A 40-year-old male patient complained of diarrhea and poor appetite for 4 months and fatigue and lower limb edema for 3 months.
History of presenting complaint
His earliest symptoms were gastrointestinal: anorexia, nausea, vomiting of green stomach contents, and green-colored diarrhea. Later, symptoms of cardiac dysfunction, such as edema, fatigue and decreased activity, were found. An ultrasonic electrocardiogram at the local hospital suggested that a 25 mm*22 mm space-occupying lesion was found in the posterior lobe of the left atrium near the mitral valve, and repeated examinations showed that the occupation gradually increased to 68 mm*48 mm. Chest computed tomography (CT) showed a right upper lung cavity and a double lung plaque, and sputum culture suggested fungal infection. He was taken to the emergency department of Peking Union Medical College Hospital for multiple system injuries.
Diagnostic symptoms
Parasitic infections: After repeated microscopic examinations of his feces, Opisthorchis sinensis eggs, Heterophyes eggs, and Echinostomatidae were found (Fig. 1). Opisthorchis sinensis can migrate in the body. In cardiac surgery, doctors found a perforation of the mitral valve. Intracardiac vegetation was thought to be related to Aspergillus infection, and parasitic infections facilitated the Aspergillus infection. However, no parasites were found in the limited myocardial tissue with histopathological examinations. Opisthorchis sinensis infection in the brain may cause convulsions and paralysis [5]. The brain MRI of the patient showed chronic infarction. Brain magnetic resonance imaging (MRI) of the patient showed chronic infarction, which may be the result of parasite immune evasion [6]. Because the damaged brain areas were functionally active areas, no biopsy could be undertaken to verify whether there were parasites. Bipolar disorder: The patient was talkative, agitative, grandiose, and overactive. He had a possible episode of depression after his father passed away and two manic episodes, which were diagnosed by local psychiatrists, with symptoms including elation, talkativeness, feeling energetic, reduced need for sleep, increased activity, dangerous driving, and risky dietary behavior during 2009 and 2013. A timeline of the history is shown in graph 4.
Differential diagnostic symptoms
Gastrointestinal symptoms suggested parasitic infections. Talkativeness, grandiosity and overactive symptoms suggested bipolar disorder. Multiorganic injuries, poor cognition and persistent behaviors might not be interpreted as bipolar disorders. Dementia: The patient had poor cognition, confabulation and euphoria, which could be interpreted as chronic encephalopathy. The patient had diarrhea and seizures for 4 months, so the clinically reasonable deduction was that his encephalopathy had developed in the last several months. However, he had been eating raw food with a parasitic infection risk for 8 years, so his changes in eating behavior could not be explained by dementia. Delirium: Delirium is defined as nonspecific acute encephalopathy syndrome with consciousness, attention, thinking, memory, mental motor behavior, and sleep cycle disorder [7]. Delirium is usually transient, lasting for a maximum of 4 weeks and persisting for 6 months in only a few patients. This patient had severe and complex physical diseases, cardiac surgery, and mental disorders, which made the patient prone to delirium. However, after his physical condition stabilized during follow-up, the patient still had poor cognition and euphoria. This is better explained by chronic encephalopathy rather than delirium. However, the agitation described during the first consultation may have partly been caused by delirium. Klüver-Bucy syndrome: Heinrich Klüver and Paul Bucy described a dramatic behavioral syndrome that includes hyperorality, placidity, hypermetamorphosis, dietary changes, altered sexual behavior, and visual agnosia, in monkeys after bilateral temporal lobectomy in 1937. It is now thought to be caused by disturbances of the temporal portions of the limbic networks that interface with multiple cortical and subcortical circuits to modulate emotional behavior and affect [8]. Subcortical infarction and inflammation of the brain might cause Klüver-Bucy syndrome, which might support episodes of docility but not hyperactivity. Schizoaffective disorder: The patient had delusions of grandeur and affective symptoms; however, no sufficient symptoms met the diagnostic criteria for schizophrenia.
Complications
Aspergillus was found in the patient’s blood. His eosinophil and leukocyte counts as well as his C-reactive protein and blood creatinine levels were also abnormal (Fig. 2). The patient had three episodes of seizures during hospitalization. A central nervous system (CNS) MRI was taken before the surgery and 2 months thereafter (Fig. 3). Subcortical infarctions were found in the bilateral centrum semiovale and the left ventricular posterior horn. His left cerebellar hemisphere showed chronic infarction. Ischemic changes of white matter in the bilateral frontal and parietal lobe were detected, and abnormal signals in ventriculus lateralis cerebri could be found.
Causes/etiology
A timeline of the history is shown in Fig. 4. Risky dietary behavior started during a manic episode and persisted even after affective symptoms were under control. Risky dietary behavior might have caused parasitic infection, which led to multiple organic injuries.
Past psychiatric history
The medical history was obtained from the patient’s mother and his sister. His father died in 1994. The patient was depressed for several months, nontalkative, sleepless, and had no appetite. Nevertheless, he recovered spontaneously. During 2009, he suddenly developed a high level of energy, required very little sleep, and undertook risky and dangerous behavior, including in his manner of driving. After a brief period, he abruptly became depressed. “Depression of manic-depressive disorder” was diagnosed by his local psychiatrists, but details of his psychopharmacologic treatment could not be provided by his mother. He stopped his prescribed medication when his symptoms were relieved. His dietary behavior changed permanently since 2009. He continued to eat raw ox-gall, snake gall, frog, and tadpole. During 2013, similar emotional symptoms reoccurred. After a few days of hyperactivity, he suddenly became depressed and very dependent on his mother’s company. He ceased working and stayed in his seaside house alone (Fig. 4).
Past medical and surgical history
The patient suffered from diabetes mellitus for 10 years. He had no surgical history.
Family and social history
The patient did not have a family history of psychiatric disorders.
Personal and forensic history
The patient came from Shandong Province in China. He was unmarried and living alone beside the seashore. He was a civil servant. He had stayed at home on leave for 8 years because of bipolar disorder.
Mental status examination
The patient had full orientation. He was euphoric, talkative, charismatic, and grandiose. He was agitated when he was interrupted. Sometimes he suddenly became angry and refused to continue the conversation. He had no insight into his disease. The total score on the MMSE (mini-mental state examination) was 24; he made errors in execution, reading, and writing. His appetite was unusually high. He frequently expressed hunger, and he ate voraciously. He expressed sexual interest, repetitively saying “I want to marry all the nurses in the hospital.”
Follow up/reviews
After treatment with Debagin 500 mg per day, olanzapine 5 mg per day and praziquantel 210 mg/kg three times a day for 3 days, the patient’s mood improved, and his physical condition was restored. However, he was still talkative and still showed eagerness for sex and raw food. Three months later, his sister said that the patient had stopped all medication since leaving the hospital. He showed overeating, and his body weight increased. He became quiet unless spoken to, but there were no significant changes in his brain MRI (graph 2). Six months later, he was still eager to eat raw food with soy sauce, but his family no longer allowed him to eat tadpoles. He became quiet but still demonstrated confabulation and grandiose symptoms. At the one-year follow-up, he still demonstrated poor cognition and confabulation. His sister said that the patient was quiet at home. He was still fond of barbecue and believed that raw food with soy sauce was delicious. However, his family restricted him from raw food, such as tadpoles and frogs.
