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Neuropediatrics 2006; 37(3): 115-120
DOI: 10.1055/s-2006-924428
Original Article

Georg Thieme Verlag KG Stuttgart · New York

Dysphagia-Gastroesophageal Reflux Complex: Complications Due to Dysfunction of Solitary Tract Nucleus-Mediated Vago-Vagal Reflex

Y. Saito1 , Y. Kawashima2 , A. Kondo1 , Y. Chikumaru3 , A. Matsui3 , I. Nagata2 , K. Ohno1
  • 1Division of Child Neurology, Institute of Neurological Sciences, Faculty of Medicine, Tottori University, Yonago, Japan
  • 2Division of Pediatrics and Perinatology, Faculty of Medicine, Tottori University, Yonago, Japan
  • 3Department of Pediatrics and Pediatric Neurology, Yokohama Ryo-iku Medical Center, Yokohama, Japan
Further Information

Publication History

Received: November 11, 2005

Accepted after Revision: June 1, 2006

Publication Date:
11 September 2006 (online)

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Abstract

We report on the complication of gastroesophageal reflux (GER) in four patients with lower brainstem dysfunction. These patients suffered from perinatal asphyxia, cerebellar hemorrhage, or congenital dysphagia of unknown origin and showed facial nerve palsy, inspiratory stridor due to vocal cord paralysis, central sleep apnea, and dysphagia, in various combinations. Naso-intestinal tube feeding was introduced in all of the patients due to recurrent vomiting and aspiration pneumonia resulting from GER. T2-weighted magnetic resonance (MR) imaging revealed symmetrical high intensity lesions in the tegmentum of the lower pons and the medulla oblongata in two of the patients, and pontomedullary atrophy in another patient. In normal subjects, lower esophageal sphincter contraction is provoked by distension of the gastric wall, through a vago-vagal reflex. Since this reflex arc involves the solitary tract nucleus, where the swallowing center is located, the association of dysphagia and GER in the present patients is thought to result from the lesions in the tegmentum of medulla oblongata. We propose the term “dysphagia-GER complex” to describe the disturbed motility of the upper digestive tract due to lower brainstem involvement. In children with brainstem lesions, neurological assessment of GER is warranted, in addition to the examination of other signs of brainstem dysfunction, including dysphagia and respiratory disturbance.

Key words

Gastroesophageal reflux - dysphagia - solitary tract nucleus - vagal reflex

References

  • 1 Altschuler S M, Boyle J T, Nixon T E, Pack A I, Cohen S. Simultaneous reflex inhibition of lower esophageal sphincter and crural diaphragm in cats.  Am J Physiol. 1985;  249 G586-G591
    PubMedGoogle Scholar
  • 2 Bavinck J N, Weaver D D. Subclavian artery supply disruption sequence: hypothesis of a vascular etiology for Poland, Klippel-Feil, and Mobius anomalies.  Am J Med Genet. 1986;  23 903-918
    CrossrefPubMedGoogle Scholar
  • 3 Birnkrant D J, Pope J F, Martin J E, Repucci A H, Eiben R M. Treatment of type I spinal muscular atrophy with noninvasive ventilation and gastrostomy feeding.  Pediatr Neurol. 1998;  18 407-410
    CrossrefPubMedGoogle Scholar
  • 4 Bohmer C J, Niezen-de Boer M C, Klinkenberg-Knol E C, Deville W L, Nadorp J H, Meuwissen S G. The prevalence of gastroesophageal reflux disease in institutionalized intellectually disabled individuals.  Am J Gastroenterol. 1999;  94 804-810
    PubMedGoogle Scholar
  • 5 Del Giudice E, Staiano A, Capano G, Romano A, Florimonte L, Miele E, Ciarla C, Campanozzi A, Crisanti A F. Gastrointestinal manifestations in children with cerebral palsy.  Brain Dev. 1999;  21 307-311
    CrossrefPubMedGoogle Scholar
  • 6 Drucker D E, Michna B A, Krummel T M, Hunt P K, Hartenberg M A, Salzberg A M. Transthoracic Nissen fundoplication for gastroesophageal reflux in patients with severe kypho-roto-scoliosis.  J Pediatr Surg. 1989;  24 46-47 47
    PubMedGoogle Scholar
  • 7 Fujita I, Koyanagi T, Kukita J, Yamashita H, Minami T, Nakano H, Ueda K. Moebius syndrome with central hypoventilation and brainstem calcification: a case report.  Eur J Pediatr. 1991;  150 582-583
    PubMedGoogle Scholar
  • 8 Frank Y, Schwartz S B, Epstein N E, Beresford H R. Chronic dysphagia, vomiting and gastroesophageal reflux as manifestations of a brain stem glioma: a case report.  Pediatr Neurosci. 1989;  15 265-268
    PubMedGoogle Scholar
  • 9 Govaert P, Vanhaeserbouck P, De Praeter C, Frankel U, Leroy J. Moebius sequence and prenatal brain ischemia.  Pediatrics. 1989;  84 570-573
    PubMedGoogle Scholar
  • 10 Jean A. Brain stem control of swallowing: neuronal network and cellular mechanisms.  Physiol Rev. 2001;  81 929-969
    PubMedGoogle Scholar
  • 11 Kawahara H, Blackshaw L A, John Dent V N. Transmitter mechanisms in vagal afferent-induced reduction of lower oesophageal sphincter (LOS) pressure in the rat.  J Autonom Nerv Sys. 1994;  49 69-80
    PubMedGoogle Scholar
  • 12 Lin Y C, Ni Y H, Chang M H. Gastroesophageal reflux disease beyond infancy.  Pediatr Int. 2004;  46 516-520
    CrossrefPubMedGoogle Scholar
  • 13 Leech R W, Brumback R A. Massive brain stem necrosis in the human neonate: presentation of three cases with review of the literature.  J Child Neurol. 1988;  3 258-262
    PubMedGoogle Scholar
  • 14 Lundgren O. Vagal control of the motor functions of the lower esophageal sphincter and the stomach.  J Autonom Nerv Sys. 1983;  9 185-197
    PubMedGoogle Scholar
  • 15 Mahony M J, Kennedy J D, Leaf A, Matthew D J, Milla P J. Brain stem glioma presenting as gastroesophageal reflux.  Arch Dis Child. 1987;  62 731-733
    PubMedGoogle Scholar
  • 16 Maisky V A, Datsenko V V, Moibenko A A, Bugaychenko L A, Pilyavskii A I, Kostyukov A I, Kalezic I, Johansson H. NO-generating neurons in the medullary cardiovascular centers of rodents and carnivores.  Comp Biochem Physiol A Mol Integr Physiol. 2003;  136 605-612
    PubMedGoogle Scholar
  • 17 Muto A, Matsui A, Saito Y, Iwamoto H, Kaneko K, Masuko K, Chikumaru Y, Saito K, Kimura S. Laryngeal dystonia in xeroderma pigmentosum.  Brain Dev. 2005;  27 598-601
    CrossrefPubMedGoogle Scholar
  • 18 Myers R E. Four patterns of perinatal brain damage and their contributions of occurrence in primates. Meldrum BS, Marsden CD Advances in Neurology, Vol. 10. New York; Raven Press 1975: 223-234
    Google Scholar
  • 19 Natsume J, Watanabe K, Kuno K, Hayakawa F, Hashizume Y. Clinical, neurophysiologic, and neuropathological features of an infant with brain damage of total asphyxia type (Myers).  Pediatr Neurol. 1995;  13 61-64
    CrossrefPubMedGoogle Scholar
  • 20 Niel J P. Reflex excitation and inhibition of the lower oesophageal sphincter induced by gastric distension in the cat.  J Autonom Nerv Sys. 1986;  16 205-209
    PubMedGoogle Scholar
  • 21 Oglivie A L, Atkinson M. Influence of the vagus nerve upon the reflex control of the lower oesophageal sphincter.  Gut. 1984;  25 253-258
    PubMedGoogle Scholar
  • 22 Patti M G, Gantert W, Way L W. Surgery of the esophagus. Anatomy and physiology.  Surg Clin North Am. 1997;  77 959-970
    CrossrefPubMedGoogle Scholar
  • 23 Pollack I F, Pang D, Kocoshis S, Putnam P. Neurogenic dysphagia resulting from Chiari malformations.  Neurosurgery. 1992;  30 709-719
    PubMedGoogle Scholar
  • 24 Reynolds J C, Ouyang A, Cohen S. A lower esophageal sphincter reflex involving substance P.  Am J Physiol. 1984;  246 G346-G354
    PubMedGoogle Scholar
  • 25 Rogers R C, Hermann G E, Travagli R A. Brainstem pathways responsible for oesophageal control of gastric motility and tone in the rat.  J Physiol. 1999;  514 369-383
    CrossrefPubMedGoogle Scholar
  • 26 Roland E H, Hill A, Norman M G, Flodmark O, MacNab A J. Selective brainstem injury in an asphyxiated newborn.  Ann Neurol. 1988;  23 89-92
    CrossrefPubMedGoogle Scholar
  • 27 Saito Y, Ezure K, Tanaka I. Activities of respiratory and non-respiratory neurons in the nucleus of solitary tract during swallowing in the rat.  J Physiol. 2002;  540 1047-1060
    CrossrefPubMedGoogle Scholar
  • 28 Sarnat H B. Watershed infarcts in the fetal and neonatal brainstem. An aetiology of central hypoventilation, dysphagia, Möbius syndrome and micrognathia.  Eur J Paediatr Neurol. 2004;  8 71-87
    CrossrefPubMedGoogle Scholar
  • 29 Schan C A, Harding S M, Haile J M, Bradley L A, Richter J E. Gastroesophageal reflux-induced bronchoconstriction.  Chest. 1994;  106 731-737
    PubMedGoogle Scholar
  • 30 Shafik A, Shafik I, El-Sibai O, Shafik A A. On the pathogenesis of gastroesophageal reflux: The concept of gastroesophageal dyssynergia.  J Thorac Cardiovasc Surg. 2005;  130 401-407
    CrossrefPubMedGoogle Scholar
  • 31 Shafik A. Recognition of a gastroesophageal reflex in dogs and its role in lower esophageal sphincter competence.  Eur Surg Res. 1998;  30 352-358
    CrossrefPubMedGoogle Scholar
  • 32 Wood J R, Camilleri M, Low P A, Malagelada J-R. Brainstem tumor presenting as an upper gut motility disorder.  Gastroenterology. 1985;  89 1141-1144
    PubMedGoogle Scholar
  • 33 Yasaki E, Saito Y, Nakano K, Katsumori H, Hayashi K, Nishikawa T, Osawa M. Characteristics of breathing patterns in Leigh and its overlap syndromes.  Neuropediatrics. 2001;  32 299-306
    Article in Thieme ConnectPubMedGoogle Scholar

Yoshiaki Saito

Division of Child Neurology
Institute of Neurological Sciences
Faculty of Medicine
Tottori University

36 - 1 Nichi-cho

Yonago 683 - 8504

Japan

Email: saitoyo@grape.med.tottori-u.ac.jp

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