Kaitlin A Greene1,2, Vivien Lu1,3, Marta San Luciano4, William Qubty5, Samantha L Irwin1, Barbara Grimes6, Amy A Gelfand7. 1. Child & Adolescent Headache Program, Department of Neurology, University of California, San Francisco, San Francisco, CA, USA. 2. Division of Pediatric Neurology, Oregon Health & Science University, Portland, OR, USA. 3. California Northstate University College of Medicine, Elk Grove, CA, USA. 4. Movement Disorders and Neuromodulation Center, Department of Neurology, University of California, San Francisco, San Francisco, CA, USA. 5. Pediatric Headache Program, Department of Neurology, Dell Medical School at the University of Texas at Austin, Austin, TX, USA. 6. Department of Epidemiology & Biostatistics, University of California, San Francisco, San Francisco, CA, USA. 7. Child & Adolescent Headache Program, Department of Neurology, University of California, San Francisco, San Francisco, CA, USA. amy.gelfand@ucsf.edu.
Abstract
BACKGROUND: Benign paroxysmal torticollis (BPT) is characterized by attacks of head tilt associated with vomiting, irritability, and/or ataxia in early childhood. BPT is associated with migraine but risk factors are unknown. Impact on quality of life is also unknown. METHODS: Parents/caregivers of children with ongoing or resolved BPT participated in telephone interviews (n = 73). Those with ongoing BPT completed the Infant Toddler Quality of Life questionnaire (ITQoL). RESULTS: Median age of children at the time of interview was 2.9 years (range 0.25-23). BPT was ongoing in 52% (n = 38). Nineteen percent (n = 14) developed migraine (median age 9.25 years, range 2.5-23) and 63% (n = 46) developed another episodic syndrome associated with migraine. Proportion of patients who developed migraine was higher among those with certain migrainous symptoms during BPT attacks vs. those without: phonophobia (58 vs. 21%, p = 0.02), photophobia and phonophobia (55 vs. 23%, p = 0.05), and photophobia, phonophobia, and motion sensitivity (60 vs. 22%, p = 0.02). ITQoL results showed significant impact of BPT on quality of life. CONCLUSIONS: Children with BPT may develop migraine or other episodic syndromes associated with migraine. Presence of migrainous features during BPT episodes may increase likelihood of developing migraine. Though characterized as "benign," BPT can significantly impact children and families. IMPACT: Benign paroxysmal torticollis (BPT) is a rare condition of early childhood characterized by episodes of head tilt associated with vomiting, irritability, ataxia, pallor, and/or malaise. This cohort study describes the phenotypic spectrum of BPT, variable treatment, natural history and association with migraine, and impact on development and quality of life. Children with BPT may go on to develop migraine or episodic syndromes that may be associated with migraine; presence of migrainous features during attacks may increase odds of developing migraine. BPT can have significant impact on quality of life, demonstrated by findings from the Infant Toddler Quality of Life questionnaire.
BACKGROUND: Benign paroxysmal torticollis (BPT) is characterized by attacks of head tilt associated with vomiting, irritability, and/or ataxia in early childhood. BPT is associated with migraine but risk factors are unknown. Impact on quality of life is also unknown. METHODS: Parents/caregivers of children with ongoing or resolved BPT participated in telephone interviews (n = 73). Those with ongoing BPT completed the Infant Toddler Quality of Life questionnaire (ITQoL). RESULTS: Median age of children at the time of interview was 2.9 years (range 0.25-23). BPT was ongoing in 52% (n = 38). Nineteen percent (n = 14) developed migraine (median age 9.25 years, range 2.5-23) and 63% (n = 46) developed another episodic syndrome associated with migraine. Proportion of patients who developed migraine was higher among those with certain migrainous symptoms during BPT attacks vs. those without: phonophobia (58 vs. 21%, p = 0.02), photophobia and phonophobia (55 vs. 23%, p = 0.05), and photophobia, phonophobia, and motion sensitivity (60 vs. 22%, p = 0.02). ITQoL results showed significant impact of BPT on quality of life. CONCLUSIONS: Children with BPT may develop migraine or other episodic syndromes associated with migraine. Presence of migrainous features during BPT episodes may increase likelihood of developing migraine. Though characterized as "benign," BPT can significantly impact children and families. IMPACT: Benign paroxysmal torticollis (BPT) is a rare condition of early childhood characterized by episodes of head tilt associated with vomiting, irritability, ataxia, pallor, and/or malaise. This cohort study describes the phenotypic spectrum of BPT, variable treatment, natural history and association with migraine, and impact on development and quality of life. Children with BPT may go on to develop migraine or episodic syndromes that may be associated with migraine; presence of migrainous features during attacks may increase odds of developing migraine. BPT can have significant impact on quality of life, demonstrated by findings from the Infant Toddler Quality of Life questionnaire.
Authors: Marta Vila-Pueyo; Gemma G Gené; Marina Flotats-Bastardes; Xabier Elorza; Cèlia Sintas; Miguel A Valverde; Bru Cormand; José M Fernández-Fernández; Alfons Macaya Journal: Eur J Paediatr Neurol Date: 2014-01-08 Impact factor: 3.140
Authors: Véronique Humbertclaude; Benjamin Krams; Erika Nogue; Nicolas Nagot; Daniel Annequin; Barbara Tourniaire; Elisabeth Tournier-Lasserve; Florence Riant; Agathe Roubertie Journal: Dev Med Child Neurol Date: 2018-06-21 Impact factor: 5.449
Authors: David W Dodick; Stewart J Tepper; Deborah I Friedman; Amy A Gelfand; Donald J Kellerman; Peter C Schmidt Journal: Headache Date: 2018-05-21 Impact factor: 5.887