Intractable yawning associated with mature teratoma of the supramedial cerebellum

Case report

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Yawning occurs in various conditions such as hypoxia, epilepsy, and sleep disorders including sleep apnea. Intractable yawning associated with a brain tumor has been rarely reported. A 19-year-old woman presented with intractable yawning. Magnetic resonance imaging showed a tumor in the supramedial cerebellum that compressed the dorsal side of the midbrain and upper pons. After subtotal removal of the tumor, the yawning completely disappeared. Postoperative MRI showed resolution of compression of the brainstem. The tumor was histologically diagnosed as a mature teratoma. The present case suggested that the intractable yawning resulted from the tumor compressing the dorsal side of the junction between the midbrain and pons.

Abbreviations used in this paper:PBN = parabrachial nucleus; PVN = paraventricular nucleus.

Yawning occurs in various conditions such as hypoxia, epilepsy, and sleep disorders including sleep apnea. Intractable yawning associated with a brain tumor has been rarely reported. A 19-year-old woman presented with intractable yawning. Magnetic resonance imaging showed a tumor in the supramedial cerebellum that compressed the dorsal side of the midbrain and upper pons. After subtotal removal of the tumor, the yawning completely disappeared. Postoperative MRI showed resolution of compression of the brainstem. The tumor was histologically diagnosed as a mature teratoma. The present case suggested that the intractable yawning resulted from the tumor compressing the dorsal side of the junction between the midbrain and pons.

Yawning occurs in various conditions such as hypoxia, epilepsy, and sleep disorders, including sleep apnea. Neural networks among the pons, hypothalamus, limbic system, and autonomic nervous system may be associated with yawning.3,6,8,9 However, intractable yawning associated with a brain tumor has rarely been reported.1

We report a rare case involving a patient who had a mature teratoma in the supramedial cerebellum and suffered from intractable yawning. After subtotal removal of the tumor, the yawning completely disappeared.

Case Report

History and Examination

A 19-year-old woman with recurrent yawning visited a local hospital, and an intracranial mass was detected on MRI. The patient visited our institution 4 weeks after symptom onset. The yawning occurred 20 times per minute and continued for approximately 60–90 minutes. Such yawning attacks occurred 2 or 3 times per week. Neurological examination demonstrated no abnormal findings. Magnetic resonance imaging depicted a high-intensity mass in the supramedial cerebellum on T1- and T2-weighted images (Fig. 1). The tumor compressed the dorsal midbrain and upper pons.

Fig. 1.
Fig. 1.

Axial T1-weighted (A), axial T2-weighted (B), and sagittal Gd-enhanced T1-weighted MR images (C) obtained when the patient presented to our institution, showing a high-intensity mass in the supramedial cerebellum that compresses the dorsal midbrain and the upper pons.

Blood gas analysis findings, including partial oxygen pressure, were within normal limits during yawning. Blood counts and biochemical data were also within normal limits. Although the patient's score was 20/24 on the Epworth Sleepiness Scale for detection of sleep disorders (normal score < 11), polysomnography showed no abnormality. Electroencephalography also demonstrated no abnormality. After improving her daily living environment according to our advice, the patient's Epworth Sleepiness Scale score declined to 9/24. However, the yawning continued for 2 years after the decrease in the Epworth Sleepiness Scale score, and then diplopia developed. Neurological examination demonstrated a right trochlear nerve palsy. Magnetic resonance imaging showed no change in the tumor.

Operation and Postoperative Course

The patient underwent removal of the tumor through an occipital transtentorial approach performed under general anesthesia. The tumor was hard and contained yellowish fat tissue and calcified components. During surgery, we found that the tumor adhered to the dorsal side of the midbrain and upper pons and involved the right trochlear nerve and the right superior cerebellar artery. Therefore, the tumor except the part adhering to the dorsal midbrain was removed. The surgical specimen was histologically diagnosed as a mature teratoma composed of fat, muscle, and nerve cells with poor heteromorphism. The yawning completely disappeared immediately after surgery and has not recurred 7 months after surgery. Postoperative MRI showed a residual tumor at the dorsal midbrain and resolution of the brainstem compression (Fig. 2).

Fig. 2.
Fig. 2.

Postoperative sagittal Gd-enhanced T1-weighted MR image revealing a residual tumor at the dorsal midbrain and resolution of compression of the brainstem.

Discussion

Based on blood gas analysis, the Epworth Sleepiness Scale score, and findings on polysomnography and electroencephalography, intractable yawning in this patient was likely not caused by hypoxia, epilepsy, or a sleep disorder such as sleep apnea. Furthermore, the yawning disappeared immediately after surgery and did not recur after surgery. These findings suggested that the yawning was associated with a tumor in the supramedial cerebellum that compressed the dorsal midbrain and upper pons.

The parabrachial area is located on the dorsal side of the junction of the midbrain and pons and plays a role in unifying the autonomic nervous system.4,5,7 In the human brain, the parabrachial area is a horseshoe-shaped band of gray matter composed of the lateral parabrachial nucleus (PBN), the Kölliker-Fuse nucleus, and the medial PBN (Fig. 3 upper). These nuclei receive important afferent fibers from the cardiovascular, respiratory, and gustatory systems and project efferent fibers toward superior centers.5 On the other hand, the paraventricular nucleus (PVN), which is located in the medial hypothalamus, is a center of the autonomic nervous system and neuroendocrine system, and it is closely associated with yawning (Fig. 3 lower).4,6 An experimental study in rats demonstrated the occurrence of yawning upon electrical or chemical stimulation of the PVN.8 Furthermore, the PVN has reciprocal connections with brainstem autonomic centers including the PBN.2 In particular, the PVN transmits signals mainly from the lateral PBN.7 In the present case, any extraordinary stimulation from the lateral PBN in the brainstem to the PVN in the hypothalamus may have led to the development of pathological yawning.

Fig. 3.
Fig. 3.

Upper: The parabrachial area is composed of the lateral PBN (a), the Kölliker-Fuse nucleus (b), and the medial PBN (c). Lower: The PVN is localized in the medial hypothalamus.

In this case, MRI showed that the tumor preoperatively compressed the dorsal side of the midbrain and upper pons, and this condition was resolved after surgery. These findings suggested that the intractable yawning might have resulted from the tumor compressing the PBN located on the dorsal side of the junction of the midbrain and pons.

Disclosure

The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Author contributions to the study and manuscript preparation include the following. Conception and design: Saura, Beppu. Analysis and interpretation of data: Uesugi, Sasaki. Critically revising the article: all authors. Reviewed submitted version of manuscript: all authors. Approved the final version of the manuscript on behalf of all authors: Saura. Administrative/technical/material support: Matsuura, Asahi.

References

  • 1

    Arai KKita KKomiyama AHirayama KSaeki NNagao K: [Progressive dysautonomia in hemangioblastoma in the region of the fourth ventricle.]. No To Shinkei 38:1952001986. (Jpn)

    • Search Google Scholar
    • Export Citation
  • 2

    Blair MLPiekut DWant AOlschowka JA: Role of the hypothalamic paraventricular nucleus in cardiovascular regulation. Clin Exp Pharmacol Physiol 23:1611651996

    • Search Google Scholar
    • Export Citation
  • 3

    Cronin TG Jr: Yawning: an early manifestation of vasovagal reflex. AJR Am J Roentgenol 150:2091988. (Letter)

  • 4

    Honda KHiguchi T: Role of midbrain parabrachial nucleus in controlling electrical activity of oxytocin and vasopressin secreting neurones in the hypothalamic supraoptic nucleus. J Reprod Dev 47:2592652001

    • Search Google Scholar
    • Export Citation
  • 5

    Kiernan JA: Barr's The Human Nervous System: An Anatomical Viewpoint ed 9BaltimoreLippincott Williams & Wilkins2009. 147148

  • 6

    Kita ISato-Suzuki IOguri MArita H: Yawning responses induced by local hypoxia in the paraventricular nucleus of the rat. Behav Brain Res 117:1191262000

    • Search Google Scholar
    • Export Citation
  • 7

    Naidich TPDuvernoy HMDelman BNSorensen AGKollias SSHaacke EM: Duvernoy's Atlas of the Human Brain Stem and Cerebellum: High-Field MRI: Surface Anatomy Internal Structure Vascularization and 3D Sectional Anatomy New YorkSpringer2009. 324

    • Search Google Scholar
    • Export Citation
  • 8

    Sato-Suzuki IKita IOguri MArita H: Stereotyped yawning responses induced by electrical and chemical stimulation of paraventricular nucleus of the rat. J Neurophysiol 80:276527751998

    • Search Google Scholar
    • Export Citation
  • 9

    Ueyama TSenba E: [Limbic system and autonomic nervous system.]. Rinsho Shinkeigaku 50:100310062010. (Jpn)

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Article Information

Address correspondence to: Hiroaki Saura, M.D., Department of Neurosurgery, Iwate Medical University, 19-1 Uchimaru, Morioka 020-8505, Japan. email: hirosau@gmail.com.

Please include this information when citing this paper: published online May 30, 2014; DOI: 10.3171/2014.4.JNS132243.

© AANS, except where prohibited by US copyright law.

Headings

Figures

  • View in gallery

    Axial T1-weighted (A), axial T2-weighted (B), and sagittal Gd-enhanced T1-weighted MR images (C) obtained when the patient presented to our institution, showing a high-intensity mass in the supramedial cerebellum that compresses the dorsal midbrain and the upper pons.

  • View in gallery

    Postoperative sagittal Gd-enhanced T1-weighted MR image revealing a residual tumor at the dorsal midbrain and resolution of compression of the brainstem.

  • View in gallery

    Upper: The parabrachial area is composed of the lateral PBN (a), the Kölliker-Fuse nucleus (b), and the medial PBN (c). Lower: The PVN is localized in the medial hypothalamus.

References

  • 1

    Arai KKita KKomiyama AHirayama KSaeki NNagao K: [Progressive dysautonomia in hemangioblastoma in the region of the fourth ventricle.]. No To Shinkei 38:1952001986. (Jpn)

    • Search Google Scholar
    • Export Citation
  • 2

    Blair MLPiekut DWant AOlschowka JA: Role of the hypothalamic paraventricular nucleus in cardiovascular regulation. Clin Exp Pharmacol Physiol 23:1611651996

    • Search Google Scholar
    • Export Citation
  • 3

    Cronin TG Jr: Yawning: an early manifestation of vasovagal reflex. AJR Am J Roentgenol 150:2091988. (Letter)

  • 4

    Honda KHiguchi T: Role of midbrain parabrachial nucleus in controlling electrical activity of oxytocin and vasopressin secreting neurones in the hypothalamic supraoptic nucleus. J Reprod Dev 47:2592652001

    • Search Google Scholar
    • Export Citation
  • 5

    Kiernan JA: Barr's The Human Nervous System: An Anatomical Viewpoint ed 9BaltimoreLippincott Williams & Wilkins2009. 147148

  • 6

    Kita ISato-Suzuki IOguri MArita H: Yawning responses induced by local hypoxia in the paraventricular nucleus of the rat. Behav Brain Res 117:1191262000

    • Search Google Scholar
    • Export Citation
  • 7

    Naidich TPDuvernoy HMDelman BNSorensen AGKollias SSHaacke EM: Duvernoy's Atlas of the Human Brain Stem and Cerebellum: High-Field MRI: Surface Anatomy Internal Structure Vascularization and 3D Sectional Anatomy New YorkSpringer2009. 324

    • Search Google Scholar
    • Export Citation
  • 8

    Sato-Suzuki IKita IOguri MArita H: Stereotyped yawning responses induced by electrical and chemical stimulation of paraventricular nucleus of the rat. J Neurophysiol 80:276527751998

    • Search Google Scholar
    • Export Citation
  • 9

    Ueyama TSenba E: [Limbic system and autonomic nervous system.]. Rinsho Shinkeigaku 50:100310062010. (Jpn)

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