Skip to main content

Advertisement

Log in

Hypersialorrhea in Wilson’s Disease

  • Original Article
  • Published:
Dysphagia Aims and scope Submit manuscript

Abstract

Hypersialorrhea, corresponding to excessive salivation is a symptom frequently reported in Wilson’s disease, especially in its neurological form. The prevalence of this frequent complaint has not been often evaluated. During a 7-month period, 87 consecutive Wilson’s disease patients answered to the simple question “do you have the sensation of excess saliva in your mouth?” to evaluate the frequency of this symptom. A sub-sample of 10 consecutive Wilson’s disease patients with drooling was recruited to undergo quantitative and qualitative measures to evaluate the mechanism of hypersialorrhea. Excessive drooling or excess saliva was found in 46 % of patients followed at the French Reference Centre. Ninety-eight percent of them presented neurological symptoms and drooling was found in only one patient without neurological symptoms. Our study showed that patients with a complaint of excessive saliva produced significantly higher quantities of saliva at rest than controls. Endoscopic examination was abnormal in six patients. A significant decrease of swallowing frequency, longer swallow latencies, and poor swallowing capacities may partly explain the salivary stasis. Oropharyngeal sensitivity disorders were present in 50 % of our patients. The decrease of the swallowing frequency observed in all patients could be related to cognitive and behavioral abnormalities with initiation difficulties objectified by longer latencies triggered by all the ingested volumes. This study confirmed the hypothesis of a multifactorial origin of hypersialorrhea in patients who have been diagnosed in Wilson’s disease. It was essential to evaluate drooling with a multidisciplinary consultation to better identify the underlying mechanisms and to implement strategies for speech therapy and therapeutic adaptation.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1

Similar content being viewed by others

References

  1. Ala A, Walker AP, Ashkan K, Dooley JS, Schilsky ML. Wilson’s disease. Lancet. 2007;369:397–408.

    Article  CAS  PubMed  Google Scholar 

  2. Gitlin JD. Wilson disease. Gastroenterology. 2003;125:1868–77.

    Article  PubMed  Google Scholar 

  3. Woimant F, Chaine P, Favrole P, Mikol J, Chappuis P. Wilson disease. Rev Neurol. 2006;162:773–81.

    Article  CAS  PubMed  Google Scholar 

  4. Kalf JG, de Swart BJ, Borm GF, Bloem BR, Munneke M. Prevalence and definition of drooling in Parkinson’s disease: a systematic review. J Neurol. 2009;256:1391–6.

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  5. Nóbrega AC, Rodrigues B, Torres AC, Scarpel RD, Neves CA, Melo A. Is drooling secondary to a swallowing disorder in patients with Parkinson’s disease? Parkinsonism Relat Disord. 2008;14:243–5.

    Article  PubMed  Google Scholar 

  6. Członkowska A, Tarnacka B, Möller JC, Leinweber B, Bandmann O, Woimant F, Oertel WH. Unified Wilson’s disease rating scale—a proposal for the neurological scoring of Wilson’s disease patients. Neurol Neurochir Pol. 2007;41:1–12.

    PubMed  Google Scholar 

  7. Dubois B, Slachevsky A, Litvan I, Pillon B. The FAB: a frontal assessment battery at bedside. Neurology. 2000;12:1621–6.

    Article  Google Scholar 

  8. Kertesz A, Davidson W, Fox H. Frontal behavioral inventory: diagnostic criteria for frontal lobe dementia. Can J Neurol Sci. 1997;24:29–36.

    CAS  PubMed  Google Scholar 

  9. Thomas-Stonell N, Greenberg J. Three treatment approaches and clinical factors in the reduction of drooling. Dysphagia. 1988;3:75.

    Article  Google Scholar 

  10. Woisard V, Andrieux MP, Puech M. Validation of a self-assessment questionnaire for swallowing disorders (Deglutition Handicap Index). Rev Laryngol Otol Rhinol. 2006;127:315–25.

    CAS  Google Scholar 

  11. Alves C, Brandão M, Andion J, Menezes R. Use of graduated syringes for measuring salivary flow rate: a pilot study. Braz Dent J. 2010;21:401–4.

    Article  PubMed  Google Scholar 

  12. Marks L, Weinreich J. Drooling in Parkinson’s disease: a novel tool for assessment of swallow frequency. Int J Lang Commun Disord. 2001;36:288–91.

    Article  PubMed  Google Scholar 

  13. Borr C, Hielscher-Fastabend M, Lücking A. Reliability and validity of cervical auscultation. Dysphagia. 2007;22:225–34.

    Article  PubMed  Google Scholar 

  14. Santamato A, Panza F, Solfrizzi V, Russo A, Frisardi V, Megna M, Ranieri M, Fiore P. Acoustic analysis of swallowing sounds: a new technique for assessing dysphagia. J Rehabil Med. 2009;41:639–45.

    Article  PubMed  Google Scholar 

  15. Ertekin C, Aydoğdu I, Yüceyar N. Piecemeal deglutition and dysphagia limit in normal subjects and in patients with swallowing disorders. J Neurol Neurosurg Psychiatry. 1996;61:491–6.

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  16. Kalf JG, Munneke M, van den Engel-Hoek L, de Swart BJ, Borm GF, Bloem BR, Zwarts MJ. Pathophysiology of diurnal drooling in Parkinson’s disease. Mov Disord. 2011;26:1670–6.

    Article  PubMed  Google Scholar 

  17. Farneti D. Endoscopic scale for evaluation of the severity of dysphagia: preliminary observations. Rev Laryngol Otol Rhinol. 2008;129:137–40.

    CAS  Google Scholar 

  18. Taly AB, Prashanth LK, Sinha S. Wilson’s disease: An Indian perspective. Neurol India. 2009;57:528–40.

    Article  CAS  PubMed  Google Scholar 

  19. Proulx M, de Courval FP, Wiseman MA, Panisset M. Salivary production in Parkinson’s disease. Mov Disord. 2005;20:204–7.

    Article  PubMed  Google Scholar 

  20. Tumilasci OR, Cersósimo MG, Belforte JE, Micheli FE, Benarroch EE, Pazo JH. Quantitative study of salivary secretion in Parkinson’s disease. Mov Disord. 2006;21:660–7.

    Article  PubMed  Google Scholar 

  21. da Silva-Júnior FP, Carrasco AE, da Silva Mendes AM, Lopes AJ, Nobre E, Souza MA, de Bruin VM. Swallowing dysfunction in Wilson’s disease: a scintigraphic study. Neurogastroenterol Motil. 2008;20:285–90.

    Article  PubMed  Google Scholar 

  22. Gulyas AE, Salazar-Grueso EF. Pharyngeal dysmotility in a patient with Wilson’s disease. Dysphagia. 1988;2:230–4.

    Article  CAS  PubMed  Google Scholar 

  23. Suzuki M, Asada Y, Ito J, Hayashi K, Inoue H, Kitano H. Activation of cerebellum and basal ganglia on volitional swallowing detected by functional magnetic resonance imaging. Dysphagia. 2003;18:71–7.

    Article  PubMed  Google Scholar 

  24. Rodrigues B, Nóbrega AC, Sampaio M, Argolo N, Melo A. Silent saliva aspiration in Parkinson’s disease. Mov Disord. 2011;26:138–41.

    Article  PubMed  Google Scholar 

  25. Kalf JG, Smit AM, Bloem BR, Zwarts MJ, Munneke M. Impact of drooling in Parkinson’s disease. J Neurol. 2007;254:1227–32.

    Article  PubMed  Google Scholar 

  26. Pehlivan M, Yüceyar N, Ertekin C, Celebi G, Ertaş M, Kalayci T, Aydoĝdu I. An electronic device measuring the frequency of spontaneous swallowing: digital phagometer. Dysphagia. 1996;11:259–64.

    Article  CAS  PubMed  Google Scholar 

  27. Wenisch E, De Tassigny A, Trocello JM, Beretti J, Girardot-Tinant N, Woimant F. Cognitive profile in Wilson’s disease: a case series of 31 patients. Rev Neurol. 2013;169:944–9.

    Article  CAS  PubMed  Google Scholar 

  28. Watanabe Y, Abe S, Ishikawa T, Yamada Y, Yamane GY. Cortical regulation during the early stage of initiation of voluntary swallowing in humans. Dysphagia. 2004;19:100–8.

    Article  PubMed  Google Scholar 

  29. Satow T, Ikeda A, Yamamoto J, Begum T, Thuy DH, Matsuhashi M, Mima T, Nagamine T, Baba K, Mihara T, Inoue Y, Miyamoto S, Hashimoto N, Shibasaki H. Role of primary sensorimotor cortex and supplementary motor area in volitional swallowing: a movement-related cortical potential study. Am J Physiol Gastrointest Liver Physiol. 2004;287:G459–70.

    Article  CAS  PubMed  Google Scholar 

  30. Pernon M, Trocello JM, Vaissière J, Cousin C, Chevaillier G, Rémy P, Kidri-Osmani K, Fougeron C, Woimant F. Could speech rate of Wilson’s disease dysarthric patient be improved in dual task condition? Rev Neurol. 2013;. doi:10.1016/j.neurol.2012.12.003.

    PubMed  Google Scholar 

  31. Nóbrega AC, Rodrigues B, Melo A. Is silent aspiration a risk factor for respiratory infection in Parkinson’s disease patients? Parkinsonism Relat Disord. 2008;14:646–8.

    Article  PubMed  Google Scholar 

  32. Miller N, Noble E, Jones D, Burn D. Hard to swallow: dysphagia in Parkinson’s disease. Age Ageing. 2006;35:614–8.

    Article  PubMed  Google Scholar 

  33. Kalf JG, Bloem BR, Munneke M. Diurnal and nocturnal drooling in Parkinson’s disease. J Neurol. 2012;259:119–23.

    Article  PubMed Central  CAS  PubMed  Google Scholar 

  34. Brodsky MB, Verdolini Abbott K, McNeil MR, Palmer CV, Grayhack JP, Martin-Harris B. Effects of divided attention on swallowing in persons with idiopathic Parkinson’s disease. Dysphagia. 2012;27:390–400.

    Article  PubMed  Google Scholar 

Download references

Conflict of interest

None.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to France Woimant.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Trocello, JM., Osmani, K., Pernon, M. et al. Hypersialorrhea in Wilson’s Disease. Dysphagia 30, 489–495 (2015). https://doi.org/10.1007/s00455-015-9627-0

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00455-015-9627-0

Keywords

Navigation