Table 2.
Case reports indicating the PWS genetic subtype
| Cases reports with genetic subtype | Genetic subtype | Gender | Age at onset Type of onset | Symptoms | Other information | Outcome | Medication | Diagnosis |
|---|---|---|---|---|---|---|---|---|
| Singh et al. [37] | Del | M | 16 sudden onsets | Acute psychosis, confusion, mood-incongruent delusions, hallucinations, anxiety, motility disturbances, mood swings | Growth hormone treatment | Complete remission of symptoms within a month | Not known |
Cycloid psychosis Bipolar affective disorder |
| mUPD | M | 26, sudden onset | Acute psychosis, mood-incongruent delusions, anxiety, mood swings | Growth hormone treatment | Complete remission of symptoms within a month | Cycloid psychosis, Bipolar affective disorder | ||
| mUPD | F | 19, sudden onset | Acute psychosis, confusion, mood-incongruent delusions, hallucinations, anxiety, mood swings | Growth hormone treatment | Complete remission of symptoms within a month | Cycloid psychosis, Bipolar affective disorder | ||
| mUPD | F | 17, sudden onset | Acute psychosis, confusion, mood-incongruent delusions, hallucinations, anxiety, mood swings | Growth hormone treatment | Complete remission of symptoms within a month | Cycloid psychosis | ||
| mUPD | M | 26, sudden onset | Acute psychosis, mood-incongruent delusions, hallucinations, anxiety, mood swings | Complete remission of symptoms within a month | Cycloid psychosis | |||
| mUPD | F | 18, sudden onset | Acute psychosis, confusion, mood-incongruent delusions, anxiety, ecstasy motility disturbances, mood swings | Growth hormone treatment | Complete remission of symptoms within a month | Cycloid psychosis, Bipolar affective disorder | ||
| mUPD | M | 24, sudden onset | Acute psychosis, confusion, mood-incongruent delusions, hallucinations, anxiety, motility disturbances, mood swings | Growth hormone treatment | Mild auditory hallucinations despite resolution of other symptoms of cycloid psychosis | Cycloid psychosis, Bipolar affective disorder | ||
| mUPD | F | 22, sudden onset | Acute psychosis, confusion, mood-incongruent delusions, hallucinations, anxiety, ecstasy, motility disturbances, mood swings | Growth hormone treatment | Complete remission of symptoms within a month | Cycloid psychosis, Bipolar affective disorder | ||
| Del | F | 34, sudden onset | Acute psychosis, confusion, mood-incongruent delusions, hallucinations, anxiety, ecstasy, motility disturbances, mood swings | Complete remission of symptoms within a month | Cycloid psychosis, Bipolar affective disorder | |||
| Del | F | 31, sudden onset | Acute psychosis, confusion, mood-incongruent delusions, hallucinations, anxiety, motility disturbances, mood swings | Complete remission of symptoms within a month | Cycloid psychosis | |||
| Del | F | 31, sudden onset | Acute psychosis, confusion, mood-incongruent delusions, hallucinations, anxiety, motility disturbances, mood swings | Complete remission of symptoms within a month | Cycloid psychosis | |||
| Briegel [38] | mUPD | F | 16, sudden | disorganized speech, reduced eye contact, signs of hallucinations, paranoid delusions, Aggressive behaviour, Increased agitation, refuse to eat | Bed wetting | Improvement 90% after 54 days of treatment |
Aripiprazole Lorazepam |
brief psychotic disorder |
| Kuppens et al. [39] | mUPD | M | Before 19 (recuurent episode at 19) | recocered | Valproic acid | |||
| Poser et al. [40] | mUPD | F |
25, within a month, 2 hospitalizations at 15 and 16 yo for depression with catatonia that responded to lorazepam and haloperidol |
Manic behaviour increasing over a month: hyperverbal, euphoric, illogical, decreased sleep Then mute and withdrawn with catatonia |
10 pounds weight loss in 10 days |
Minimal improvement with pharmacological therapy Improvement after ECT and full recovery after 6 months |
Aripiprazole, Lamotrigine Lithium, Lorazepam, Zisprasidone 8 electroconvulsive therapy |
|
| Sinnema et al. [41] | mUPD | F | 17 | optic and acoustic hallucinations, delusions, aggression, motor agitation, increase of obsessive behaviour, increase of food obsession | bipolar | Stayed hospitalised 2 years, and had psychotic episodes all her further life | Anti-psychotic, Mood stabilizing agents, antidepressants | |
| Chiou and Tsai [42] | mUPD | M | 15 | Reference delusion, Physical aggression | Skin picking, ADHD | Great improvement after treatment | Risperidone 0.5 mg | |
| Del | M | 15, acute, medication-induced |
Decreased need for sleep Physical violence |
Autistic spectrum symptoms ADHD |
carbamazepine | Medication-induced manic episode | ||
| Hergüner and Mukaddes [43] | mUPD | F | 13, acute medication induced | Auditory and visual hallucination, paranoid delusions, social withdrawal, hypoactivity, echolalia, excessive sleep and decreased appetite | Symptoms completely resolved after discontinuation of fluoxetine | Medication induced psychotic symptoms | ||
| Verhoeven et al. [35], Verhoeven et al. [33], Verhoeven et al. [20] | mUPD | M |
14 Within 2 weeks |
Probable hallucinatory experiences, mood instability Increase in obsessional features |
Stabilization of behaviour Marked effect of treatment |
Thioridazine 200 mg with little effect, Lithium added | Cycloid psychosis | |
| mUPD | M | Within 2 weeks |
Emotional turmoil, mood swings, anxieties Confusion |
Marked and long-lasting effect of treatment. No occurrence of major behavioural problems over of a period of more than 2 years | Valproic acide 1500 mg | Cycloid psychosis but originaly diagnosed as bipolar affective disorder | ||
| mUDP | M | Within 2 weeks |
Emotional turmoil, mood swings, anxieties, confusion Paranoid ideations |
Complete disappearance of psychotic symptoms within 6 weeks following start of lithium Stabilization and no new episode until the end of follow-up period (1 year) |
Lithium | Cycloid psychosis | ||
| Del | M | Within 2 weeks |
Possible Perceptual disturbances, paranoid ideations, confusion Emotional turmoil, mood swings, anxieties Obsessive rituals |
Moderate stabilization over a period of 12 months until the end of follow-up |
Lithium Valproic acid carbamazepine |
Cycloid psychosis | ||
| mUPD | F | Subacute onset |
Hallucinations; perceptual disturbances, Paranoid ideation Emotional turmoil; Mood swings, anxieties; confusion |
Marked improvement |
Lithium Valproic acid |
Cycloid psychosis | ||
| mUPD | F | Subacute onset | Hallucinations; paranoid ideation, emotional turmoil; Mood swings, anxieties; confusion Hyperactivity, Increased obsessive rituals | Marked improvement | Valproic acid | Cycloid psychosis | ||
| mUPD | F | Subacute onset | Paranoid ideation, emotional turmoil; Mood swings, anxieties; confusion hyperactivity, increased obsessive rituals | hypothyroidism | Moderate improvement |
Lithium Valproic acid |
Cycloid psychosis | |
| mUPD | M | Subacute onset | Hallucinations, confusion, mood swings, increased obsessive rituals, paranoid ideation | Not evaluated | Valproic acid | Cycloid psychosis | ||
| mUPD | F | Subacute onset |
Emotional turmoil; Mood swings Paranoid ideation Confusion Anxieties; Hyperactivity, Increased obsessive rituals |
Marked improvement |
Lithium Valproic acid |
Previous diagnosis: depression Last: cycloid psychosis |
||
| mUPD | M | Subacute onset | Hallucinations, Probable perceptual disturbances and emotional turmoil, confusion, mood swings, anxieties, hyperactivity, increased obsessive rituals | Total recovery | Pipanperone |
Previous diagnosis: delirious state Last: cycloid psychosis |
||
| Boer et al. [18] | Del | F | 21 | Antipsychotic, Mood stabiliser, antidepressant | paranoid delusion | |||
| mUPD | M | 28 |
Antipsychotic, Mood stabiliser antidepressant |
Paranoid delusion and hallucinations | ||||
| mUPD | F | 29 | Antipsychotic | Paranoid delusion and hallucinations | ||||
| mUPD | F | 22 | Antidepressant | Paranoid delusion and hallucinations | ||||
| mUPD | F | 36 | Antipsychotic, Mood stabiliser, Antidepressant | Paranoid delusion and hallucinations | ||||
| mUPD | F | 29 | Antidepressant | Paranoid delusion and hallucinations | ||||
| Vogels et al. [44], Vogels et al. [19] | ICD | M | 15, Acute | |||||
| mUPD | M | 13, Acute | ||||||
| mUPD | F | 19, Abrupt | ||||||
| mUPD | M | 13, Acute | ||||||
| mUPD | F | 16, Acute |
Anxiety, agitation, restlessness, overactivity, confusion Aggressive behaviour, refused food and drinks Delusion |
One of the episodes was preceded by fever and diarrhoeas during several weeks | After treatment with thioridazine, several mild psychotic episodes during the 13 next years. Then recurrent episodes again | Haloperidol, orphanedrine Thioridazine, Sodium valproate, risperidone, fluvoxamine | ||
| mUPD | M | 19, Abrupt | ||||||
| Beardsmore et al. [16] | mUPD | F | 11, sudden | Food and drinks refusal, bedridden, refused human approach, Self-neglect | Relapsing episodes, Remission between episodes | Risperidone | Cycloid psychosis | |
| mUPD | M | 13, sudden | Relapsing episodes, Remission between episodes | Risperidone | Cycloid psychosis | |||
| Del | M | 12, Not sudden | Agitation Mild anxiety, delusions, Hallucination | Remission with some residual symptoms | Risperidone | Transient psychotic disorder | ||
| Del | M | 14, Not sudden | Agitation, mild anxiety, delusions, hallucination | Remission with some residual symptoms | Risperidone | Transient psychotic disorder | ||
| Beardsmore et al. [16] | mUPD | F | < 33 | Bipolar affective disorder with psychotic symptoms | ||||
| Del | F | 23 | Depressive episode with psychotic symptoms | |||||
| Del | M | 33 | Depressive episode with psychotic symptoms | |||||
| Clarke [17] | Del | F | 20 | Delusion, paranoid ideation, unresponsive, withdrawn | thioridazine | Paranoid schizophrenia | ||
| Trl | F | 14 | Disorientated, disordered speech, withdrawn, paranoid delusions | Several episodes. Good recovery between first episodes. Mental state stabilized following treatment with risperidone and carbamazepine |
Trifluoperazine, Sulpiride, Risperidone Carbamazepine |
Previous diagnosis: acute confusional state Cycloid psychosis | ||
| mUPD | M |
Aggression Delusion |
Level of functioning has declined after illness | Chlorpromazine, Procyclidine Trifluoperazine | ||||
| Del | M | 12 |
Delusion, agitation, disturbed sleep, anxiety, apathy Refuse to eat and drink |
Several psychotic episodes. Improvement at age 22 after treatment with haloperidol, sulpiride and fluoxetine | haloperidol, sulpiride and fluoxetine | Cycloid psychosis | ||
| Whittaker et al. [13] | mUPD | F | 17 |
Querulous, tearful, anxious, withdrawn, paranoid thoughts Disrupted time orientation, sleep, appetite and concentration |
||||
| Watanabe et al. [45] | Del | F | 19 | anorexia, insomnia, guilt feelings, ideas of being doomed, ostracized and persecuted, and stupor alternating with agitation | Near-monthly rhythm, followed by a spontaneous remission in 7–18 days |
Recurrent brief episodes Monthly rhythm |
||
| Ewald et al. [12] | Del | F | 2 episodes of 3–4 weeks | Persecutory delusions, auditory hallucination, anxiety, psychomotor excitement | ||||
| Clarke [11] | Del | M | 17, acute | Agitation, incoherent speech, labile emotions, hallucination (auditory and visual) | Completely resolved after 10 days. No recurrence after 3 years, even after chlorpromazine was discontinued | 200 mg chlorpromazine | ||
| Del | F | 24, acute | Agitation, anxiety, unable to sleep, refuse to eat, withdrawn | Temper outburst worst perimenstrually | Good recovery after 4 weeks but remain suspicious | Chlorpromazine, then dydrogesterine and fluphenazine | ||
| Del | F | acute |
Anxious, restless Delusion, auditory hallucinations |
discharged after 2 weeks but still abnormal beliefs. No return to premorbid level of functioning | thioridazine | |||
| Bhate et al. [46] | Del | F |
Restless both day and night, sleeping for only 1 or 2 h Episodes of psychosis with hallucinations and delusions |
Frequent visit to the toilet, but incontinent |