Mitsuda psychosis and holodysphrenia revisited: An atypical psychosis in a patient with parieto-occipital paroxysmal electroencephalographic activity and high unconjugated bilirubin.

Abstract

Recent works have found a positive correlation between bilirubin concentration and schizophrenia symptoms.1-3 It seems that unconjugated bilirubin (UCB), through its neurotoxic effect,4 may be particularly interesting as a biomarker candidate in the distinction between schizophrenia and bipolar disorder.5, 6 We present a patient with atypical psychosis, high UCB levels, and electroencephalographic (EEG) changes. A 47-year-old woman was seen in a psychiatric emergency room following 3 days of mutism, anorexia, and insomnia. Over the last month there had also been mental confusion, ruinous persecutory delusion, and auditory hallucinations (two voices commenting on her behavior). We found no changes either in the patient's mood or memory. One year previously, her mother, suffering from an undisclosed mental disorder, had committed suicide. At examination, the patient presented a slightly reddish jaundice. Laboratory tests revealed high levels of UCB (1.57 mg/dL) with hemoglobinuria (0.50 mg/dL). EEG revealed frequent right parieto-occipital paroxysmal activity, but brain computed tomography and lumbar puncture were negative. The patient was diagnosed with psychotic disorder due to medical condition and treated with sodium valproate 1500 mg and olanzapine 10 mg. Full remission of symptoms was achieved and the patient was referred for further study. At internal medicine consultation, no hepatic pathology was found and she was diagnosed with self-limited hemolysis of undisclosed origin, with UCB level rapidly returning to normal values. At the neurology consultation, a new EEG found no abnormal activity at all. At the psychiatry community outpatient clinic, there was no clinical recurrence. After 12 months of follow-up, there were no neuropsychiatric signs or symptoms, so the patient was sent back to the general practitioner with neither antipsychotic nor antiepileptic medication. This clinical vignette suggests classic holodysphrenia, an endogenous group of psychoses with relapsing/remitting course and profound disintegration of mental activity during episodes,7 corresponding nowadays to the DSM-5's brief psychotic disorder or schizophreniform disorder. Some holodysphrenia cases were related to quite understudied medical conditions, thus overlapping Mitsuda's (atypical) psychoses,8 somewhere in between the schizophrenia and the epileptic psychosis spectrum.9 This case emphasizes the importance of a high level of clinical suspicion and of careful medical examination of every patient at the psychiatric emergency room. Furthermore, it also highlights the plausible correlation between high UCB levels and psychosis, especially on the schizophrenia spectrum, which shall be the object of further study. The protocol for the research project has been approved by the Ethics Committee of Lisbon Psychiatric Hospital Center and it conforms to the provisions of the Declaration of Helsinki. Informed consent was attained from the patient on publishing this report. The author declares no conflicts of interest.