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Brain Perfusion Asymmetry in Patients with Oral Somatic Delusions

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Brain Perfusion Asymmetry in Patients with Oral Somatic Delusions Eur Arch Psychiatry Clin Neurosci (2013) 263:315–323 DOI 10.1007/s00406-013-0390-7 ORIGINAL PAPER Brain perfusion asymmetry in patients with oral somatic delusions Yojiro Umezaki • Ayano Katagiri • Motoko Watanabe • Miho Takenoshita • Tomomi Sakuma • Emi Sako • Yusuke Sato • Akira Toriihara • Akihito Uezato • Hitoshi Shibuya • Toru Nishikawa • Haruhiko Motomura • Akira Toyofuku Received: 19 August 2012 / Accepted: 15 January 2013 / Published online: 29 January 2013 Ó The Author(s) 2013. This article is published with open access at Springerlink.com Abstract Oral cenesthopathy is a somatic delusion or brain among the patients. The quantitative analysis con- hallucination involving the oral area and is categorized as a firmed that the regional cerebral blood flow values on the delusional disorder, somatic type. The pathophysiology of right side were significantly larger than those on the left this intractable condition remains obscure. In this study, we side for most areas of the brain in the patients. A com- clarified the pathophysiology of oral cenesthopathy by parison of the R/(R ? L) ratios in both groups confirmed evaluating regional brain perfusion. We performed single the significant brain perfusion asymmetry between the two photon emission computed tomography (SPECT) using sides in the callosomarginal, precentral, and temporal 99mTc-ethylcysteinate dimer in 16 subjects (cenesthopa- regions in the patients. Qualitative evaluation of the thy:control = 8:8). The SPECT images were visually SPECT images revealed right [ left brain perfusion assessed qualitatively, and quantitative analyses were also asymmetry in broad regions of the brain. Moreover, the performed using a three-dimensional stereotactic region- quantitative analyses confirmed the perfusion asymmetry of-interest template. The visual assessment revealed a between the two sides in the frontal and temporal areas. right [ left perfusion asymmetry in broad areas of the Those may provide the key for elucidation of the patho- physiology of oral cenesthopathy. Y. Umezaki (&) Á A. Katagiri Á M. Watanabe Á H. Motomura Á Keywords Oral cenesthopathy Á Delusional disorder A. Toyofuku somatic type Á SPECT Á Brain perfusion asymmetry Psychosomatic Dentistry, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8549, Japan e-mail: [email protected] Introduction M. Takenoshita Á T. Sakuma Á E. Sako Psychosomatic Dentistry Clinic, Dental Hospital, Tokyo Medical Oral cenesthopathy is a somatic delusion or hallucination and Dental University, Tokyo, Japan involving the oral area and is categorized as a delusional disorder, somatic type (DDST). Patients complain of unu- Y. Sato sual sensations without corresponding abnormal findings in Complete Denture Prosthodontics, Graduate School of Medical and Dental Sciences, Tokyo Medical and Dental University, the oral area, such as excessive mucus secretion, a slimy Tokyo, Japan sensation in the mouth, or a feeling of coils or wires being present within the mouth. A. Toriihara Á H. Shibuya Because of their firm conviction that their annoying Department of Diagnostic Radiology and Oncology, Graduate School of Medical and Dental Sciences, symptoms have a somatic base, patients often visit dental Tokyo Medical and Dental University, Tokyo, Japan clinics, without consulting a psychiatrist. Most of these patients are reluctant to see a psychiatrist, even if they are A. Uezato Á T. Nishikawa recommended to consult one. To make matters worse, the Department of Psychiatry and Behavioral Sciences, Graduate School of Medical and Dental Sciences, Tokyo Medical and symptoms in most cases are drug-resistant, resulting in Dental University, Tokyo, Japan dentist shopping. 123 316 Eur Arch Psychiatry Clin Neurosci (2013) 263:315–323 Previous case reports examining the regional cerebral duration of the symptoms ranged from 10 to 72 months blood flow (rCBF) of patients with DDST have demon- (29.8 ± 20.4 months). All patients were right-handed as strated hypoperfusion in the left temporal and left parietal defined by the Edinburgh inventory [26]. lobes [10, 24, 27, 34]; this hypoperfusion was successfully SPECT was performed after obtaining verbal consent reversed by pharmacological treatment or modified elec- from the patients. Written informed consent was obtained troconvulsive therapy (mECT). We also previously repor- from all the patients for this study. ted a case of oral cenesthopathy in which a rightward asymmetry of blood flow in the temporal area of the brain Control subjects disappeared after mECT [33]. Unlike the relatively consistent results described in case Eight age- and sex-matched subjects (75.8 ± 5.8 years, 2 reports, statistical SPECT studies analyzing multiple men and 6 women) without symptoms of oral cenesthop- DDST patients are very few and have reported inconsistent athy were enrolled as the control subjects. The inclusion results. Tateno et al. [32] reported that patients with oral criteria were (1) referral for brain perfusion SPECT cenesthopathy exhibited hyperperfusion in the thalamus imaging for dementia screening, (2) no neurological or and right anterior cingulate, as compared to patients with psychiatric symptoms, (3) HDS-R score [25, and (4) no depression. Another recent study examining DDST patients detectable abnormalities visible on MRI examination of the reported hyperperfusion in the left post-central gyrus and brain, except for a high signal intensity on T2-weighted right paracentral lobule, which are regarded as the primary images that was not significant. All data were collected sensory areas [25]. from the subjects’ medical records. The handedness of the Although some studies have examined the relationship control subjects was not identified. between functional brain images and DDST, most of these This study was conducted with the approval of the studies have been case reports. Moreover, to the best of our Ethical Committee of Tokyo Medical and Dental knowledge, the qualitative and quantitative differences University. between the rCBF in patients with oral cenesthopathy and normal control subjects have never previously been ana- Brain perfusion SPECT lyzed. The purpose of this study was to clarify the patho- physiology of oral cenesthopathy through qualitative and The subjects were requested to remain in a comfortable quantitative evaluations of brain perfusion using 99mTc- supine position with their eyes closed in a quiet room. Data ECD SPECT. acquisition was begun after a bolus injection of 600 MBq of 99mTc-ECD via the right brachial vein. First, the passage from the heart to the brain was monitored using a rectan- Methods gular large-field, dual-head gamma camera (E.CAM Sig- nature; Toshiba, Tokyo, Japan) equipped with low-energy, Patients with oral cenesthopathy high-resolution, parallel-hole collimators. Data acquisition consisted of a sequence of 100 frames at a rate of 1 s/frame The subjects were outpatients of the Department of Psy- using a 128 9 128 matrix. Five minutes after the injection chosomatic Dentistry, Tokyo Medical and Dental Univer- of 99mTc-ECD, SPECT images were obtained using the sity, Tokyo, Japan. Diagnosis of oral cenesthopathy had same gamma camera equipped with fan-beam collimators. been made based on the chief complaints of the subjects of The energy window was set at 140 keV ± 15 %, and 45 experiencing abnormal oral sensations. The patients’ com- step-and-shoot images were obtained throughout 180 plaints were limited to the oral area. All of the patients were degrees of rotation (128 9 128 matrix, 1.72 mm/pixel) able to maintain their social standard of living, although they with an acquisition time of 30 s/step. All the images were were somewhat troubled by bizarre sensations. There were reconstructed using ‘ordered subsets expectation maximi- no other psychiatric symptoms, and a psychiatrist confirmed zation’ (OSEM) and then smoothed three dimensionally the absence of either depression or schizophrenia. No cog- using a Butterworth filter. The reconstructed images were nitive deficits were detected, and the measured revised corrected for gamma ray attenuation using the Chang Hasegawa dementia scale (HDS-R) scores of the patients method. were [25. No abnormalities were observed on magnetic To quantify the rCBF, the Patlak plot method [20, 21] resonance image (MRI) of the brain, except for a high signal was applied to the 99mTc-ECD cerebral blood perfusion intensity on the T2-weighted images that was not significant. SPECT images to measure the mean CBF. Quantitative The demographic data of the patients are listed in flow-mapping images were then obtained from the quali- Table 1. The patients consisted of 2 men and 6 women tative cerebral perfusion SPECT images using the Patlak ranging in age from 66 to 83 years (75.9 ± 6.0 years). The plot graphical analysis and Lassen’s correction [7, 19]. 123 Eur Arch Psychiatry Clin Neurosci (2013) 263:315–323 317 Data analysis was composed mainly of elderly women. The sex and age distributions, shown in this table, were consistent with Visual assessment those in our previous paper [33]. The patients’ chief complaints (for example, sticky saliva or bubbles flowing A physician specialized in the field of nuclear medicine behind their dentures or wires for the mandibular incisors who was unaware of the patient grouping evaluated all the when removing dentures) were strange, bizarre, and hard to qualitative SPECT images of all the 16 study subjects. The empathize with. Moreover, the symptoms were persistent,
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