Skip to main content

Table 2 Case reports indicating the PWS genetic subtype

From: Psychotic illness in people with Prader–Willi syndrome: a systematic review of clinical presentation, course and phenomenology

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