Resting-state hyperperfusion in the whole brain: A case of malignant catatonia that improved with electric convulsion therapy Shin Kurose a,b,c, ⁎, Akihiro Koreki a , Michitaka Funayama b , Eriko Takahashi a , Masataka Kaji a , Kamiyu Ogyu a,c , Shotaro Takasu a,c , Teruki Koizumi a,c , Hisaomi Suzuki a , Mitsumoto Onaya a , Masaru Mimura c a Department of Psychiatry, National Hospital Organization Shimofusa Psychiatric Medical Center, Chiba, Japan b Department of Neuropsychiatry, Ashikaga Red Cross Hospital, Ashikaga, Japan c Department of Neuropsychiatry, Keio University School of Medicine, Tokyo, Japan Dear Editors, The first detailed description of catatonia dates back to the 19th cen- tury (Kahlbaum, 1874). Since then, catatonia has been recognized as one of the most severe forms among psychiatric conditions. It also causes fatal medical complications in some cases (Funayama et al., 2018; Mann et al., 1986). Therefore, understanding the mechanisms be- hind catatonia, which have not yet been fully elucidated, is extremely crucial. Although there have been several imaging studies that revealed abnormalities in the caudate, orbitofrontal cortex, parietal cortex, premotor cortex and motor cortex in catatonia, these results have not been consistent (Walther et al., 2017). Walther et al. (2017) suggested that this inconsistency was caused by the fact that catatonia includes several subtypes and various degrees of severity, and they also demon- strated an association between increased catatonia severity and higher perfusion in the supplementary motor area (SMA) using arterial spin la- beling (ASL). These studies were limited to cross-sectional ones and targeted only patients with mild catatonia rather than those with malignant catatonia, the severest form of catatonia, which is accompanied by autonomic in- stability and hyperthermia and frequently causes severe medical com- plications (Fink and Tayler, 2003; Mann et al., 1986). To date, however, there had been no neuroimaging study to investigate this con- dition. Therefore, we analyzed a patient with malignant catatonia using ASL in the period of before and after treatment. A 61-year-old man with schizophrenia with an onset at age 20 was admitted to our hospital with a condition of psychosis and catatonic ex- citement, i.e., excessive and purposeless motor activities, impulsivity, frenzy and incoherent speech. At admission, his physical findings, labo- ratory data and head computed tomography (CT) scan were normal. Immediately after being hospitalized, his psychiatric conditions deterio- rated into mutism and stupor. On the second day of his hospital stay, he developed malignant catatonia (Fink and Tayler, 2003), presenting with hyperthermia, tachycardia, muscle rigidity, and high levels of serum creatine kinase (5822 IU/L), which was treated with lorazepam (given orally, 3 mg/day). He had several medical complications, including uri- nary tract infection, deep vein thrombosis, megacystis and megacolon. Although his medical conditions improved with antibiotics and antico- agulant therapy, his malignant catatonia remained. Therefore, we ad- ministered electroconvulsive therapy (ECT). The score on the Bush- Francis Catatonia Rating Scale (69, worst; 0, best) (Bush et al., 1996) was 21 on the day of the first ECT session and decreased to 0 after he re- ceived 10 sessions of ECT. We acquired whole-brain resting state cerebral blood flow (rCBF) measurements using magnetic resonance imaging (MRI) with ASL both before and 61 days after the 10 ECT sessions. ASL is the latest mo- dality of measuring rCBF and provides an absolute measure of rCBF. This case study adhered to the declaration of Helsinki and was approved by the ethics committee of National Hospital Organization Shimofusa Psy- chiatric Medical Center. His family provided written informed consent as a deputy before the first MRI was conducted, and the patient pro- vided written informed consent after treatment (before the second MRI) was conducted. Images were acquired using a 1.5 T MR system (SIGNA Explorer, GE Medical Systems) and a 12-channel headcoil. ASL was performed using a three-dimensional spiral fast spin-echo sequence with background suppression for perfusion imaging, which covered the entire brain. The ASL sequence consisted of a three-dimensional, multi-delay pseudo-continuous ASL (PCASL), with a fast spin-echo acquisition with background suppression. The labeling plane was set at the base of the brain without information from MR angiography. The imaging protocol was as follows: repetition time (TR) = 4841 ms, echo time (TE) = 10.5 ms, locations = 36, FOV = 24 × 24 cm, voxel size= 4.08 × 4.08 × 4 mm 3 , post-labeling delay (PLD) = 2.025 s, labeling duration = 1.5 s, number of excitations (NEX) = 2, acquisition time = 2 min 35 s. MRI with ASL revealed a remarkable increase in the rCBF in the whole brain, especially in the bilateral SMA, the bilateral anterior cingu- late cortex (ACC), the left insular cortex, the left amygdala, the hypo- thalamus and the left striatum (Fig. 1). This abnormal increase in rCBF was improved after completion of 10 sessions of ECT, and the symptoms of malignant catatonia remitted (Fig. 1). Our results revealed that hyperperfusion was observed not only in the SMA, which was previously reported in mild catatonia (Walther et al., 2017), but also in the whole brain, particularly the ACC, insula, amygdala, hypothalamus, and striatum. The ACC, insula, amygdala, and hypothalamus are core components of the central autonomic net- work (CAN), which plays a prominent role in regulating the autonomic Schizophrenia Research xxx (xxxx) xxx ⁎ Corresponding author at: Department of Neuropsychiatry, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-0016, Japan. E-mail address: s.kurose.0513@keio.jp (S. Kurose). SCHRES-08341; No of Pages 2 Contents lists available at ScienceDirect Schizophrenia Research journal homepage: www.elsevier.com/locate/schres Please cite this article as: S. Kurose, A. Koreki, M. Funayama, et al., Resting-state hyperperfusion in the whole brain: A case of malignant catatonia that improved with el..., Schizophrenia Research, https://doi.org/10.1016/j.schres.2019.05.042 https://doi.org/10.1016/j.schres.2019.05.042 0920-9964/© 2019 Elsevier B.V. All rights reserved.