimitrijeviclidija benignparoxysmaltorticollisininfancydiagnosticerrorpossibility
AT stankovicanita benignparoxysmaltorticollisininfancydiagnosticerrorpossibility
AT balovbiljana benignparoxysmaltorticollisininfancydiagnosticerrorpossibility