LAUSR

To the Editor: A 13-year-old Iraqi male youngster, with a positive maternal history of bipolarity, was admitted to our inpatient facility for escalating aggression and socially flouting behaviors. He had a 5-day history of decreased need for sleep (2–3 hours/night); hyperactivity with pacing and roaming; verbosity with a coprophrasia; oversexualized behaviors trying to disrobe in public; and extreme mood swings. He began to endorse grandiose delusions of divine revelations and persecutory delusions against his classmates. Work-up for firstepisode psychosis was performed, including toxic screen, neuroimaging, and electroencephalogram but was unrevealing. He had no history of previous depressive episodes. He has been always described to be wellmannered with an outstanding academic achievement. Apart from final examinations, there seemed no current psychosocial stressors. He was diagnosed with bipolar I disorder with Young Mania Rating Scale of 35. Lithium, titrated up to 1000 mg/d (on 3 divided doses), was accordingly prescribed with tangible therapeutic response at a serum level of 1.1 mmol/L. Renal functions, thyroid functions, S. Ca, and Electrocardiograph were all within normal. After 4 weeks of treatment, he was noticed to develop a painless dystonic syndrome with truncal rigidity and flexion to the right, especially while walking, with frequent falls, that disappears when lying down. He was indifferent to the posture. Neuroconsult was summoned. Conversion and catatonia were ruled out on clinical grounds. A diagnosis of lithium-induced Ekbom syndrome was entertained. Naranjo Adverse Drug Reaction Probability Scale scored 8 (“Probable”). Quetiapine XR was introduced at 300-mg nocte, uptitrated to 800 mg/d over 4 days. Lithium was gradually tapered down at 200 mg every 2 days. After 2 weeks, axial dystonia totally disappeared. Manic symptoms were well controlled on quetiapine with great tolerability apart from gaining 2 kgs. Sixteen weeks have elapsed at time of writing this report and patient is in remission (Young Mania Rating Scale , 12) with no EPS. A viva voce parents’ consent was obtained beforehand to report this case anonymously. Ekbom syndrome1 or pleurothotonus describes lateral flexion of the trunk (. 100) due to spasm in paraspinal musculature. Pisa syndrome (PS) is a truncal dystonia characterized by twisting and bending of upper thorax, with involuntary flexion of the neck and head, to one side (commonly right side) relieved by passive mobilization or supine position. Tilting symptoms occurring bilaterally may be labeled as “metronome PS.” It may be seen in extrapyramidal disorders (PD, multiple system atrophy) or as a rare extrapyramidal side effect by neuroleptics or Acetylcholine esterase inhibitors-is in Alzheimer’s disease with an incidence of 0.04%. Hence, some form of cholinergic-dopaminergic imbalance and/or serotonergic-noradrenergic dysfunction would seem to be involved in pathogenesis. Clinically, 2 forms of PS are recognized, that is, acute and tardive dystonia. Risk factors2 have been identified and include old age, female gender, long-standing antipsychotic treatment, chronic psychosis, polypharmacy, and organicity. Discontinuation of culprit agent or dose reduction is typically recommended. Anticholinergic drugs are generally effective in circa 40% of cases. Physiotherapy with postural retaining is crucial for recovery.3 Mechanistically, lithium blocks Dopamine.4 Postsynaptic activation of dopamine receptors is mediated by G-protein–coupled receptors or G-protein coupled receptor. Chronic administration of lithium can alter the functionality of G-protein coupled receptor subunits. Because of this, dopaminergic neurotransmission is inhibited. Moreover, lithium increases GABA levels in cerebrospinal fluid. At the presynaptic level, lithium facilitates GABA release. At the postsynaptic level, it upregulates GABA-B receptors. Boosting GABAergic neurotransmission would further block Dopamine tone. Likewise extrapyramidal side effects have invariably been reported with lithium therapy.5 Lithium is generally underutilized in CAP population.6 It is FDA-approved for bipolar mood disorder aged 12. A recent randomized controlled trial attested to efficacy and safety for ages as young as 7 years. That said, atypical antipsychotic has a bigger effect size for juvenile bipolar disorder.7 American Journal of Therapeutics 0, 1–2 (2020)