Neurocase
The Neural Basis of Cognition

ISSN: 1355-4794 (Print) 1465-3656 (Online) Journal homepage: http://www.tandfonline.com/loi/nncs20

A case of musical anhedonia due to right
putaminal hemorrhage: a disconnection syndrome
between the auditory cortex and insula
Masayuki Satoh, Natsuko Kato, Ken-ichi Tabei, Chizuru Nakano, Makiko Abe,
Risa Fujita, Hirotaka Kida, Hidekazu Tomimoto & Kiyohiko Kondo
To cite this article: Masayuki Satoh, Natsuko Kato, Ken-ichi Tabei, Chizuru Nakano, Makiko
Abe, Risa Fujita, Hirotaka Kida, Hidekazu Tomimoto & Kiyohiko Kondo (2016): A case of musical
anhedonia due to right putaminal hemorrhage: a disconnection syndrome between the
auditory cortex and insula, Neurocase
To link to this article: http://dx.doi.org/10.1080/13554794.2016.1264609

Published online: 07 Dec 2016.

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Date: 16 December 2016, At: 06:47

NEUROCASE, 2016
http://dx.doi.org/10.1080/13554794.2016.1264609

A case of musical anhedonia due to right putaminal hemorrhage: a disconnection
syndrome between the auditory cortex and insula
Masayuki Satoha, Natsuko Katob, Ken-ichi Tabeia, Chizuru Nakanoa, Makiko Abea, Risa Fujitaa, Hirotaka Kidaa,
Hidekazu Tomimotoa,b and Kiyohiko Kondoc
a
Department of Dementia Prevention and Therapeutics, Graduate School of Medicine, Mie University, Tsu, Japan; bDepartment of Neurology,
Graduate School of Medicine, Mie University, Tsu, Japan; cDepartment of Neurology, Yoka Hospital, Yabu, Japan

ABSTRACT

ARTICLE HISTORY

A 63-year-old, right-handed professional chorus conductor developed right putaminal hemorrhage, and
became unable to experience emotion while listening to music. Two years later, neurological examination revealed slight left hemiparesis. Neuromusicological assessments revealed impaired judgment of
“musical sense,” and the inability to discriminate the sound of chords in pure intervals from those in
equal temperament. Brain MRI and tractography identified the old hemorrhagic lesion in the right
putamen and impaired fiber connectivity between the right insula and superior temporal lobe. These
findings suggest that musical anhedonia might be caused by a disconnection between the insula and
auditory cortex.

Received 25 May 2016
Accepted 17 November
2016

Introduction
It is generally accepted that the emotional processing of music
has two components: emotion perception and emotional
experience (Juslin & Västfjäll, 2008). Emotion perception
means the recognition of expressed emotions in music without necessarily feeling an emotion (Juslin & Västfjäll, 2008).
Emotional experience is the subjective experience of emotion
(Juslin & Västfjäll, 2008). The reported literature on the impairment of emotion perception and emotional experience of
music reveals a double dissociation, in other words, these
two cognitive domains have, at least in part, independent
neural networks in the brain (Satoh, Nakase, Nagata, &
Tomimoto, 2011). Cases have been reported in which one of
these two domains was impaired and another preserved
(Griffiths, Warren, Dean, & Howard, 2004; Hirel et al., 2014;
Matthews, Chang, May, Engstrom, & Miller, 2009; Mazzoni,
Moretti, Pardossi, Vista, & Muratorio, 1993; Peretz & Gagnon,
1999; Satoh et al., 2011).
Selective loss of the emotional experience of music was
named musical anhedonia by the present author (Satoh et al.,
2011). Musical anhedonia is characterized by the following: (i)
normal otolaryngological function, (ii) normal perception of
elementary musical components and emotion perception of
music, (iii) impaired capacity to respond emotionally to music,
and (iv) normal emotional experience to other auditory stimuli
and sensory modalities such as environmental sounds and
vision. Musical anhedonia was first presented as an acquired
disorder, but this concept was widened to include congenital
impairment of the emotional experience of music (MasHerrero, Zatorre, Rodriguez-Fornells, & Marco-Pallares, 2014;
Zatorre, 2015). In this article, we use the term “musical anhedonia” in its original meaning, namely the acquired impairment of emotional experience of music due to cerebral
CONTACT Masayuki Satoh

bruckner@clin.medic.mie-u.ac.jp

© 2016 Informa UK Limited, trading as Taylor & Francis Group

KEYWORDS

musical anhedonia;
putaminal hemorrhage;
insula; auditory cortex;
disconnection

damage. Until now, only four cases of musical anhedonia
had been reported. The first case was a 24-year-old amateur
guitarist (Mazzoni et al., 1993). After a hemorrhage due to an
arteriovenous malformation at the right temporoparietal lobe,
he lost the esthetic pleasure of listening to music. The second
case was a 52-year-old radio announcer (Griffiths et al., 2004).
Due to an infarction of the left insula partly extending into the
frontal lobe and amygdala, emotional response to specific
music was lost. The third case was a 71-year-old retired teacher (Satoh et al., 2011), who developed an infarction of the
right temporoparietal lobe. After the cerebrovascular event, he
found himself unable to experience emotion while listening to
music even that which he had enjoyed prior to the illness. The
fourth case was recently reported in French (Hirel et al., 2014).
The patient was a 43-year-old amateur musician; due to an
ischemic lesion of the right superior temporal lobe, the patient
showed musical anhedonia. While the neural basis of musical
anhedonia is controversial, according to a similar symptom in
the vision system called “visual hypoemotionality” (Bauer,
1982; Habib, 1986), it is hypothesized that an auditory-limbic
disconnection is responsible.
We present a patient with musical anhedonia who was a
professional chorus conductor. He suffered from a right putaminal hemorrhage, and subsequently lost the esthetic pleasure of
listening to music. Neuroimaging studies suggest that a disconnection between the insula and auditory cortex is, in part,
responsible for the occurrence of musical anhedonia.

Case report
History
In May, 2011, a 63-year-old professional chorus conductor
being treated for hypertension developed a right putaminal

2

M. SATOH ET AL.

hemorrhage. He was treated conservatively, and 1 month later
was transferred to a rehabilitation hospital. At the end of July,
he returned home with slight left hemiparesis which was
measured by manual muscle testing, and mild hypoesthesia
on the left side of his body. He was able to independently
perform activities of daily life, such as dressing, eating, bathing, going out using public transportation, and shopping.
When he returned to his musical profession, he found himself
unable to have any emotional experience while listening to
music. He could not elicit interest from any type music, even
his favorite genre. When he conducted his chorus group, he
felt much different than prior to the cerebrovascular event;
these changes are described below. Before the illness he felt
great joy and “chills” when the harmony sounded complete.
He subsequently lost this sensation, and thus lacked self-confidence as to whether the harmony was good or not. He felt
that the sound was dull and lacked freshness, though he could
acoustically recognize that the harmony was correct in its
physical sound properties. He could recognize familiar music
and discriminate subtle differences in the timbre of voices,
although it had become somewhat difficult to separately
identify the alto and tenor voices in soprano-alto-tenor-base
(SATB) chorus. He also found it difficult to speak in noisy
situations. These symptoms suggested the existence of the
disturbance of cocktail party effect. None of his chorus members noticed this impairment. During rehearsals, the patient
frequently sang melodies and phrases in order to make choir
members understand his willing. His singing was musically
appropriate, so the members could easily understand that.
His musical performances were excellent as before the illness,
and the audience was satisfied when attending his concerts.
No hearing deficit was found during consultation with an
otolaryngologist. He was able to enjoy pictures in an art
museum, movies in the theater, watching comic dialogues
on television, and eating delicious foods, as he did before
the cerebrovascular event. His symptoms remained
unchanged for 2 years. In April, 2013, he was referred to us
for assessment of his impaired musical appreciation.

Neurological and neuropsychological examinations
Neurological examination revealed slight left hemiparesis and
mild hypoesthesia on the left side of his body, but there was
no evidence of unilateral neglect, aphasia, apraxia, dyscalculia,
or visual discrimination difficulties. No hearing deficit was
found during consultations with an otolaryngologist.
Neuropsychological assessments revealed a normal score on
the Mini-Mental State Examination (27/30) and a perfect score
on a Japanese version of Raven’s Colored Progressive Matrices
(36/36) (Raven, 1995). To assess memory function, we performed the Japanese version of the Rivermead Behavioral
Memory Test (RBMT). The RBMT yielded a normal score: a
standard profile score of 22 (mean ± SD; 19.73 ± 2.93) and a
screening score of 10 (9.15 ± 1.78). He clearly remembered
daily and social events. Therefore, we reasonably concluded
that the patient had normally functioning short- and longterm memory. He could correctly copy the figure of a cube.
When the patient was asked to verbally name as many different animals as he could in 1 min, the patient responded with

25 words where the lower limit of normal is between 8 and 10
(Satoh, Takeda, & Kuzuhara, 2009). His letter fluency beginning
with “ka” was also excellent (17 words/min.). The results of the
Trail Making Test – A and B were also normal (A 146 sec., mean
± SD; 173.2 ± 65.8; B 143 sec., mean ± SD; 230.2 ± 89.5). Given
that the quality of his musical performance was at the same
level as before the cerebrovascular event, we can conclude
that his frontal and executive function was also preserved.
Speech audiometry revealed a normal score on single sounds
(146/148). To assess recognition of environmental sounds, a
previously described test was used (Satoh, Takeda, &
Kuzuhara, 2007; Satoh et al., 2005, 2011). Twenty-four familiar
environmental sounds were presented, and the patient correctly stated what each sound meant (24/24). Assessment of
responses to other sounds showed that he felt comfortable
when listening to recorded natural sounds. Unpleasant stimuli
such as disgusting smells, the sound of scratching a blackboard, pictures of wars or disasters, and physical pain caused
him discomfort. We may say that these negative stimuli normally caused in negative feeling to the patient. For the assessment of emotional experience we used the International
Affective Picture System (IAPS) which is a set of normative
emotional stimuli for experimental investigations of emotion
and attention (Center for the Study of Emotion and Attention,
1999). The two primary components are affective valance
(ranging from pleasant to unpleasant) and arousal (ranging
from calm to excited). To assess these dimensions, the SelfAssessment Manikin, which is an affective rating system, was
used. According to the guidelines of Meagher’s report
(Meagher, Arnau, & Rhudy, 2001), we used 39 IAPS slides
(pleasant 10; unpleasant 9; neutral 20), when compared to
the mean value of the IAPS, the patient scored within normal
limits: total valence 5.0 ± 1.9 (mean ± SD; 5.2 ± 1.5) and total
arousal 4.7 ± 2.0 (mean ± SD; 4.1 ± 1.7). The results of pleasant,
unpleasant, and neutral visual stimuli were also normal: pleasant valence 7.1 (mean ± SD; 7.4 ± 1.6) and arousal 6.2 (mean
± SD; 4.4 ± 2.2), unpleasant valence 2.1 (mean ± SD; 3.4 ± 1.7)
and arousal 6.6 (mean ± SD; 6.6 ± 1.9), and neutral valence 5.3
(mean ± SD; 4.9 ± 1.3) and arousal 3.1 (mean ± SD; 2.7 ± 1.7).
Thus, he was able to normally experience emotion to visual
stimuli, and the preservation of pleasure towards a variety of
other stimuli suggested that his loss of pleasure was restricted
to listening to music.

Neuromusicological assessments
Perception and expression of music were investigated by tests
used in our previous studies of amusic patients (Satoh et al.,
2005, 2007, 2011) and by “The Montreal Battery of Evaluation
of Amusia (MBEA) (Peretz, Champod, & Hyde, 2003). For the
former tests (Satoh et al., 2005, 2007, 2011), we performed the
following tasks regarding musical perception: pitch discrimination, discrimination and recognition of familiar songs, discrimination of unfamiliar phrases, rhythm discrimination,
meter discrimination, discrimination of isolated chords, and
judgment of tonality (Table 1). For further details, please see
the aforementioned papers (Satoh et al., 2005, 2007, Satoh
et al., 2011). The results are summarized in Table 2, the
patient’s score was within the normal range for all

NEUROCASE

Table 1. Assessment of musical abilities.
Perception of music
Pitch discrimination (Hz)
Discrimination and recognition of familiar songs (/30)
Discrimination of unfamiliar phrases (/30)
Rhythm discrimination (/30)
Meter discrimination (/4)

Discrimination of isolated chords (/14)
Judgment of tonality (/20)

Expression of music
Singing familiar songs

After presenting a pure tone of 440 Hz as the standard, we presented a second tone and
measured the range in which the standard and second tones were heard as the same
pitch.
Thirty familiar nursery songs, all in a major key, were chosen and in twenty of these songs,
a part of the melody was locally altered. Two different response modes were used: (i)
song decision; (ii) “familiar-unfamiliar” decision.
The tonal memory test of Seashore’s measures of musical talents (Seashore, Lewis, &
Saetveit, 1960) was used. Subjects were presented with two serial phrases and were
asked which tone was altered in the second phrase.
The rhythm test from Seashore’s measures of musical talents (Seashore et al., 1960) was
used. Thirty pairs of rhythm patterns were presented, and subjects were asked to make
a same-difference discrimination.
We used items from the meter test of an assessment of musical capacity for school
children (`Mashino, Hamano, & Motegi, 1979). Following an example of two pieces of
music that had double or triple time, the examiner presented four pieces of music that
had double or triple time. The subject was then required to answer “two” or “three” for
each piece of music.
A pair of chords in which the interval was 1 s was presented and subjects were asked to
make a same-different discrimination. We used an item from the harmony test from an
assessment of musical capacity for schoolchildren (Mashino et al., 1979).
We made 20 kinds of sequences of 3 chords in which 2 chords had subdominant to
dominant seventh progression. The last chords of eight tasks were the tonic chords of
each key which induced a natural feeling of an end. In 12 tasks, the last chords were
irrelevant to the preceding 2 chords, which induced an unfinished, peculiar feeling.
Subjects were asked to make a finished/unfinished response for each task.
The patient and control subjects were asked to sing 10 well-known Japanese nursery songs
without accompaniment with the help of written lyrics.
Subjects were required to vocally reproduce single tones presented by the examiner. The
examination was performed under two conditions: (i) while the tone was sounding, and
(ii) immediately following the end of the tone presentation.
Subjects were presented with three two-note phrases and were asked to sing each phrase.
The phrases consisted of two notes whose pitches were separated by 3, 4, or 5 degrees
(e.g., D-F sharp, D-G, and D-A).
Ten simple rhythm patterns 2 bars long in 4/4 time were presented and subjects were
required to reproduce the rhythm by hand clapping.

Reproduction of single sounds
Reproduction of two-note phrases
Reproduction of simple rhythm

Table 2. Results for musical abilities.
Perception of music
Pitch discrimination (Hz)
Discrimination and recognition of
familiar songs (/30)
Discrimination of unfamiliar phrases
(/30)
Rhythm discrimination (/30)
Meter discrimination (/4)
Discrimination of isolated chords (/14)
Judgment of tonality (/20)
Expression of music
Singing familiar songs
Reproduction of single sounds
Reproduction of two-note phrases
Reproduction of simple rhythm

3

Present case

Control

439–441
(−3.939–3.930
cents)
30

434–446
(−23.770–23.448
cents)
27.7 (26–30)

30

21.8 (17–27)

26
4
14
20

24.8 (20–29)
3.8 (3–4)
12.6 (11–14)
15.6 (12–19)

Well
Well
Well
Well

Well
Well
Well
Well

components. For the evaluation of music perception, we also
used” The MBEA (Peretz et al., 2003). The MBEA aims to assess
musical ability in brain-damaged patients and consists of six
tasks: scale, contour interval, rhythm, meter, and memory task.
Our patient scored within normal limits on all tasks: Scale 27
(mean ± SD; 27 ± 2.3, cutoff 22), Different contour 29 (mean ±
SD; 27 ± 2.2, cutoff 22), Same contour 30 (mean ± SD; 26 ± 2.4,
cutoff 21), Rhythmic contour 29 (mean ± SD; 27 ± 2.1, cutoff
23), Metric 30 (mean ± SD; 26 ± 2.9, cutoff 20), and Incidental
memory 27 (mean ± SD; 27 ± 2.3, cutoff 23).

In order to evaluate the perceptual capacity of musical
characteristics, we performed portions of the Musical
Aptitude Profile (Gordon, 1995), a test to assess musical sensitivity, including: Part I, Phrasing; Part II, Balance; Part III, Style.
These tests were designed to evaluate the musical aptitudes
of elementary, junior, and senior high school students, and
consisted of 30 pairs of identical musical phrases which were
played by a violin and/or a cello. In each pair, one performance sounded more musically accomplished than the other.
Differences between the two performances consisted of timbre, legato or staccato, crescendo or decrescendo, accelerando
or ritardando, playing carefully or crudely, the way of finishing
the phrase, and the “agogic” which is the term for a type of
accent based on duration (lingering on a note in order to
stress it) (Sadie, 1994). The subject was asked to determine
which of the two renditions of the same musical phrase made
better “musical sense.” The word “musical sense” means
appropriate and suitable characteristics of music including
intonation, articulation, melodic contour, tempo, timbre,
phrasing, ending, and style (Gordon, 1995). These tests can
reveal a more subtle and complex capacity for perception of
the musical characteristics which produce emotional experiences. For the normative data, we used results from senior
high school students that were highest among the tested
elementary, junior, and senior high school students. If the
patient scored below the normative average, we could assume
that his ability to perceive esthetical characteristics of music

4

M. SATOH ET AL.

was impaired. The results of these tests were as follows: Part
I-Phrasing test 23 (mean ± SD; 23 ± 3.79, percentile 44%), Part
II-Balance 19 (mean ± SD; 23.2 ± 3.96, percentile 15%), and
Part III Style 23 (mean ± SD; 23 ± 4.13, percentile 44%). The
scores of the patient fell within or below the normative means.
Given that our patient was a professional musician and the
normative data were those of nonmusicians, we can reasonably conclude that the ability to perceive the subtle characteristics of “musical sense” was impaired in this patient.
The patient complained that while conducting a chorus he
could not judge if the harmony was acoustically correct or not.
To assess this ability, we performed a chord-type discrimination
task. There are several types of chords in music. A chord with pure
intervals consists of tones whose pitches are completely equal to
their physiological properties based on the fundamental tone. For
example, an octave is a tone whose pitch has double frequency,
and a tone with a 5-degree interval (ex. “G” on “C,” 701.955 cents)
has a one and a half difference of frequency (Table 3). The standard Western temperament today is equal temperament, in which
the octave is divided into 12 uniform semitones (Sadie, 1994). In
equal temperament, the pitch interval with 5 degrees is slightly
narrower than pure interval which is acoustically correct (Table 3).
Therefore, the sound of chords with equal temperament is not as
clear as those with pure intervals, and professional musicians
usually can discriminate whether a chord is of pure intervals or
not. For the assessment, we played a pair of chords, one was pure
and the other was equal temperament. Using Musical Instrument
Digital Interface (MIDI) function of Cubase Essential 4 (Steinberg),
we made triad of C major in the timber of AKOUSTIK PIANO
(Native Instruments). By using the tuning function, we made C

Table 3. Triad on C in pure interval and equal temperament.
Pure interval
C
E
G

Equal temperament

Hz

Cent

Hz

Cent

264.0
330.0
396.0

0
386.314
701.955

261.6
329.6
392.0

0
400.024
700.189

major triads with pure intervals and in equal temperament
(Table 3), and stored in the personal computer in Wave files. We
prepared four kinds of stimuli with pair of triads: pure intervalpure interval, pure interval-equal temperament, equal temperament-pure interval, and equal temperament-equal temperament.
These stimuli were randomly arranged, and sounded two times
(total number of stimuli was eight). We asked the patient whether
the chord was in the pure interval, or not. Five professional
musicians could easily and completely discriminate the chords
with pure interval and equal temperament. The ratio of correct
answers given by the patient was 62.5% (correct 5). Among three
incorrect answers, one was replied as different though the pair of
chords was the same with pure intervals, and two replied as the
same, though they were different. We thus concluded that the
patient had impaired perception of the subtle differences in the
sound of chords.

Neuroimaging assessments
Duvernoy’s atlas (Duvernoy, 1991) was used to estimate the
location of the patient’s brain lesion. Two years after the onset
of the cerebrovascular event, fluid attenuated inversion recovery images of MRI scans revealed that the old hemorrhagic
lesion was situated in the right putamen (Figure 1). Subcortical
fiber degeneration was observed between the superior temporal gyrus and the posterior two thirds of the right insula.
N-isopropyl-p-[123I] iodoamphetamine (123I-IMP) single photon
emission computed tomography imaging showed decreased
regional cerebral blood flow (rCBF) in the right insula and
temporal lobe (Figure 2).
For the tractography by diffusion tensor imaging (DTI), the
resulting averaged DTI dataset was preprocessed (non-brain
tissue removal, eddy current (an induced electric current formed
within the body of a conductor when it is exposed to a time
varying magnetic field) and motion correction) and then analyzed using FMRIB Diffusion Toolbox (FDT 3.0) from FSL 5.0.9
(Smith et al., 2004; Woolrich et al., 2009). The processing step we
need to take is to correct for eddy currents, which occur due to
the changing gradient field directions, and head motion.

Figure 1. FLAIR MRI two years after the cerebrovascular event. FLAIR: fluid attenuated inversion recovery, R: right.

NEUROCASE

5

Figure 2. 123I-IMP SPECT imaging shows decreased rCBF in the right insula and temporal lobe. 123I-IMP: N-isopropyl-p-[123I] iodoamphetamine, rCBF: regional
cerebral blood flow, R: right, SPECT: single photon emission computed tomography.

Figure 3. Colored encoding diffusion tensor imaging (CEDTI) map using FMRIB Diffusion Toolbox. Red, green, and blue color show right-left, anterior-posterior, and
rostal-caudal fibers, respectively. The image level of (x, y, z) is (66, 51, 25). The depicted fiber tracking between the insula and superior temporal gyrus is decreased
in the right hemisphere (white arrows). R: right.

Following eddy current correction we can now use DTIFit to fit
tensors to the data and determine a variety of values include the
fractional anisotropy at each voxel as well as the principle diffusion direction (Figure. 3).

Discussion
The patient was a professional musician that, due to a putaminal
hemorrhage in the right hemisphere, lost the esthetic pleasure of
listening to music. This symptom was formerly named “musical
anhedonia” (Satoh et al., 2011), and is to our knowledge only the
fifth case presenting with this symptom (Table 4). The abilities of
our patient are summarized as follows: (i) normal perception and
expression of music, specifically, the quality of his performances
was unchanged, (ii) normal speech audiometry and environmental sound recognition, (iii) impairment of judgment of “musical

sense,” (iv) inability to discriminate the sound of chords in pure
versus equal temperament, (v) impairment listening to the inner
vocal parts, such as alto and tenor, while conducting a chorus,
and when experiencing the cocktail party effect during conversation, (vi) right putaminal hemorrhage and impairment of fiber
connections between the insula and superior temporal lobe
including the auditory cortex, and (vii) intact response to visual
and other auditory stimuli and sensory modalities. In the following section, we discuss the mechanism of musical anhedonia as a
disconnection syndrome between the insula and auditory cortex.
In recent years, the function of the insula has been an
important topic in neurology. The insula, as a multimodal
area, has a major role as a convergence zone implicated in
the coordination between internal and external information
through
emotional
subjective
awareness
(Ibańez,
Gleichgerrcht, & Manes, 2010). The insular cortex has

○: preserved; ×: impaired; AVM: arteriovenous malformation; F: female; M: male; np: not particular; R: right.

Diagonosis
Site of lesion

Right temporoparietal
Left frontal region, amygdala, insula
Right temporoparietal
Right superior temporal lobe
Subcortical region of right temporal lobe
Bilateral temporal and left frontal
Bilateral temporal
×
×
×
×
×
○
○
○
○
○
○
○
×
×
Amateur guitarist
np
np
Amateur
Professional chorus conductor
np
np
Ambidextrous
R
R
R
R
R
R
M
M
M
M
M
F
M
24
52
71
43
63
40
30
1993
2004
2011
2014
2015
1999
2009
Mazzoni
Griffiths
Satoh
Hirel
Present case
Peretz
Matthews

J Neurol Neurosurg Psychiatry
J Neurol Neurosurg Psychiatry
Neurocase
Rev Neurologique
Neurocase
Neurocase
Neurocase

Musical emotion

Perception Experience
Musical history
Handedness
Age (year-old) Sex
Year
Author

Journal

AVM, hemorrhage
Infarction
Infarction
Infarction
Putaminal hemorrhage
Infarction
Egeneration

M. SATOH ET AL.

Table 4. Summary of reported cases of musical anhedonia with associated pattern of symptoms.

6

connections with the primary and secondary somatosensory
areas, anterior cingulate cortex, amygdala, prefrontal cortex,
superior temporal gyrus, temporal pole, orbitofrontal cortex,
frontal operculum, parietal operculum, primary auditory cortex, auditory association cortex, visual association cortex,
olfactory bulb, hippocampus, entorhinal cortex, thalamus,
medial geniculate body, and motor cortex (Craig, 2009;
Ibańez et al., 2010; Mutschler et al., 2009; Nagai, Kishi, &
Kato, 2007). The insula is known as a multi-sensory integration
site (Figure 4) (Ibańez et al., 2010; Nagai et al., 2007). The
insular cortex affects autonomic function, and involved in
interoceptive awareness, pain, temperature perception, olfactory, gustatory, vestibular sensation, auditory processing, language, and emotional processing (Mutschler et al., 2009).
Koelsch (2014) reported that grey matter loss of posterior
insula was associated with impaired recognition of emotions
expressed by music in the patient with frontotemporal lobar
degeneration. This finding reinforced the opinion that the
insula has a causal role in music-evoked emotions (Koelsch,
2014).
The neural architecture for encoding the biological value of
music consists of a neural network for perceptual coding of
music, emotion and reward processing, and higher-order cognitive processing (Clark, Downey, & Warren, 2014). Based on
the present and reported literature, musical anhedonia might
be caused by a disconnection between the insula and auditory
cortex. The Griffith’s case had an infarction of the left insula
(Griffiths et al., 2004). In contrast, our first case with musical
anhedonia (Satoh et al., 2011) and Mazzoni’s case (Mazzoni
et al., 1993) had a lesion at the subcortical area of the right
temporoparietal region, possibly including the fibers from/to
the auditory cortex. The present case of musical anhedonia is
the first to clearly demonstrate impairment of the fiber connections between the insula and auditory cortex using tractography. The esthetic pleasure in listening to music, for
example, the deep impression consisting of shivering and/or
tearing, is related to emotional and autonomic nervous
responses. All cases of musical anhedonia have presented
with a lesion at the insula and/or its subcortical region,
which impaired the input of information from the auditory
cortex to the insula resulting in disrupted integration of auditory, emotional, and autonomic nervous function. The lateralization of musical anhedonia is unknown. Among the five
cases mentioned above, four cases (Hirel et al., 2014;
Mazzoni et al., 1993; Satoh et al., 2011; and the present
Satoh case) had the responsible brain lesion in the right
cerebral hemisphere; only the case reported by Griffiths et al.
(2004) had a lesion in the left hemisphere. Of the pre-illness
musical abilities, only the present case was a professional
musician. In terms of handedness, four patients were righthanded (Griffiths et al., 2004; Hirel et al., 2014; Satoh et al.,
2011; and the present Satoh case), while the Mazzoni et al.
(1993) case was ambidextrous. Much remains to be learned
about the relationship between the cognitive processing of
esthetic pleasure and hemispheric lateralization, pre-illness
musical ability, and handedness.
What is the role of the insula in the occurrence of musical
anhedonia? While this is an ongoing question, the insula
might participate in the high level esthetic perception of

NEUROCASE

7

Figure 4. Diagram showing the neural circuit between the association cortex, limbic system (representing the emotional system), hypothalamus (representing the
autonomic nervous system), thalamus (forming the sensory input), and the insula (acting as the multi-sensory integration site). The “X” shows the region thought to
be impaired in the present case. Figure adapted from Lippincott’s Illustrated Reviews: Neuroscience.

listening to music. The present patient showed impaired judgment of “musical sense” and the perception of chords with
pure intervals, both of which are very sensitive finely honed
abilities obtained by long-term professional training. Thus, as a
multi-sensory integration site which connects many kinds of
information, the insula might play a key role in the deep
impressions of artistic appreciation.
In summary, we reported a case of musical anhedonia
caused by right putaminal hemorrhage. Neuroimaging
showed a disconnection between the insula and auditory
cortex in the right hemisphere. Recent understanding of the
insula suggests that this region plays an important role in
integrating multi-sensory information. Thus, we can interpret
the present and reported cases of musical anhedonia as a
disconnection syndrome between the insula and auditory
cortex.

Disclosure statement
No potential conflict of interest was reported by the authors.

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