Abstract
This paper reports the longitudinal clinical, neurocognitive, and neuroradiological findings in an adolescent patient with nonprogressive motor and cognitive disturbances consistent with a diagnosis of developmental coordination disorder (DCD). In addition to prototypical DCD, the development of mastication was severely impaired, while no evidence of swallowing apraxia, dysphagia, sensorimotor disturbances, abnormal tone, or impaired general cognition was found. He suffered from bronchopulmonary dysplasia and was ventilated as a newborn for 1.5 months. At the age of 3 months, a ventriculoperitoneal shunt was surgically installed because of obstructive hydrocephalus secondary to perinatal intraventricular bleeding. At the age of 5 years, the patient’s attempts to masticate were characterized by rough, effortful, and laborious biting movements confined to the vertical plane. Solid food particles had a tendency to get struck in his mouth and there was constant spillage. As a substitute for mastication, he moved the unground food with his fingers in a lateral direction to the mandibular and maxillary vestibule to externally manipulate and squeeze the food between cheek and teeth with the palm of his hand. Once the food was sufficiently soft, the bolus was correctly transported by the tongue in posterior direction and normal deglutition took place. Repeat magnetic resonance imaging (MRI) during follow-up disclosed mild structural abnormalities as the sequelae of the perinatal intraventricular bleeding, but this could not explain impaired mastication behavior. Quantified Tc-99m-ethylcysteinate dimer single-photon emission computed tomography (Tc-99m-ECD SPECT), however, revealed decreased perfusion in the left cerebellar hemisphere, as well as in both inferior lateral frontal regions, both motor cortices, and the right anterior and lateral temporal areas. Anatomoclinical findings in this patient with DCD not only indicate that the functional integrity of the cerebellocerebral network is crucially important in the planning and execution of skilled actions, but also seem to show for the first time that mastication deficits may be of true apraxic origin. As a result, it is hypothesized that “mastication dyspraxia” may have to be considered as a distinct nosological entity within the group of the developmental dyspraxias following a disruption of the cerebellocerebral network involved in planned actions.
Similar content being viewed by others
References
Seikel JA, King DW, Drumright DG. Anatomy and physiology for speech, language, and hearing. New York: Delmar; 2010.
Lund JP. Mastication and its control by the brainstem. Crit Rev Oral Biol Med. 1991;2:33–64.
Soboleva U, Laurina L, Slaidina A. The masticatory system—an overview. Stomatologija. 2005;7:77–80.
Momose I, Nishikawa J, Watanabe T, Sasaki Y, Senda M, Kubota K, et al. Effect of mastication on regional cerebral blood flow in humans examined by positron-emission tomography with 15O-labelled water and magnetic resonance imaging. Arch Oral Biol. 1997;42:57–61.
Onozuka M, Fujita M, Watanabe K, Hirano Y, Niwa M, Nishiyama K, et al. Mapping brain region activity during chewing: a functional magnetic resonance imaging study. J Dent Res. 2002;81:743–6.
Heilman KM, Gonzalez Rothi LJ, Hanna-Pladdy B. The forelimb apraxias. In: Mariën P, Abutalebi J, editors. Neuropsychological research: a review. Hove: Psychology; 2008. p. 162–83.
Goldenberg G. Apraxia. Neuropsychology and behavioral neurology. In: Goldenberg G, Miller BL, editors. Handbook of clinical neurology, vol. 88. 3rd ed. Amsterdam: Elsevier; 2008. p. 323–37.
Snijders JT, Tellegen PJ, Laros JA. Snijders-Oomen Niet-Verbale Intelligentietest (SON-R). Verantwoording en handleiding. Groningen: Wolters Noordhoff; 1988.
Wechsler D. Wechsler Intelligence Scale for Children. 3rd ed. London: The Psychological Corporation; 2002.
Beery KE. The Developmental Test of Visual–Motor Integration: administration, scoring, and teaching manual. 3rd ed. Cleveland: Modern Curriculum; 1989.
Osterrieth PA. Le Test de Copie d’une Figure Complexe. Neuchatel: Delachaux & Niestle; 1944.
Kleber EW, Kleber G. Differentieller Leistungstest—KE. Göttingen: Hogrefe; 1974.
Vos PG. Bourdon–Vos test. 2nd ed. Lisse: Swets & Zeitlinger; 1998.
Van Bon WHJ, Hoekstra JG. Taaltest voor Kinderen. Lisse: Swets & Zeitlinger; 1982.
Vieijra J, König CE, Gardien CJ, de Vries M. PINOK neuropsychologisch onderzoek bij kinderen. Amsterdam: Pearson Test; 1994.
Bakke M, Bergendal B, McAllister A, Sjögreen L, Åsten P. Development and evaluation of a comprehensive screening for orofacial dysfunction. Swed Dent J. 2007;31:75–84.
Darley F, Spriestersbach D. Test for assessing nonverbal oral movement control and sequencing. Diagnostic methods in speech pathology. New York: Harper & Row; 1978.
Schmahmann JD, Gardner R, MacMore J, Vangel MG. Development of a brief ataxia rating scale (BARS) based on a modified form of the ICARS. Movement Disord. 2009;24:1820–8.
Heaton RK, Chelune GJ, Talley JL, Kay GG, Curtis G. Wisconsin Card Sorting Test (WCST) manual: revised and expanded. Odessa: Psychological Assessment Resources; 1993.
Reitan RM. Validity of the trail making test as an indicator of organic brain damage. Percept Motor Skill. 1958;8:271–6.
Golden JC. Stroop Color and Word Test. Chicago: Stoelting Co; 1978.
Wechsler D. Manual for the Wechsler Memory Scale-Revised. New York: The Psychological Corporation; 1987.
Mariën, Mampaey E, Vervaet A, Saerens J, De Deyn PP. Normative data for the Boston Naming Test in native Dutch-speaking Belgian elderly. Brain Lang. 1998;65:447–67.
Graetz P, De Bleser R, Willmes K. De Akense Afasie Test. Lisse: Swets & Zeitlinger; 1992.
Schmahmann JD, Sherman JC. The cerebellar cognitive affective syndrome. Brain. 1998;121:561–79.
Mariën P, Wackenier P, De Surgeloose D, De Deyn PP, Verhoeven J. Developmental coordination disorder: disruption of the cerebello-cerebral network evidenced by SPECT. Cerebellum. 2010;9:405–10.
Pinder GL, Faherty AS. Issues in pediatric feeding and swallowing. In: Caruso AJ, Strand EA, editors. Clinical management of motor speech disorders in children. New York: Thieme Medical Publishers Inc; 1999. p. 281–8.
Wilson EM, Green JR. The development for jaw movement for mastication. Early Hum Dev. 2009;85:303–11.
Tuch BE, Nielson JM. Apraxia of swallowing. Bull Los Angeles Neurol Soc. 1941;6:52–4.
Meadows JC. Dysphagia in unilateral cerebral lesions. JNNP. 1973;36:853–60.
Daniels SK, Brailey K, Foundas AL. Lingual discoordination and dysphagia following acute stroke: analysis of lesion localisation. Dysphagia. 1999;14:85–92.
Daniels SK. Swallowing apraxia: a disorder of the praxis system? Dysphagia. 2000;15:159–66.
Cakmakci H, Pekcevik Y, Yis U, Unalp A, Kurul S. Diagnostic value of proton MR spectroscopy and diffusion-weighted MR imaging in childhood inherited neurometabolic brain diseases and review of the literature. Eur J Radiol. 2010;74:e161–71.
Mariën P, Verhoeven J, Engelborghs S, Rooker S, Pickut BA, De Deyn PP. A role for the cerebellum in motor speech planning? Evidence from a case with the foreign accent syndrome. Clin Neurol Neurosurg. 2006;108:518–22.
Miller NG, Reddick WE, Kocak M, Glass JO, Löbel U, Morris B, et al. Cerebellocerebral diaschisis is the likely mechanism of postsurgical posterior fossa syndrome in pediatric patients with midline cerebellar tumours. ANJR Am J Neuroradiol. 2010;31:288–94.
Mariën P, de Smet E, De Smet HJ, Wackenier P, Dobbeleir A, Verhoeven J. “Apraxic dysgraphia” in a 15-year-old left-handed patient: disruption of the cerebello-cerebral network involved in the planning and execution of graphomotor movements. Cerebellum. 2012. doi:10.1007/s12311-012-0395-1.
Acknowledgments
The authors thank Drs. Miche De Meyer, Patrick Santens, and Peggy Wackenier for their helpful advice and B&B for their continuous support. We also thank an anonymous reviewer for his/her valuable suggestions on an earlier version of the manuscript.
Conflict of Interest
The authors of the manuscript, Peter Mariën, Annelies Vidts, Wim Van Hecke, Didier De Surgeloose, Frank De Belder, Paul M. Parizel, Sebastiaan Engelborghs, Peter P. De Deyn, and Jo Verhoeven, explicitly disclose no conflicts of interests.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Mariën, P., Vidts, A., Van Hecke, W. et al. Mastication Dyspraxia: A Neurodevelopmental Disorder Reflecting Disruption of the Cerebellocerebral Network Involved in Planned Actions. Cerebellum 12, 277–289 (2013). https://doi.org/10.1007/s12311-012-0420-4
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12311-012-0420-4