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Neuropediatrics 2012; 43(01): 027-036
DOI: 10.1055/s-0032-1307457
Original Article
Thieme Medical Publishers 333 Seventh Avenue, New York, NY 10001, USA.

Botulinum Toxin Type A and B for the Reduction of Hypersalivation in Children with Neurological Disorders: A Focus on Effectiveness and Therapy Adherence

A. Sebastian Schroeder
1   Department of Paediatric Neurology and Developmental Medicine, Hauner Children's Hospital, Campus Innenstadt, University of Munich, Germany
2   Integrated Center for Research and Treatment of Vertigo,, Balance and Ocular Motor Disorders, IFBLMU Campus Großhadern, University of Munich, Germany
,
Theresia Kling
1   Department of Paediatric Neurology and Developmental Medicine, Hauner Children's Hospital, Campus Innenstadt, University of Munich, Germany
,
Kristina Huß
1   Department of Paediatric Neurology and Developmental Medicine, Hauner Children's Hospital, Campus Innenstadt, University of Munich, Germany
,
Ingo Borggraefe
1   Department of Paediatric Neurology and Developmental Medicine, Hauner Children's Hospital, Campus Innenstadt, University of Munich, Germany
,
Inga K. Koerte
3   Institute of Clinical Radiology, University of Munich, Germany
,
Astrid Blaschek
1   Department of Paediatric Neurology and Developmental Medicine, Hauner Children's Hospital, Campus Innenstadt, University of Munich, Germany
,
Klaus Jahn
2   Integrated Center for Research and Treatment of Vertigo,, Balance and Ocular Motor Disorders, IFBLMU Campus Großhadern, University of Munich, Germany
,
Florian Heinen
1   Department of Paediatric Neurology and Developmental Medicine, Hauner Children's Hospital, Campus Innenstadt, University of Munich, Germany
2   Integrated Center for Research and Treatment of Vertigo,, Balance and Ocular Motor Disorders, IFBLMU Campus Großhadern, University of Munich, Germany
,
Steffen Berweck
4   Specialist Centre for Paediatric Neurology, Epilepsy Centre for Children and Adolescents, Vogtareuth, Germany
› Author Affiliations
Further Information

Publication History

24 October 2011

01 February 2012

Publication Date:
19 March 2012 (online)

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Abstract

Botulinum toxin (BoNT) is an established treatment option to reduce hypersalivation in children with chronic neurological disorders. Objective of this study was (1) to discriminate differences in efficacy and safety of repeated interventions using BoNT with a focus on different preparations used and (2) to look for effectiveness and treatment adherence from a qualitative research perspective in this single-center cohort study. We prospectively assessed goal attainment scaling, drooling severity and frequency score and the number of towels/day before, and 4 to 8 weeks after intervention. A parent questionnaire assessed therapy-related effects on quality of life retrospectively. A total of 19 out of 34 patients received repeated injections of BoNT (106 total). Mean dose: 95 units onabotulinumtoxinA (Botox®), 2383 units rimabotulinumtoxinB (Neuro-/Myobloc®). Outcome parameters showed a distinct reduction in all treatment groups with a higher efficacy of riB. The child's need for care was reduced in 79% and social interaction improved in 84%. Main reason for discontinuation was “not enough effect” and “formation of antibodies.” riB showed to be more effective in reducing hypersalivation, but antibody-formation seems to be clinically relevant. Despite clinical efficacy treatment adherence is influenced by personal and environmental factors of parents and caretakers balancing the short-term clinical benefit versus the burden of intervention.

Keywords

hypersalivation - sialorrhea - children - botulinum toxin type A - botulinum toxin type B - treatment adherence
 

  • References

  • 1 Tahmassebi JF, Curzon ME. Prevalence of drooling in children with cerebral palsy attending special schools. Dev Med Child Neurol 2003; 45 (9) 613-617
    CrossrefPubMedGoogle Scholar
  • 2 Senner JE, Logemann J, Zecker S, Gaebler-Spira D. Drooling, saliva production, and swallowing in cerebral palsy. Dev Med Child Neurol 2004; 46 (12) 801-806
    PubMedGoogle Scholar
  • 3 Tahmassebi JF, Curzon ME. The cause of drooling in children with cerebral palsy: hypersalivation or swallowing defect?. Int J Paediatr Dent 2003; 2: 106-111
    PubMedGoogle Scholar
  • 4 Erasmus CE, Van Hulst K, Rotteveel LJ , et al . Drooling in cerebral palsy: hypersalivation or dysfunctional oral motor control?. Dev Med Child Neurol 2009; 51 (6) 454-459
    CrossrefPubMedGoogle Scholar
  • 5 Van der Burg JJ, Jongerius PH, Van Hulst K, Van Limbeek J, Rotteveel JJ. Drooling in children with cerebral palsy: effect of salivary flow reduction on daily life and care. Dev Med Child Neurol 2006; 48 (2) 103-107
    CrossrefPubMedGoogle Scholar
  • 6 van der Burg JJ, Jongerius PH, van Limbeek J, van Hulst K, Rotteveel JJ. Drooling in children with cerebral palsy: a qualitative method to evaluate parental perceptions of its impact on daily life, social interaction, and self-esteem. Int J Rehabil Res 2006; 29 (2) 179-182
    CrossrefPubMedGoogle Scholar
  • 7 Little SA, Kubba H, Hussain SS. An evidence-based approach to the child who drools saliva. Clin Otolaryngol 2009; 34 (3) 236-239
    CrossrefPubMedGoogle Scholar
  • 8 Meningaud JP, Pitak-Arnnop P, Chikhani L, Bertrand JC. Drooling of saliva: a review of the etiology and management options. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006; 101 (1) 48-57
    CrossrefPubMedGoogle Scholar
  • 9 Dogu O, Apaydin D, Sevim S, Talas DU, Aral M. Ultrasound-guided versus 'blind' intraparotid injections of botulinum toxin-A for the treatment of sialorrhoea in patients with Parkinson's disease. Clin Neurol Neurosurg 2004; 2: 93-96
    PubMedGoogle Scholar
  • 10 Lipp A, Trottenberg T, Schink T, Kupsch A, Arnold G. A randomized trial of botulinum toxin A for treatment of drooling. Neurology 2003; 61 (9) 1279-1281
    CrossrefPubMedGoogle Scholar
  • 11 Mancini F, Sandrini G, Moglia A, Nappi G, Pacchetti C. A randomised, double-blind, dose-ranging study to evaluate efficacy and safety of three doses of botulinum toxin type A (Botox) for the treatment of spastic foot. Neurol Sci 2005; 26 (1) 26-31
    CrossrefPubMedGoogle Scholar
  • 12 Ondo WG, Hunter C, Moore W. A double-blind placebo-controlled trial of botulinum toxin B for sialorrhea in Parkinson's disease. Neurology 2004; 1: 37-40
    PubMedGoogle Scholar
  • 13 Naumann M, So Y, Argoff CE , et al; Therapeutics and Technology Assessment Subcommittee of the American Academy of Neurology . Assessment: Botulinum neurotoxin in the treatment of autonomic disorders and pain (an evidence-based review): report of the Therapeutics and Technology Assessment Subcommittee of the American Academy of Neurology. Neurology 2008; 70 (19) 1707-1714
    CrossrefPubMedGoogle Scholar
  • 14 Jongerius PH, Rotteveel JJ, van den Hoogen F, Joosten F, van Hulst K, Gabreëls FJ. Botulinum toxin A: a new option for treatment of drooling in children with cerebral palsy. Presentation of a case series. Eur J Pediatr 2001; 160 (8) 509-512
    CrossrefPubMedGoogle Scholar
  • 15 Banerjee KJ, Glasson C, O'Flaherty SJ. Parotid and submandibular botulinum toxin A injections for sialorrhoea in children with cerebral palsy. Dev Med Child Neurol 2006; 48 (11) 883-887
    CrossrefPubMedGoogle Scholar
  • 16 Jongerius PH, van den Hoogen FJ, van Limbeek J, Gabreëls FJ, van Hulst K, Rotteveel JJ. Effect of botulinum toxin in the treatment of drooling: a controlled clinical trial. Pediatrics 2004; 114 (3) 620-627
    CrossrefPubMedGoogle Scholar
  • 17 Reid SM, Johnstone BR, Westbury C, Rawicki B, Reddihough DS. Randomized trial of botulinum toxin injections into the salivary glands to reduce drooling in children with neurological disorders. Dev Med Child Neurol 2008; 50 (2) 123-128
    PubMedGoogle Scholar
  • 18 Suskind DL, Tilton A. Clinical study of botulinum-A toxin in the treatment of sialorrhea in children with cerebral palsy. Laryngoscope 2002; 112 (1) 73-81
    CrossrefPubMedGoogle Scholar
  • 19 van der Burg JJ, Jongerius PH, van Limbeek J, van Hulst K, Rotteveel JJ. Social interaction and self-esteem of children with cerebral palsy after treatment for severe drooling. Eur J Pediatr 2006; 165 (1) 37-41
    CrossrefPubMedGoogle Scholar
  • 20 Wilken B, Aslami B, Backes H. Successful treatment of drooling in children with neurological disorders with botulinum toxin A or B. Neuropediatrics 2008; 39 (4) 200-204
    Article in Thieme ConnectPubMedGoogle Scholar
  • 21 Simpson DM, Gracies JM, Graham HK , et al; Therapeutics and Technology Assessment Subcommittee of the American Academy of Neurology . Assessment: Botulinum neurotoxin for the treatment of spasticity (an evidence-based review): report of the Therapeutics and Technology Assessment Subcommittee of the American Academy of Neurology. Neurology 2008; 70 (19) 1691-1698
    CrossrefPubMedGoogle Scholar
  • 22 Thomas-Stonell N, Greenberg J. Three treatment approaches and clinical factors in the reduction of drooling. Dysphagia 1988; 3 (2) 73-78
    CrossrefPubMedGoogle Scholar
  • 23 Palisano RJ. Validity of goal attainment scaling in infants with motor delays. Phys Ther 1993; 73 (10) 651-658 , discussion 658–660
    PubMedGoogle Scholar
  • 24 Scheffer AR, Erasmus C, van Hulst K, van Limbeek J, Jongerius PH, van den Hoogen FJ. Efficacy and duration of botulinum toxin treatment for drooling in 131 children. Arch Otolaryngol Head Neck Surg 2010; 136 (9) 873-877
    CrossrefPubMedGoogle Scholar
  • 25 Basciani M, Di Rienzo F, Fontana A, Copetti M, Pellegrini F, Intiso D. Botulinum toxin type B for sialorrhoea in children with cerebral palsy: a randomized trial comparing three doses. Dev Med Child Neurol 2011; 53 (6) 559-564
    CrossrefPubMedGoogle Scholar
  • 26 Dressler D, Benecke R. Autonomic side effects of botulinum toxin type B treatment of cervical dystonia and hyperhidrosis. Eur Neurol 2003; 49 (1) 34-38
    CrossrefPubMedGoogle Scholar
  • 27 Tintner R, Gross R, Winzer UF, Smalky KA, Jankovic J. Autonomic function after botulinum toxin type A or B: a double-blind, randomized trial. Neurology 2005; 65 (5) 765-767
    CrossrefPubMedGoogle Scholar
  • 28 Kranz G, Paul A, Voller B , et al . Long-term efficacy and respective potencies of botulinum toxin A and B: a randomized, double-blind study. Br J Dermatol 2011; 164 (1) 176-181
    CrossrefPubMedGoogle Scholar
  • 29 Berweck S, Schroeder AS, Lee SH, Bigalke H, Heinen F. Secondary non-response due to antibody formation in a child after three injections of botulinum toxin B into the salivary glands. Dev Med Child Neurol 2007; 49 (1) 62-64
    CrossrefPubMedGoogle Scholar
  • 30 Erasmus CE, Scheffer AR, van Hulst K , et al . Does motor performance matter in botulinum toxin efficacy for drooling?. Pediatr Neurol 2011; 45 (2) 95-99
    CrossrefPubMedGoogle Scholar
  • 31 Erasmus CE, Van Hulst K, Scheffer AR, Van Limbeek J, Van Den Hoogen FJ, Rotteveel JJ , et al. . What could predict effectiveness of Botulinum Toxin to treat drooling: A search for evidence of discriminatory factors on the level of body functions or structures. Eur J Paediatr Neurol, 2011 DOI: 10.1016/j.ejpn.2011.06.002
    PubMed
  • 32 Brashear A, Bergan K, Wojcieszek J, Siemers ER, Ambrosius W. Patients' perception of stopping or continuing treatment of cervical dystonia with botulinum toxin type A. Mov Disord 2000; 15 (1) 150-153
    CrossrefPubMedGoogle Scholar
  • 33 Linder-Lucht M, Kirschner J, Herrmann J , et al. Why do children with cerebral palsy discontinue therapy with botulinum toxin A?. Dev Med Child Neurol 2006; 4: 319-320
    PubMedGoogle Scholar
  • 34 Cesario S, Morin K, Santa-Donato A. Evaluating the level of evidence of qualitative research. J Obstet Gynecol Neonatal Nurs 2002; 31 (6) 708-714
    PubMedGoogle Scholar