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

psychiatric disorder. After admission, an initial treatment of oral haloperidol (1 mg/day) was administered, rather than second-generation antipsychotics (which are associated with tendency of metabolic syndrome and increased risk of stroke), along with a physical work-up to test for possible organic etiology. The auditory hallucination was still noted and haloperidol was increased to 5 mg/day. A brain magnetic resonance imaging (MRI) scan revealed no evidence of an interval change or new intracranial lesions, compared with MRI scans performed 2 years prior. Functional MRI (fMRI) was not performed, so we failed to specify the relation between clinical hallucinations and fMRI modifications. The complete blood cells, electrolyte levels, and renal and liver function were normal. The antiphospholipid, lupus anticoagulant, IgG immunoglobulin, and anti-nuclearand anti-double-stranded DNA autoantibodies were also all within the normal range. During her hospital stay, the patient’s psychotic symptoms gradually improved on haloperidol (5 mg/day); for progressive extrapyramidal symptoms, haloperidol was replaced with chlorpromazine (25 mg/day) after discharge a few weeks later, with a fair response. No exacerbation of the psychosis was noted during follow-ups over the next 3 years at our clinic. In our patient, idiopathic Sneddon syndrome was diagnosed. She had deafness and MRI showed multiple cerebral infarctions. The livedo reticularis was noted over her four limbs and the skin biopsy revealed thrombosis of subcutaneous arterioles. Systemic lupus erythematosus, polyarteritis nodosa, cryoglobulinemia, livedoid vasculitis, cold agglutinin disease, arteriosclerosis, and others were all excluded. During this hospitalization, her autoimmune factors were normal and Sneddon syndrome was still considered. Her psychotic symptoms were fully remitted under a low dose of antipsychotics (haloperidol at 5 mg/day in the ward, and subsequent chlorpromazine at 25 mg/day in the clinic), without recurrence of psychosis over the following 3 years. There may be some debate about whether the patient’s symptoms indicated a comorbid primary psychosis or were secondary to Sneddon syndrome. However, the evidence suggests that this is not a comorbid primary psychosis, because of the absence of a family history of psychiatric disorder, the relatively late onset of the psychotic symptoms, and a lack of concomitant delusions. We hypothesize that the psychotic symptoms were secondary to Sneddon syndrome. Suicide risk should be considered in cases where Sneddon syndrome is the diagnosis; a low dose of antipsychotics may be effective for controlling the symptoms and preventing side-effects. The protocol for the research project has been approved by TMU-JIRB (Taipei Medical University-Joint Institutional Review Board, No. N201604035, protocol version/date: version 1.0/2016/04/05, informed consent forms: waiver of informed consent), and it conforms to the provisions of the Declaration of Helsinki.