compulsive singing, which we propose as yet another
Compulsive Singing:
Another Aspect of Punding
in Parkinson’s Disease
Christophe Bonvin, MD, Judit Horvath, MD,
Blaise Christe, PhD, Theodor Landis, MD,
and Pierre R. Burkhard, MD
We report on two patients with advanced Parkinson’s disease
who were exhibiting a peculiar and stereotyped behavior
characterized by an irrepressible need to sing compulsively
when under high-dose dopamine replacement therapy. Shar-
ing many features with punding, this singing behavior is pro-
posed as a distinct manifestation of the dopamine dysregula-
tion syndrome in Parkinson’s disease.
Ann Neurol 2007;62:525–528
Chronic dopamine replacement therapy in Parkinson’s
disease (PD) is associated with a variety of motor, non-
motor, and neuropsychiatric complications, the latter
culminating in peculiar, spectacular, and sometimes di-
sastrous behavior disorders. These involve a combina-
tion of addiction, impulse-control disorders, and manic
or psychotic manifestations, now collectively referred to
as the dopamine dysregulation syndrome (DDS).1,2
Sharing core features with drug addiction, DDS im-
plies repeated, unnecessary, and sometimes deleterious
daily intakes of dopaminergic agents at amounts far be-
yond those required by the objective motor impair-
ment, leading to severe dyskinesia, euphoria, aggressiv-
ity, hallucinations, confusion, or frank psychosis. DDS
may also be associated with impulse-control disorders,
such as pathological gambling, compulsive shopping,
bulimia, or hypersexuality, or with complex psychomo-
tor stereotypies involving repeated, automatic, and pur-
poseless behaviors referred to as punding,3,4 a term
originally coined to describe stereotypical behaviors in
psychostimulant addicts.5
In this report, we describe two PD patients who de-
veloped, in the context of increased dopamine replace-
ment therapy, an abnormal behavior characterized by
From the Department of Neurology, Geneva University Hospitals
and Faculty of Medicine, Geneva, Switzerland.
Received Apr 27, 2007, and in revised form Jun 25. Accepted for
publication Jun 29, 2007.
Published online Aug 14, 2007, in Wiley InterScience
(www.interscience.wiley.com). DOI: 10.1002/ana.21202
Address correspondence to Dr Burkhard, Department of Neurology,
Geneva University Hospitals, 1211 Geneva 14, Switzerland.
E-mail: pierre.burkhard@hcuge.ch
distinct feature of DDS-related punding.
Case Reports
Patient 1
Patient 1 is a 70-year-old university professor and am-
ateur piano player who developed typical L-dopa (LD)–
responsive PD more marked on the left side. Brain
magnetic resonance imaging was normal, and the 123I-
ioflupane single-photon emission computed tomogra-
phy scan showed reduced radiotracer uptake in the
right striatum more prominent in the putamen. There
was no evidence of cognitive impairment (Mini-Mental
State Examination score, 30/30). After 6 years of PD
duration and escalating dosages of dopaminergic agents
(cabergoline 4mg/day, LD/carbidopa 800/200mg/day,
and tolcapone 300mg/day), she developed dyskinesia
and unpredictable motor and nonmotor fluctuations.
When “off,” besides severe parkinsonian features, she
experienced generalized pain, depression, anxiety, and
panic attacks. When “on,” she was mobile and had
moderate dyskinesia and occasional visual hallucina-
tions. At the same time, while being treated with 1,268
4
L-dopa equivalent units (LEU), she exhibited a repet-
itive, compulsive behavior characterized by singing
endlessly when “on.” It started with an irrepressible
urge to hum the rhythm and then the main melody of
Francesca di Foix, a jocular opera written in 1831 by
Gaetano Donizetti. She had heard this rarely produced
piece in Milan years ago, and although she did not
particularly like it, she had an obsessive need to repeat
this song again and again for hours. Even though it
was disruptive, preventing sleep and social interactions,
singing was reported as pleasant and associated with a
feeling of calmness and relief. If interrupted, she be-
came irritated. She later developed clear addiction to
LD, leading to disabling dyskinesia, visual hallucina-
tions, and other behavioral symptoms typical of DDS
including hypersexuality, euphoria, low frustration tol-
erance, mood swings, walkabouts, and social with-
drawal. Several attempts to decrease her daily intake of
dopaminergic agents proved unsuccessful, generating
prolonged and unbearable “off” periods. All symptoms
improved minimally after quetiapine (25mg twice
daily) had been introduced.
Patient 2
This otherwise healthy 71-year-old painter was diag-
nosed with PD at age 66, which started with left-sided
bradykinesia and rigidity. He grew up in a family of
musicians and used to spend time listening to classical
music and singing willingly. There was no cognitive
decline (Mini-Mental State Examination score, 30/30).
Over the following 5 years, the disease progressed sig-
nificantly; he complained of fatigue, motor and mental
Bonvin et al: Singing, Punding, and PD 525
slowness, micrographia, and imprecision in his paint-
ings. Brain magnetic resonance imaging showed mini-
mal diffuse atrophy. 123I-ioflupane single-photon emis-
sion computed tomography scan confirmed a severely
reduced radiotracer uptake in the striatum bilaterally
more prominent in the right putamen. LD/benserazide
100/25mg three times a day was introduced with a re-
markable clinical response on motor symptoms. Some-
what paradoxically, within a year, major depressive
symptoms developed with suicidal thoughts, anxiety,
and insomnia, leading to the successive initiation of
various antidepressant drugs. At the same time, LD/
benserazide was increased to 500/125mg daily and
pramipexole (2mg daily) was added. Gradually, he pre-
sented mild motor fluctuations associated with mood
swings. He began eating excessively leading to a weight
gain of 7kg over 2 years. His art production became
repetitive and disorganized. Concomitantly, while be-
ing treated with 634 LEU, he started to hum repeat-
edly the same melody, initially once a week, then sev-
eral times daily, mostly in the evening and only during
“on” periods. Although he asserted singing exclusively
Mozart’s 7th Serenade (“Haffner,” KV 250), his wife
reported also about 10 different poorly elaborated
songs. This stereotyped behavior was reported as irre-
pressible and gave him a sensation of relief and “peace
of mind.” On demand, he could stop singing for short
periods but felt somewhat frustrated, demonstrating
some aggressive behavior toward his spouse. There
were no concomitant auditory or visual hallucinations.
This phenomenon exacerbated dramatically when LD/
benserazide was increased to 1,000/250mg daily (1134
LEU) mostly because of unverified complaints of pro-
longed “off” periods, that is, the patient losing control
over the compulsion and singing almost unendingly all
day. Although he denied it, his spouse reported re-
peated intakes of additional rescue doses of LD, sup-
porting an addictive behavior toward dopaminergic
agents. Eventually, compulsive singing improved, but
did not disappear, when LD/benserazide was reduced
to the minimal daily doses (500/125mg) preventing
the worst “off” periods.
Results and Discussion
In addition to other features of DDS, as detailed in the
Table, both patients developed a peculiar, stereotyped,
and compulsive behavior characterized by an urge to
sing repeatedly the same song, in the setting of ad-
vanced and fluctuating PD and in the absence of sig-
nificant cognitive impairment or prior psychiatric con-
dition. The main precipitating factor appeared to be
related to dopamine replacement therapy, as demon-
strated by the singing compulsion being present when
the patients were “on,” by the worsening of the phe-
nomenon when the dosage of dopaminergic agents was
increased to more than 800 LEU, as previously report-
526 Annals of Neurology Vol 62 No 5 November 2007
ed,4 and conversely, by an improvement when doses
were decreased. In addition, both patients exhibited
clear features of addiction to LD, which was marked in
Patient 1 and more subtle in Patient 2. The respective
role of LD and dopamine agonists in triggering the
compulsive singing is difficult to appreciate because, in
both patients, the phenomenon developed concomi-
tantly with the initiation or an increase of both thera-
peutic agents.
The phenomenon herein described shares a number
of features with what has been previously reported un-
der the term punding, a stereotyped, repetitive, pur-
poseless, and disruptive behavior performed during
hours, if not days, and accompanied with a form of
fascination related to the ongoing activities. Just as in
our patients, punding is associated with a feeling of
relief when produced and of frustration when pre-
vented. It has been proposed that punding-associated
compulsive thoughts and activities may stem in pre-
morbid occupation or hobbies and remain selective for
one or a few types of behaviors over time.3 Our pa-
tients support this hypothesis because, although neither
was professional singer or musician, both have been ex-
tensively exposed to music. Whereas the compulsive
singing was associated with a soothing, pleasant feeling,
and even a mild degree of euphoria in Patient 1, our
patients tend to withdraw from social interactions as
opposed to manic patients who engage simultaneously
in multiple activities. Thus, compulsive singing devel-
oped as an isolated, elaborate, and selective feature, un-
related to mania or psychosis. Finally, although the
singing behavior was fully recognized by both patients
as inadequate and socially disruptive, they were unable
to stop singing for more than a few seconds to min-
utes, partly because the singing-induced sensation of
pleasure felt was overwhelming.
To the best of our knowledge, this phenomenon has
not been consistently identified in PD thus far, al-
though Evans and colleagues did mention a 56-year-
old musician exhibiting various compulsive behaviors
including singing songs with invented lyrics.4 Despite
some similarities, compulsive singing appears clearly
different from other forms of vocalizations related to
parkinsonism or other movement disorders. For exam-
ple, Friedman6,7 described a PD patient with involun-
tary humming stereotypies occurring only as an “off”
phenomenon6 and a nondemented PD patient produc-
ing annoying sounds by sucking air between his teeth,
regardless of being “on” or “off.”7 Voon8 also reported
a patient with vascular parkinsonism on low doses of
LD exhibiting a prominent repetitive humming and
throat-clearing behavior, which disappeared when LD
was discontinued.8 Finally, compulsive singing differs
from klazomania, which refers to a compulsive shout-
ing behavior initially reported in encephalitis lethar-
 Table. Demographic, Clinical Data, and Dopamine Dysregulation Syndrome Criteria and Manifestations
(according to Lawrence and colleagues2) in Reported Patients
Characteristics Patient 1 Patient 2

Age, yr 70 71
Sex F M
PD duration, yr 9 5
LEU at the time of compulsive singing 1,268 634
Diagnostic criteria for DDS
PD with documented LD responsiveness Yes Yes
Need for increased doses of DRT in excess Yes Yes
Drug-seeking behavior, unwillingness to reduce DRT Yes No
Impairment of social or occupational functioning Yes Yes
Development of hypomaniac, maniac, or cyclothymic affective syndrome Yes No
in relation with DRT
Development of withdrawal state with dysphoria, irritability, depression, Yes Yes
or anxiety with reduction of DRT
Duration of disturbance ⬎ 6 months Yes Yes
Behavioral manifestations related to DDS
Punding Yes Yes
Euphoria or hypomania Yes No
Compulsive eating No Yes
Hypersexuality Yes No
Pathological gambling or shopping No No
Heightened aggression Yes Yes
Craving and withdrawal Yes No
Psychosis No No
Compulsive singing Yes Yes
LEU ⫽ L-dopa equivalent units; DDS ⫽ dopamine dysregulation syndrome; PD ⫽ Parkinson’s disease; LD ⫽ L-dopa; DRT ⫽
dopamine replacement therapy.

gica9 and later used for some complex vocal tics in
Tourette’s syndrome.10
Pathophysiological mechanisms underlying the ste-
reotyped behaviors seen in punding, induced either by
amphetamine abuse or dopaminergic agents in PD, are
believed to be similarly related to alterations of the
brain’s reward and motor systems at the level of both
the dorsal and the ventral striatum. Various scenarios
have been proposed.2 It appears indisputable that some
excessive or sensitized dopaminergic stimulation is a
necessary prerequisite for stereotypies to occur,11 and
in animals, D1 and D2 dopamine receptors have been
shown to be involved in the production of stereotyp-
ies.12 Moreover, PET and functional magnetic reso-
nance imaging studies conducted in humans have cor-
related pleasure and reward from music listening with a
significant activation of the ventral tegmental area and
accumbens nucleus, as well as of the hypothalamus, in-
sula, and orbitofrontal cortex.13–15 These findings sug-
gest that music listening may recruit similar neural cir-
cuitry of reward and emotions as other pleasure-
inducing stimuli like food and sex, and this may also
be the case for singing. Interestingly, music perception
and cognition have been located in the right superior
temporal and prefrontal cortices, and singing seizures
have been attributed to right frontal or temporal foci.
One may therefore speculate that the left-sided pre-
dominance of parkinsonism in our patients, and there-
fore the more marked dopamine deficiency in the right
dopaminergic pathways, may play, via an increased
sensitization to dopaminergic agents on the more de-
nervated striatum, a facilitating role in the production
of this selective, punding-like singing behavior. Finally,
dopamine may not be the only neurotransmitter in-
volved in the production of punding, as suggested by
the poor response usually obtained with the use of do-
pamine receptor blockers and as exemplified by Patient
1. It has been recently reported that quetiapine, an

Bonvin et al: Singing, Punding, and PD 527

atypical antipsychotic drug that interacts not only with
D1 and D2, but also with serotonin 5-HT2 receptors,
may paradoxically induce stereotyped behaviors in
PD.16 This observation, in addition to the known ben-
eficial effect of serotoninergic agents for obsessive-
compulsive disorders in non-PD patients, suggests an
additional involvement of the serotonin system, and
perhaps other neurotransmitters, in the pathogenesis of
punding.
References
1. Evans AH, Lees AJ. Dopamine dysregulation syndrome in Par-
kinson’s disease. Curr Opin Neurol 2004;17:393–398.
2. Lawrence AD, Evans AH, Lees AJ. Compulsive use of dopa-
mine replacement therapy in Parkinson’s disease: reward sys-
tems gone awry? Lancet Neurol 2003;2:595– 604.
3. Friedman JH. Punding on levodopa. Biol Psychiatry 1994;36:
350 –351.
4. Evans AH, Kazenschlager R, Paviour D, et al. Punding in Par-
kinson’s disease: its relation to the dopamine dysregulation syn-
drome. Mov Disord 2004;19:397– 405.
5. Rylander G. Psychoses and the punding and chorefirom syn-
dromes in addiction to central stimulant drugs. Psychiatr Neu-
rol Neurochir 1972;75:203–212.
6. Friedman JH. Involuntary humming in autopsy-proven Parkin-
son’s disease. Mov Disord 1993;8:401.
528 Annals of Neurology Vol 62 No 5 November 2007
7. Friedman JH. More on repetitive behaviors in Parkinson’s dis-
ease. Mov Disord 2005;20:509.
8. Voon V. Reply: repetitive behaviors in Parkinson’s disease. Mov
Disord 2005;20:509 –510.
9. Wohlfart G, Ingwvar DH, Hellberg AM. Compulsory shouting
(Benedek’s “klazomania”) associated with oculogyric spasms in
chronic epidemic encephalitis. Acta Psychiatr Scand 1961;36:
369 –377.
10. Jankovic J, Mejia NI. Tics associated with other disorders. Adv
Neurol 2006;99:61– 68.
11. Evans AH, Pavese N, Lawrence AD, et al. Compulsive drug use
linked to sensitized ventral striatal dopamine transmission. Ann
Neurol 2006;59:852– 858.
12. Nakazato T, Akiyama A. Behavioral activity and stereotypy in
rats induced by L-DOPA metabolites: a possible role in the
adverse effects of chronic L-DOPA treatment in Parkinson’s
disease. Brain Res 2002;930:134 –142.
13. Jeffries KJ, Fritz JB, Braun AR. Words in melody: an H215O
PET study of brain activation during singing and speaking.
NeuroReport 2003;14:749 –754.
14. Menon V, Levitin DJ. The rewards of music listening: response
and physiological connectivity of the mesolimbic system. Neu-
roImage 2005;28:175–184.
15. Blood AJ, Zatorre RJ. Intensely pleasurable responses to music
correlate with activity in brain regions implicated in reward and
emotion. Proc Natl Acad Sci U S A 2001;98:11818 –11823.
16. Miwa H, Morita S, Nakanishi I, Kondo T. Stereotyped behav-
iors or punding after quetiapine administration in Parkinson’s
disease. Parkinsonism Relat Disord 2004;10:177–180.