One of the earliest papers I came across on the phenomenon was by Dr. Daniel Jacome in a 1984 issue of the Journal of Neurology, Neurosurgery and Psychiatry. Dr. Jacome described the case of a “musically naive patient with dominant fronto-temporal and anterior parietal infarct developed transcortical mixed aphasia. From early convalescence, he exhibited elated mood with hyperprosody and repetitive, spontaneous whistling and whistling in response to questions”. In addition to the whistling, Jacome also reported that the individual spontaneously sang without any error in melody, lyrics, pitch, and rhythm. The man also developed the desire to spend long periods of time listening to music.
Compulsive whistling was also reported in a 2012 issue of BMC Psychiatry by Dr. Rosaura Polak and her colleagues. Their paper reported the case of a 65-year-old man who started whistling compulsively following a heart attack. The heart attack had caused some brain damage due to a lack of oxygen to the brain. Prior to the cardiac arrest, the man had never displayed any obsessive-compulsive symptoms or psychiatric complaints. He was treated with clomipramine (a seretonin reuptahe inhibitor) and this decreased time spent compulsively whistling. The authors concluded that:
“This case shows that the whistling can be explained in the context of compulsivity with its repetitive character. It illustrates that the compulsive behavior can be present as an independent symptom of cortico-striatal dysfunction, and may not always belong to frontal syndrome, punding or OCD. Finally, this case illustrates that pharmacological treatment with clomipramine is effective and suggests that similar cases of compulsivity may benefit from this treatment”
A paper published in a 2000 issue of the Journal of the Korean Neurological Association
examining 25 patients with fronto-temporal dementia
(20 women and five men with an average age of 56 years) noted that compulsive behavior is one of the commonest early manifestations of the condition. The researchers analyzed their symptoms and compulsive behaviors and 22 of the patients (88 percent) showed various compulsive behaviors including “reading signboards, stereotypy of speech, ordering, hoarding, washing, checking, counting, singing, and wandering a fixed route.”
However, no real detail was provided in relation to the compulsive singing. Other papers – such as one in a 2002 issue of European Psychiatry
by Dr. F. Muratori and colleagues – have reported compulsive singing in people that have Kleine-Levin syndrome (i.e., recurrent primary hypersomnia
where individualscan lapse into a deep sleep
at any time without warning, sometimes lasting as long as 16 hours).
One of the most interesting and detailed papers on compulsive singing is a 2007 paper by Dr. Christophe Bonvin and colleagues in the Annals of Neurology. They reported two case studies of individuals with advanced Parkinson's disease who exhibited “a peculiar and stereotyped behavior characterized by an irrepressible need to sing compulsively when under high-dose dopamine replacement therapy.” They argued that the compulsive singing behavior shared many features with punding (i.e., repetitive behavior that is a side effect of some drugs). Here is a brief summary of the two cases:
Patient 1: “A 70-year-old female university professor and amateur piano player while being treated with 1,268 L-dopa equivalent units (LEU)…exhibited a repetitive, compulsive behavior characterized by singing endlessly…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 became irritated…All symptoms improved minimally after quetiapine (25mg twice daily) had been introduced”.
Patient 2: “A 71-year-old male painter…[that] grew up in a family of musicians and used to spend time listening to classical music and singing willingly…While being treated with 634 LEU, he started to hum repeatedly the same melody, initially once a week, then several times daily, mostly in the evening…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 irrepressible 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)…[This resulted in] the patient losing control over the compulsion and singing almost unendingly all day…Eventually, compulsive singing improved, but did not disappear, when LD/benserazide was reduced to the minimal daily doses (500/125mg)”.
The authors noted that both of these patients developed a peculiar, stereotyped, and compulsive behavior characterized by an urge to sing repeatedly the same song. They also concluded that in both cases:
“[The] compulsive singing developed as an isolated, elaborate, and selective feature, unrelated to mania or psychosis…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 minutes, 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 [Parkinson’s disease] thus far…Moreover, PET and functional magnetic resonance imaging studies conducted in humans have correlated pleasure and reward from music listening with a significant activation of the ventral tegmental area and accumbens nucleus, as well as of the hypothalamus, insula, and orbitofrontal cortex. These findings suggest that music listening may recruit similar neural circuitry of reward and emotions as other pleasure inducing stimuli like food and sex, and this may also be the case for singing”.
In 2010, Dr. Hiroshi Kataoka and Dr. Satoshi Ueno described the case of an 82-year old woman (also with Parkinson disease) who started to sing compulsively (in the absence of any other types of pathologic behavior) following treatment with pergolide. In the journal Cognitive and Behavioral Neurology
, the authors reported that she would hum the same melody and sing songs repeatedly. When she stopped taking her ergolide medication, the compulsive singing and humming considerably subsided. Drs. Kataoka and Ueno suggested that a dopamine agonist in the patient’s medication may have contributed to her compulsive singing. The same phenomenon was also reported in three Parkinson’s patients treated with dopamine agonists by a Dr. C. Borrue-Fernandez at a Spanish conference on treating Parkinson’s disease in 2011.
It would appear from the few papers that have been published on compulsive singing that it almost always occurs alongside or as a consequence of other primary medical conditions and that some excessive or sensitized dopaminergic stimulation is a necessary prerequisite for such musical stereotypies to occur.
References and further reading
Bonvin, C., Horvath, J., Christe, B., Landis, T., & Burkhard, P. R. (2007). Compulsive singing: another aspect of punding in Parkinson's disease. Annals of Neurology, 62, 525-528.
Borrue-Fernandez, C. (2011). Compulsive singing as an Impulse Control Disorder in dopamine agonist treated patients: Review of three cases. The 15th Congress of the European Federation of Neurological Societies.
Jacome, D. E. (1984). Aphasia with elation, hypermusia, musicophilia and compulsive whistling. Journal of Neurology, Neurosurgery and Psychiatry, 47, 308-310.
Kataoka, H., & Ueno, S. (2010). Compulsive singing associated with a dopamine agonist in Parkinson disease. Cognitive and Behavioral Neurology, 23(2), 140-141.
Muratori, F., Bertini, N., & Masi, G. (2002). Efficacy of lithium treatment in Kleine–Levin syndrome. European Psychiatry, 17, 232–3.
Polak, A. R., van der Paardt, J. W., Figee, M., Vulink, N., de Koning, P., Olff, M., & Denys, D. (2012). Compulsive carnival song whistling following cardiac arrest: a case study. BMC Psychiatry, 12(1), 75.
Yoon, S. J., Jeong, J. H., Kang, S. J., & Na, D. L. (2000). Compulsive behaviors and presenting symptoms of frontotemporal dementia. Journal of the Korean Neurological Association, 18, 681-686