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Do individuals with Williams syndromepossess absolute pitch?Pastora Martínez-Castilla a , María Sotillo b & Ruth Campos ba Department of Developmental and Educational Psychology,Universidad Nacional de Educación a Distancia (UNED), Madrid,Spainb Department of Basic Psychology, Universidad Autónoma deMadrid, Madrid, Spain

Available online: 06 Dec 2011

To cite this article: Pastora Martínez-Castilla, María Sotillo & Ruth Campos (2011): Doindividuals with Williams syndrome possess absolute pitch?, Child Neuropsychology,DOI:10.1080/09297049.2011.639755

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Child Neuropsychology, 2011, iFirst, 1–19http://www.psypress.com/childneuropsychISSN: 0929-7049 print / 1744-4136 onlinehttp://dx.doi.org/10.1080/09297049.2011.639755

Do individuals with Williams syndrome possess absolute

pitch?

Pastora Martínez-Castilla1 , María Sotillo2, and Ruth Campos2

1Department of Developmental and Educational Psychology, Universidad Nacional deEducación a Distancia (UNED), Madrid, Spain2Department of Basic Psychology, Universidad Autónoma de Madrid, Madrid, Spain

Although absolute pitch (AP) is a rare skill in typical development, individuals with Williams syn-drome (WS) are often referred to as possessing this musical ability. However, there is paucity ofresearch on the topic. In this article, 2 studies were conducted to evaluate AP in WS. In Study 1, sevenmusically trained individuals with WS, 14 musically trained typically developing controls matchedfor chronological age, and 2 experienced musicians with AP completed a pitch-identification task.Although the task was a classical assessment of AP, it required participants to have musical knowledge,and the availability and accessibility of musically trained individuals with WS is very low. In Study 2,a paradigm suitable for evaluating AP in individuals without musical training was used, which madeit possible to evaluate a larger group of participants with WS. A pitch memory test for isolated toneswas presented to 27 individuals with WS, 54 typically developing peers matched for chronologicalage, and the 2 musicians with AP. Both individuals with WS and their controls obtained low results inthe two studies. They showed an arbitrary pattern of response, and their performance was far from thatof musicians with AP. Therefore, participants with WS did not appear to possess AP. Unlike what isusually claimed, results suggest that AP is not a remarkable ability in WS and that, as in the typicallydeveloping population, this musical ability is also rare in individuals with WS.

Keywords: Williams syndrome; Absolute pitch; Pitch memory; Pitch identification; Musical skills.

Williams syndrome (WS) is a neurodevelopmental disorder caused by a microdeletion onchromosome 7 (band 7q11.23) (Ewart et al., 1993) and with an incidence ranging from1 in 20,000 to 1 in 7,500 live births (Morris, Demsey, Leonard, Dilts, & Blackburn,1988; Strømme, Bjørnstad, & Ramstad, 2002). Along with mild-to-severe general intel-lectual disability, individuals with WS present an uneven cognitive profile (Bellugi, Wang,& Jernigan, 1994; Mervis & John, 2010; Mervis, Morris, Bertrand, & Robinson, 1999;

This research was funded by a grant to the first author from the Ministry of Education and Science ofthe Spanish Government (AP2003-5098). The manuscript has been proofread thanks to funds from the plan forpromoting research in the Universidad Nacional de Educación a Distancia. We would also like to thank theWilliams Syndrome Association of Spain and the parents and participants who collaborated in this research.

Address correspondence to Pastora Martínez-Castilla, Universidad Nacional de Educación a Distancia(UNED), Department of Developmental and Educational Psychology, Faculty of Psychology, C/ Juan del Rosal,n◦ 10, Madrid, 28040, Spain. E-mail: pastora.martinez@psi.uned.es

© 2011 Psychology Press, an imprint of the Taylor & Francis Group, an Informa business

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2 P. MARTÍNEZ-CASTILLA ET AL.

Udwin & Yule, 1991). Although they exhibit significant impairments in visuospatial con-struction, numerical cognition, planning, and problem solving, they also present relativelygood functioning abilities regarding auditory rote memory, face processing, language, andsociability (e.g., Bellugi, Lichtenberger, Jones, Lai, & George, 2000; Mervis et al., 2000).This unusual pattern of weaknesses and strengths in higher cognitive processes has beentaken as evidence to support theories that claim the existence of innate and independentlyfunctioning cognitive modules (e.g., Pinker, 1994, 1999).

Music has also been included among the strengths of the WS cognitive profile. Thus,individuals with WS are traditionally described as having innate and outstanding musicalskills (e.g., Lenhoff, 1998; Levitin et al., 2004). Music teachers and relatives of individualswith WS have pointed out their musical interests and abilities (e.g., Dykens, Rosner, Ly, &Sagun, 2005; Lenhoff, 1996, 1998; Lenhoff, Wang, Greenberg, & Bellugi, 1997; Levitin& Bellugi, 1998; Levitin et al., 2004; Stambaugh, 1996). In addition to these reports, aca-demic research has also contributed to including music among the phenotypic strengthsin WS (Bellugi et al., 2000). Rhythmic reproduction and pitch discrimination skills ofindividuals with WS have been found to be equivalent to those of younger typically devel-oping children matched for mental age (Don, Schellenberg, & Rourke, 1999; Levitin &Bellugi, 1998). Moreover, it has been reported that individuals with WS perform as well astypically developing individuals matched for chronological age (CA) on timbral identifica-tion, rhythm discrimination, and perception of expressive phrasing and melodic elaboration(Hopyan, Dennis, Weksberg, & Cytrynbaum, 2001; Levitin & Bellugi, 2006).

However, research on musical abilities in WS has also provided results that bringinto question the idea that individuals with WS have enhanced musical skills. Impairmentsfor discriminating pitch and rhythm and for identifying the emotional resonance of musicalexcerpts have been found in WS (Don et al., 1999; Hopyan et al., 2001). Singing accuracyproblems have also been reported in individuals with WS (Martínez-Castilla & Sotillo,2008). In addition, the existence of a musical global integration deficiency has been sug-gested (Deruelle, Schön, Rondan, & Mancini, 2005). Finally, individuals with WS havebeen found to perform lower than typically developing peers matched for CA on differentmusical tasks, including tonal and rhythmic memory, and melody and rhythm reproduction(Martens, Reutens, & Wilson, 2010; Martínez-Castilla, Sotillo, & Campos, 2011).

Regardless of the previous results, it is generally accepted that, in spite of theircognitive deficits, individuals with WS present remarkable musical skills (e.g., Lenhoff,2006; Levitin, 2005; Levitin & Bellugi, 2006; McPherson & Hallam, 2009). Furthermore,findings that support the outstanding musical abilities of individuals with WS have beeninterpreted as evidence that in WS basic auditory processing could be intact and that musi-cal ability is preserved, or relatively preserved, and constitutes an independent functioningmodule (Don et al., 1999; Levitin & Bellugi, 1998; Levitin et al., 2004). Results regardingabsolute pitch (AP) have contributed to this view.

AP is referred to as the ability to identify or produce musical pitch without anyreference tones and it occurs in only 1 in 10,000 people (e.g., Miyazaki, 1988; Takeuchi& Hulse, 1993; Ward, 1999). Although most musical tasks do not require the possessionof AP, it is generally considered a useful and desirable ability for musicians (Takeuchi& Hulse, 1993). Furthermore, this rare ability has sometimes been considered a sign ofmusical talent and has often been described as an amazing or exceptional gift (Chin, 2003;Deutsch, 2002; Levitin & Rogers, 2005). For more than a century, AP has awakened theinterest of researchers, who have tried to explain the reasons why only some people possessthis ability (Chin, 2003; Takeuchi & Hulse, 1993). Although the way AP is acquired is still

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ABSOLUTE PITCH IN WS 3

unclear, this ability seems to arise from the interaction of genetic and environmental factorsduring development (Baharloo, Johnson, Service, Gitschier, & Freimer, 1998; Baharloo,Service, Risch, Gitschier, & Freimer, 2000; Zatorre, 2003). This has led researchers toconsider AP as an interesting model for investigating the general mechanisms of neuraldevelopment and cognition (Zatorre, 2003).

Lenhoff, Perales, and Hickok (2001a, 2001b) found noteworthy abilities for AP inWS. These authors evaluated this skill in 5 individuals with WS recruited from a musiccamp. All participants had received prior musical training and therefore knew the namesof musical tones. Participants were asked to identify notes presented with a piano or a key-board set to piano tones. Their performance was near ceiling (mean = 97.5%). From theseresults, Lenhoff et al. (2001a, 2001b) suggested that the incidence of AP in individualswith WS could be higher than in the typically developing population. Specifically, consid-ering that the number of individuals with WS in the United States and Canada is estimatedat 4,500 and that the 5 study participants had AP, the incidence of AP in WS would beapproximately 1/1,000 (Lenhoff et al., 2001b). This rate would be 10 times greater thanthe rate suggested for the typically developing population (Takeuchi & Hulse, 1993; Ward,1999). Lenhoff et al. (2001b) also noted that as more individuals with WS study music,learn to name the notes and are eventually evaluated, the incidence will be even higher.

Furthermore, Lenhoff et al. (2001a, 2001b) noticed that 4 of the 5 participants withWS began their musical training after the age of 6, despite the general assumption thatindividuals who possess AP started their musical training between the ages of 3 and 6(Baharloo et al., 1998; Levitin & Zatorre, 2003; Russo, Windell, & Cuddy, 2003; Takeuchi& Hulse, 1993). This observation led the authors to suggest that the typical early childhoodcritical period for AP acquisition may be extended in individuals with WS. The authors alsosuggested that individuals with WS could have AP abilities comparable to or even higherthan those of musicians with AP. As a whole, Lenhoff et al. (2001a, 2001b) concludedthat individuals with WS have exceptional abilities for AP, which would support the viewthat they possess musical intelligence related to pitch (Gardner, 1983). The fact that thismusical ability seemed to be excellent in the face of serious limitations in other domainswas interpreted by the authors as evidence of cognitive modularity (Lenhoff et al., 2001b).

Neuroanatomical studies have also provided data regarding AP in WS. However,results have been contradictory. Thus, the neuroanatomical correlate of AP observed in thetypically developing population has only been found in WS in one study (Hickok, Bellugi,& Jones, 1995). In typically developing individuals, this ability has been associated withdifferences in brain structures related to auditory processing. Specifically, professionalmusicians with AP show stronger leftward planum temporale asymmetry than nonmusi-cians or musicians without AP (Schlaug, Janke, Huang, & Steinmetz, 1995). This increasedleftward asymmetry seems to be due to a smaller-than-average right planum temporale andcould be genetically determined (Keenan, Thangaraj, Halpern, & Schlaug, 2001; Wilson,Lusher, Wan, Dudgeon, & Reutens, 2009). It is important to note that Hickok et al. (1995)found the same neuroanatomical asymmetry in 3 out of a sample of 4 participants withWS, although to a lesser extent than in musicians with AP. This neuroanatomical findinghas therefore been considered as further support for the idea that individuals with WS pos-sess AP. It has also led to suggesting that in individuals with WS musical abilities couldconstitute a neuromodule (Sacks, 1995).

Nevertheless, other research has not found the neuroanatomical correlate of AP inindividuals with WS. In postmortem examinations, Galaburda and Bellugi (2000) didnot find increased leftward planum temporale asymmetry in 2 out of 4 cases analyzed.

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4 P. MARTÍNEZ-CASTILLA ET AL.

Furthermore, no asymmetry was found in such a brain structure in WS, which differs frombrains of typically developing individuals. In a subsequent study, Eckert et al. (2006) tookneuroanatomical magnetic resonance images from a large group of 42 individuals with WS.They observed a pattern opposite the one described by Hickok et al. (1995). Eckert et al.reported significantly reduced leftward asymmetry of the planum temporale in WS com-pared with typically developing individuals matched for CA. This reduction was related toan increased right planum temporale. These results have been replicated by Chiang et al.(2007).

It should also be considered that the only behavioral study in which AP was evaluatedin WS included only 5 participants (Lenhoff et al., 2001a). Therefore, its conclusions thatindividuals with WS possess excellent AP abilities may be restricted by its small samplesize. This limitation is a frequent problem with investigations of a rare disorder such asWS because the syndrome’s low incidence makes it difficult to recruit a larger number ofparticipants. This difficulty is even more pronounced in studies focused on AP becausethe classical assessment of this skill involves labeling pitches, which requires participantsto have knowledge of musical nomenclature (Lenhoff et al., 2001a). The availability andaccessibility of individuals with WS who also have musical training is very low.

Although classical tests of AP ask participants to label tones (or to produce musicalnotes by names) (Takeuchi & Hulse, 1993), AP involves the ability not only to namemusical tones but also to encode durable long-term representations of target stimuli (Ross,Gore, & Marks, 2005; Ross, Olson, Marks, & Gore, 2004). Thus, the two-componentmodel of AP proposes that AP consists of long-term pitch memory and pitch labeling(Levitin, 1994; Levitin & Rogers, 2005, Zatorre, 2003). Following on from this view, adifferent paradigm for testing AP — specifically, for testing the ability to encode long-termrepresentations of pitch — without requiring subjects to know or use conventional musicalnomenclature has been successfully used in the literature (e.g., Bachem, 1954; Ross et al.,2004; Ross & Marks, 2009; Ross, Olson, & Gore, 2003; Siegel, 1974; Takeuchi & Hulse,1993). This paradigm tests pitch memory for isolated tones by giving participants a targetto remember, followed by a long delay or an interval of tonal interference that destroystheir short-term trace of the stimulus. It is important to consider that AP possessors andnonpossessors perform similarly on tasks that involve echoic or short-term memory butthat only the former are able to encode and maintain an accurate long-term representationof pitch chroma (Bachem, 1954; Siegel, 1974). Therefore, because the paradigm describedcancels out the possibility of having participants using a short-term trace of the tone andforces them to rely on durable representations of the stimuli, it allows for distinguishingbetween AP possessors and nonpossessors (Ross et al., 2004). Two tasks have beendeveloped under this paradigm: a task in which participants have to discriminate the targetfrom a comparison tone, and one in which they have to reproduce the target by adjustingan oscillator (Takeuchi & Hulse, 1993). Importantly, as explained, this paradigm does notrequire the labeling of tones, making it possible to assess participants who do not havemusical training.

As previously mentioned, evaluating AP in WS with classical tests is problematicbecause it is difficult to find individuals with WS who have musical knowledge. However,a paradigm focused on assessing pitch memory, such as the one previously described, issuitable for testing AP in individuals with WS who do not have musical training. In turn,this paradigm allows for AP assessment in a larger sample of participants. Thus, it shouldbe noted that, although it is generally assumed that individuals with WS are AP possessorsand that this assumption has contributed to the idea that they have special musical skills

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ABSOLUTE PITCH IN WS 5

and, consequently, to claims of modularity (e.g., Sacks, 1995; Semel & Rosner, 2003), APhas only been assessed in a small sample of individuals with WS (Lenhoff et al., 2001a,2001b). Therefore, an evaluation of AP in a larger number of participants with WS seemsto be a valuable contribution to the literature.

The current article assessed AP in WS using both a classical task and the previouslydescribed paradigm in order to ascertain whether individuals with WS possess exceptionalAP abilities, as claimed in the literature, or whether, on the contrary, the results found byLenhoff et al. (2001a, 2001b) cannot be generalized to the population of individuals withWS. Two studies were conducted. In the first study, 7 participants with WS with musicaltraining were assessed with a pitch-identification task, that is, labeling tones. In the secondstudy, a larger group of 27 participants with WS was assessed using a pitch memory taskfor isolated tones.

STUDY 1

Method

Participants. The sample included 7 individuals with WS (age range: 15 to32 years) and a control group (CG) of 14 typically developing peers. All participants withWS exhibited the clinical features of the WS phenotype (e.g., Bellugi et al., 2000). In addi-tion, all of them had a positive FISH (fluorescent in situ hybridization) test to confirm genedeletion and the WS diagnosis. It is considered that conclusions about the excellent musi-cal abilities of individuals with WS can only be inferred when their musical performanceis compared with that of typically developing individuals matched for CA (Hopyan et al.,2001). Thus, this is the procedure that should be used when evaluating the claim that, withinthe cognitive profile of individuals with developmental disorders, a specific ability consti-tutes a strength in absolute terms (e.g., Karmiloff-Smith, Brown, Grice, & Paterson, 2003;Karmiloff-Smith & Thomas, 2003). This is the case of the abilities for AP in WS since theyhave been claimed to be exceptional (Lenhoff et al., 2001a, 2001b). As aforementioned,the aim of this study was to evaluate this claim. Therefore, the WS group and the CG wereindividually matched for CA. Consequently, no significant differences in CA were foundbetween the groups (p = .91). In addition to the WS group and the CG, two experiencedmusic students (14 and 16.4 years old) who claimed to be AP possessors participated inthe study as a standard for comparison. As reported by parents, no participants sufferedfrom hearing impairment or had any other clinical diagnosis. Participants with WS wererecruited through the Williams Syndrome Association of Spain. Control participants wererecruited through education centers (schools and universities) and other typical settings(work and neighbors), and the music students were recruited through a conservatory.

The cognitive levels of participants in both the WS group and the CG were evalu-ated. The Escala de inteligencia de Wechsler para adultos III (Wechsler Intelligence Scalefor Adults-III [WAIS-III]; Wechsler, 1970/2001) was administered to participants aged17 years and older, and the Escala de inteligencia de Wechsler para niños IV (WechslerIntelligence Scale for Children-IV [WISC-IV]; Wechsler, 1974/2005) was used for theyounger participants. Intelligence quotients (IQs) in the WS group were significantly lowerthan those in the CG in terms of full-scale IQ, verbal IQ, and performance IQ (p < .001 forall comparisons). Table 1 shows the descriptive characteristics of both the WS group andthe CG.

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6 P. MARTÍNEZ-CASTILLA ET AL.

Table 1 Descriptive Characteristics of the WS Group and the CG of Study 1.

WS group CG

n 7 14Gender (M/F) 3/4 5/9Mean CA 21.96 (6.8) 21.49 (5.7)Full-scale IQ 51.14 (4.41) 117.50 (8.39)Verbal IQ 58 (4.44) 115.64 (7.73)Performance IQ 53.14 (4.06) 114.64 (9.64)

Note. Values in parentheses represent standard deviations.

All participants, both in the WS group and the CG, had received prior musical train-ing and therefore all of them had knowledge of musical nomenclature. Due to the specialeducation needs of individuals with WS in the field of music (Stambaugh, 1996), it wasimpossible to match the groups exactly with regard to musical training. However, the musi-cal training levels in the WS group and the CG were intended to be relatively homogeneous.Following on from this, because no participants with WS had received musical training inthe formal context of a conservatory, we explicitly avoided including participants withformal conservatory training in the CG. Therefore, the musical training of the control par-ticipants was mainly informal. The characteristics of the participants’ musical training,including that of the experienced music students who claimed to possess AP, are shown inTable 2.

Task and Procedure. Participants were presented with a pitch-identification task,the most commonly used method of assessing AP (Takeuchi & Hulse, 1993). Specifically,they were asked to label a total of 18 pitches without having previous reference tones.No feedback was given after participants’ answers to avoid the use of relative pitch cueswhen identifying the pitches. The tones corresponded to notes from the Western equal-tempered scale (A4 = 440), ranging from A3 to F#5. They were played with a tunedSamick piano and were digitally recorded (sampling frequency of 22.05 KHz) with a lap-top (HP, Intel Pentium M Processor 1.60 GHz 800 MHz, SoundMAX Integrated DigitalAudio sound card). All of them had a duration of 1 second and an intensity of 70 dB, aschecked and modified, if necessary, with PRAAT (Boersma & Weenink, 2004).

Only chroma, but not octave information, was taken into account when calculatingparticipants’ accuracy in the task. Octave information was ruled out because AP possessorsmake octave errors (e.g., Bachem, 1937, 1955; Lockhead & Byrd, 1981). As performed byLenhoff et al. (2001a, 2001b), three-quarters of a point was given for answers that wereoff by a semitone. This criterion was adopted to solve the possible problem of identifyingAP in individuals who consistently err by a semitone in the same direction (Bermudez &Zatorre, 2009; Takeuchi & Hulse, 1993). Responses differing by more than a semitone weregiven zero points, while correct chroma responses were given one point. The percentageof correct responses was calculated on the basis of these scoring criteria. As suggested byBermudez and Zatorre, mean absolute deviation was also used as a measure of AP identifi-cation accuracy. This measure was calculated by taking the absolute value of the semitonedeviation from the correct answer, after having discarded the octave information, and thenobtaining the mean of these values across all trials for each participant. Because the possi-ble range of this measure is 0 to 6 semitones, a mean absolute deviation of 0 corresponds to

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ABSOLUTE PITCH IN WS 7

Tabl

e2

Cha

ract

eris

tics

ofPa

rtic

ipan

ts’

Mus

ical

Tra

inin

gin

the

WS

Gro

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Stud

ents

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Poss

ess

AP.

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ents

n7

142

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trai

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102–

109

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ean

6.21

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1)4.

76(2

.32)

9(0

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Age

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208–

245–

6

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12.6

9(4

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1)

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8 P. MARTÍNEZ-CASTILLA ET AL.

perfect AP identification, while a value of 3 indicates a perfectly random response pattern(Bermudez & Zatorre, 2009).

All participants were individually assessed. The task was presented to participantsat a comfortable listening level via the laptop used for recording the tones. Participantswith WS were assessed in a quiet room of their home. Control participants were assessedin their residences, the experimenter’s residence, or in a room in their education centers,depending on the way in which they had been contacted. The experienced music studentswere evaluated in a room at their conservatory.

Statistical analyses were performed with SPSS 14.0.

Results

Means and standard deviations of the two variables calculated to measure the accu-racy of AP identification in the WS group and the CG are shown in Table 3. Scores obtainedby the experienced music students who claimed to possess AP are also reported for descrip-tive comparison purposes. As shown in Table 3, the percentage of correct responses wasgenerally low both in the WS and the control groups. However, the self-reported AP pos-sessors made a nearly perfect identification of chroma. With respect to mean absolutedeviation, the WS group and the CG obtained values near the cutoff point for a com-pletely random response pattern, whereas the self-reported AP possessors showed nearlyno deviation from the correct answer.

Figure 1 shows histograms of semitone deviation from the correct response, afteroctave information was discarded. Because a deviation of -6 semitones yields the samepitch class as a deviation of +6 semitones, as done in previous research (Levitin, 1994),errors of ± 6 semitones were distributed evenly between the two extreme categories. Whileself-reported AP possessors showed the expected distribution around the exactly correctresponses, both the WS and control groups presented arbitrary responses distributed at alldistances from the correct tone.

To compare the WS groups and the CG, Mann-Whitney tests were conducted for thepercentage of correct responses and for the mean absolute deviation (scores obtained bythe self-reported possessors were not included in these analyses due to the small samplesize of this group). No significant differences were found between groups in any of thesevariables (p > .05). Correlations between variables of accuracy of AP identification andparticipants’ IQs were also calculated. No significant correlation was found (p > .05).

Table 3 Mean Scores (SD) of Accuracy Measurements of AP Identification.

WS group CG

Experienced musicstudents

(self-reported APpossessors)

Percentage of correctresponses

28.57 (7.08) 26.19 (8.06) 97.92 (2.95)

Mean absolute deviation 2.76 (0.46) 2.72 (0.41) 0.08 (0.12)

Note. Values in parentheses represent standard deviations.

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ABSOLUTE PITCH IN WS 9

Self-identified AP possessors

0

20

40

60

80

100

Semitone Deviation

Per

cent

age

of R

espo

nses

WS group

0

20

40

60

80

100

Semitone Deviation

Per

cent

age

of R

espo

nses

CG

0

20

40

60

80

100

–6 –5 –4 –3 –2 –1 0 1 2 3 4 5 6

–6 –5 –4 –3 –2 –1 0 1 2 3 4 5 6

–6 –5 –4 –3 –2 –1 0 1 2 3 4 5 6Semitone Deviation

Per

cent

age

of R

espo

nses

Figure 1 Histograms of semitone deviations from the correct response for each group (octave informationdiscarded).

Discussion

Individuals with WS obtained a very low percentage of correct answers (less than30% correct). In addition, their mean absolute deviation showed a random response pat-tern (approximately three semitones). This pattern of response was also shown when thedistribution of semitone deviation was examined. These results contrast with those typi-cally obtained by AP possessors, who score over 90% correct with deviations less thanone semitone when presented with a pitch-labeling task (Bermudez & Zatorre, 2009; Chin,2003; Miyazaki, 1988). The two self-reported AP possessors in this study scored approxi-mately 98% correct and showed almost no deviation from the correct response. Therefore,in contrast to the conclusions obtained by Lenhoff et al. (2001a), results from this study

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10 P. MARTÍNEZ-CASTILLA ET AL.

suggest that AP abilities of individuals with WS are not comparable to those of musicianswith AP. Furthermore, our results suggest that individuals with WS do not possess AP.

It should be noted that the low results obtained by participants with WS cannotbe accounted for by their cognitive deficits because no significant correlation was foundbetween the accuracy of AP identification and the participants’ IQs. Moreover, the CGalso obtained low results. In fact, the pattern of results found for the CG was the sameas that found for the WS group. Thereby, as for individuals with WS, the percentage ofcorrect answers obtained by control participants was well below the level reached by APpossessors (e.g., Chin, 2003). In addition, control participants also responded randomly,as shown by the mean absolute deviation and the distribution of semitone deviation. Thus,although there were slight differences in the results found for the WS group and the CG(individuals with WS obtained a slightly higher percentage of correct answers than theircontrol peers, but the CG showed a slightly lower mean absolute deviation than the WSgroup), these differences were not significant. Far from indicating proficiency for identify-ing tones without a reference pitch, these results would suggest that neither the CG nor theWS group in this study possessed AP. The low incidence of this ability in typically devel-oping individuals could explain why participants of the CG did not present AP (Bachem,1955; Takeuchi & Hulse, 1993; Ward, 1999). It should also be considered that the con-trol participants started their musical training late (between 8 and 24 years), beyond thecritical period for acquiring AP (Baharloo et al., 1998; Levitin & Zatorre, 2003; Russoet al., 2003; Takeuchi & Hulse, 1993). In addition, their musical training was generallyinformal. Therefore, it seems unlikely that control participants received the type of musi-cal training that may lead to AP, that is, musical training focused on making associationsbetween particular auditory stimuli and their respective category labels (Levitin & Zatorre,2003). Individuals with WS also received late musical training. Furthermore, consideringtheir cognitive profile and general difficulties with learning music (Stambaugh, 1996), it isalso unlikely that individuals with WS received musical training focused on associationsbetween tones and their labels. Therefore, as for the CG, both the age at which participantswith WS commenced their musical training and the type of musical training they receivedcould explain why the individuals with WS in this study did not possess AP. Nevertheless,it should be noted that the fact that individuals with WS with late musical training did notacquire AP would not support the conclusions of Lenhoff et al. (2001a, 2001b) regardingan extended critical period in WS for acquiring AP.

In short, results of the current study are strikingly different from those obtainedby Lenhoff et al. (2001a, 2001b). These differences cannot be explained by methodol-ogy because a similar approach was used in both studies: The scoring criterion was thesame, and the tones were administered with a piano in both studies; although a keyboardset to piano tones was also used in some cases in Lenhoff et al.’s study (2001a, 2001b).Regardless, the use of piano should have facilitated the task because this is the easiestinstrument in which to identify pitch (Bachem, 1937; Lockhead & Byrd, 1981; Takeuchi& Hulse, 1993). The pitch register used, however, was different in both studies. Whilein Lenhoff et al.’s study, pitches were chosen from Octaves 2 through 6, in the currentstudy, pitches were presented from the central pitch register. Nevertheless, it should benoted that pitch identification is more accurate in the central pitch register (Bachem, 1937;Takeuchi & Hulse, 1993). Therefore, the use of an easier pitch register to identify pitchcannot explain the lower results obtained by participants with WS in this study. On thecontrary, with an easier task, a higher performance would have been expected. The numberof items administered also differed between the two studies. However, it is unlikely that this

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variable accounts for the striking differences found in the studies’ results. In fact, assumingthat individuals with WS have exceptional AP abilities (Lenhoff et al., 2001a, 2001b), it isdifficult to explain why the participants with WS in the present study had low scores on atask with a timbre and pitch register that would have made pitch identification easier forAP possessors (Bachem, 1937; Lockhead & Byrd, 1981; Takeuchi & Hulse, 1993).

Differences in the participants’ musical training may instead explain why individualswith WS in Lenhoff et al.’s (2001a, 2001b) study showed AP, while participants withWS in the current study did not. Detailed information on the musical training received byparticipants with WS of the study conducted by Lenhoff et al. (2001a, 2001b) was notavailable. Thus, it may be that participants with WS in Lenhoff et al.’s (2001a, 2001b)study received more musical training focused on making associations between auditorystimuli and their category labels; that is, the type of musical training that may lead toacquiring AP (Levitin & Zatorre, 2003).

It is also important to consider that, despite the classical homogenous descriptionsof the WS cognitive profile, heterogeneity in cognitive functions within WS has also beenreported (e.g., Pezzini, Vicari, Volterra, Milani, & Ossella, 1999; Porter & Coltheart, 2005).Therefore, the variability associated with WS may also explain the differences between thestudies. This variable becomes more important when considering the small sample sizeof the two studies. In turn, the small sample size hinders the generalization of results.As previously mentioned, this restriction was motivated by the difficulty of finding indi-viduals with WS who know musical nomenclature and can perform a classical AP taskin which pitch labeling is required. With the aim of addressing this limitation and furthercontributing to our knowledge regarding whether individuals with WS possess exceptionalAP abilities, we conducted a second study that used a paradigm suitable for testing AP inindividuals with WS who had no musical knowledge.

STUDY 2

Method

Participants. Individuals with WS, typically developing individuals of the CGand experienced music students who took part in Study 1 also participated in this study.Additionally, the sample of Study 2 was enlarged to include individuals without any musi-cal training: 20 additional individuals with WS (aged 12 to 32 years) and 40 typicallydeveloping individuals matched for CA. The rationale of the matching criterion was thesame as that used in the previous study, and no significant differences were found in thisvariable (p = .95). Therefore, there were four groups of participants: musically trained indi-viduals with WS (n = 7), musically untrained individuals with WS (n = 20), a musicallytrained CG (n = 14) and a musically untrained CG (n = 40). In addition to these, as alreadymentioned, the two music students with AP from Study 1 also participated in Study 2.Musically untrained participants in the WS group and the CG were recruited through thesame settings as those described in Study 1. All participants with WS presented the clin-ical phenotype of the syndrome. They also had a genetic diagnosis confirmed by FISH.As reported by parents, none of the new participants, either in the WS group or the CG,suffered from hearing impairment or had any other clinical diagnosis. The cognitive levelof all musically untrained participants was evaluated with the Wechsler scales, as in theprevious study. Full-scale IQ, verbal IQ, and performance IQ were significantly lower inindividuals with WS than in their control peers (p < .001 for all comparisons). Descriptive

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Table 4 Descriptive Characteristics of the Musically Untrained Participants in Study 2.

WS group CG

n 20 40Gender (M/F) 10/10 23/17Mean CA 19.55 (5.94) 19.59 (6.09)Full-scale IQ 50.65 (5.28) 110.63 (9.44)Verbal IQ 58.55 (6.57) 110.53 (8.52)Performance IQ 53.00 (6.17) 106.45 (9.49)

Note. Values in parentheses represent standard deviations.

characteristics of the new participants (i.e., the musically untrained participants) who tookpart in this study are shown in Table 4.

Task and Procedure. The task of this study was designed according to the pre-viously described paradigm for assessing AP in individuals without musical training.Considering the visuospatial difficulties of individuals with WS (e.g., Bellugi, Sabo, &Vaid, 1988; Farran & Jarrold, 2003; Mervis et al., 1999), a pitch memory task in whichparticipants had to adjust an oscillator after being presented with a tone was ruled out.Instead, a memory task for isolated tones was used. In the task, participants were presentedwith an isolated tone, followed by a distracting melody. Another isolated tone was thenpresented. Participants were asked to discriminate whether the two tones were the sameor different. In short, participants had to make same/different judgments about a targetstimulus after a retention interval filled with interfering melodies.

The task consisted of two examples, two practice items and 16 experimental items.Half the items were the same and half of them were different. For the different items, thestandard and comparison tones were one semitone apart. The isolated tones were notesfrom the Western equal-tempered scale (A4 = 440) ranging from A#3 to F5 and with aduration of 1 second. All were produced as tones in Study 1. The distracting melodieswere excerpts from recording of famous piano pieces that lasted between 8.7 and 10.8 sec-onds. For the interstimulus interval, melodies were preferred to randomly selected tonesto make the task more attractive to participants; however, to prevent the perception of amelody’s tonality biasing the task, none of the isolated tones was the tonic of the inter-fering melody. There was also 2 seconds of silence between each isolated tone and thedistracting piano melody. Therefore, the total interstimulus interval ranged from 12.7 to14.8 seconds. PRAAT (Boersma & Weenink, 2004) was used to concatenate the materialsfor each item (i.e., tone - silence - distracting melody - silence - tone).

As previously mentioned, AP possessors and nonpossessors differ in their abilitiesto encode and maintain long-term representations of tones, but not in their short-termmemory for pitch (Bachem, 1954; Ross et al., 2004; Siegel, 1974). Consequently, AP pos-sessors perform significantly better than nonpossessors on tasks of memory for isolatedtones when the short-term trace of the tones is destroyed, forcing participants to rely onlong-term representations of the stimuli (Ross et al., 2004). The higher performance ofAP possessors compared to nonpossessors on pitch memory tests for isolated tones hasbeen reported at interfering intervals of 10 to 15 seconds with a 1 semitone differencebetween tones (Siegel, 1974). As aforementioned, the same conditions were used in thecurrent study, which ensured that the task was able to distinguish between AP possessorsand nonpossessors.

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Table 5 Results Obtained by Each Group in the Pitch Memory Task for Isolated Tones.

Group MeanStandarddeviation

Musically untrained Williams syndrome group 54.38 9.75Musically trained Williams syndrome group 50 8.07Musically untrained control group 55.31 13.32Musically trained control group 60.27 14.21Music students with AP 87.5 0

General procedures regarding assessment conditions and data analysis were the sameas those used in Study 1.

RESULTS

Percentage of correct responses was calculated for each group. Results for the musi-cally trained and untrained individuals with WS and their control peers are shown inTable 5. Scores obtained by music students with AP are also reported for descriptive com-parison purposes. As shown in Table 5, both musically trained and musically untrainedparticipants with WS performed at chance. The same pattern was found for the two controlgroups. In contrast, the music students with AP scored well above chance.

Although the groups’ performances were very similar, a 2 × 2 analysis of vari-ance (ANOVA) with diagnosis group (WS group vs. CG) and musical training (musicallytrained vs. musically untrained) as between-subject factors was run to test for possibledifferences between groups, (as in Study 1, scores obtained by music students with APwere not considered for statistical analyses due to the small sample size of that group).Neither the principal effects nor the interaction between variables was statistically signif-icant (p > .05). Correlations between scores on the task and participants’ IQs were alsocalculated. No significant correlation was found (p > .05).

DISCUSSION

Participants with WS performed at chance in the memory task for isolated tones usedin this study. Their poor results contrast with those of the two music students with AP, whoobtained high results that were well above the chance level. As explained, the task requiredparticipants to encode and maintain long-term representations of pitch because only APpossessors are able to successfully use such abilities (Ross et al., 2004). Therefore, resultssuggest that individuals with WS do not possess AP, at least as referred to the ability toencode durable representations of tones.

Scores obtained by participants with WS were similar to those of the control partici-pants. No significant differences were found between groups. In addition, no significantcorrelation was found between the participants’ scores and their intelligence measure-ments. As in Study 1, this result suggests that the poor results of participants with WScannot be explained by their cognitive deficits. It also suggests that neither participants withWS nor their control peers could be considered AP possessors. These results are not sur-prising, considering the well-known rarity of AP (Bachem, 1955; Takeuchi & Hulse, 1993;Ward, 1999). Nor were significant differences found as a function of musical training.

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None of the participants who took part in Study 1 had AP. Therefore, this explains why themusical training variable showed no significant effects.

As already mentioned, results of Study 1 also led us to conclude that participants withWS were not AP possessors. Nevertheless, the study’s small sample size made it difficultto ascertain whether results should be generalized to other individuals with WS. However,the fact that the same type of results was found in Study 2, which had a considerably largersample, supports and strengthens our previous conclusions regarding the lack of AP in WS.From our point of view, results of Study 2 provide a significant contribution to the literaturebecause they represent the first time that AP has been assessed in a relatively large group ofindividuals with WS. This assessment was possible because of the use of a novel paradigmthat did not require participants to have any musical knowledge (Ross et al., 2004). Thisparadigm allowed us to solve the main problem associated with the use of classical tasksof AP in WS, that is, the difficulty of finding individuals with WS who can name musicaltones and, consequently, the small sample size of studies that use these tasks.

Finally, it should be noted that, although the paradigm focused on the ability toencode durable representations of chroma, we do not deny the possibility that the twomusic students with AP could have solved the task by using a verbal labeling strategy, thatis, they could have used the musical nomenclature to label the stimuli in order to main-tain representations of chroma. However, it should also be considered that AP possessorshave different encoding strategies for pitch and that these strategies are not limited to ver-bal labeling. Thus, AP possessors may use multiple codes (e.g., auditory, kinesthetic, andvisual imagery) for long-term representations of chroma (Zatorre & Beckett, 1989). Furtherstudies should therefore ascertain whether musically trained individuals with AP tend touse a verbal-labeling strategy when presented with tasks under the paradigm employed inthis study. In any case, it is important to note that the paradigm does not require partic-ipants to have any musical knowledge. In fact, this paradigm has already proven usefulfor identifying AP in nonmusician adults and in 5-year-old children with minimal musicexperience (Ross et al., 2003; Ross & Marks, 2009).

GENERAL DISCUSSION

In this section, Studies 1 and 2 are synthesized in order to provide a comprehensiveview of the article. Results are further discussed and conclusions are drawn.

This research aimed to ascertain whether individuals with WS possess exceptionalAP abilities. Two studies were conducted. In the first one, participants were presented witha classical AP task (labeling tones). In the second study, a pitch memory task for isolatedtones was used. Individuals with WS obtained low results in both studies. They showed anarbitrary pattern of response, and their performance was far from that of musicians with AP.Therefore, results from the two studies presented here lead us to conclude that individualswith WS do not possess exceptional AP abilities.

Only one previous study has evaluated AP in WS (Lenhoff et al., 2001a, 2001b). Thatstudy employed a procedure similar to the one used here in Study 1. However, results of thetwo studies were strikingly different. The sample size was small in both studies; therefore,no solid conclusions could be drawn from them. The difficulties of finding individualswith WS with musical training who can perform a classical AP task that requires toneidentification explained the small sample size of both Lenhoff et al.’s (2001a, 2001b) studyand Study 1 reported here. This limitation was solved in Study 2 by using an AP task thatdid not require participants to make use of any musical knowledge. This approach allowed

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us to evaluate a larger group of individuals with WS because both participants with andwithout musical training were able to complete the task. No participant with WS presentedAP in Study 2 either. Therefore, these results, obtained from a considerably larger groupof participants, provided further evidence against the idea that individuals with WS haveexcellent AP abilities.

As previously mentioned, individuals with WS present high variability in their cog-nitive functions (e.g., Pezzini et al., 1999; Porter & Coltheart, 2005). This variability haseven been shown in the neuroanatomical correlate associated with AP. Increased leftwardplanum temporale asymmetry has only been reported in a few cases, and many more indi-viduals with WS present reversed asymmetry or symmetrical plana (Eckert et al., 2006;Martens et al., 2010). Therefore, as observed with the neuroanatomical correlate of AP, itmay be that only a few individuals with WS possess AP. If this is the case, Lenhoff et al.’sparticipants with WS would not be representative of the majority of individuals with WS.Consequently, the results found for such participants should not be generalized to the entirepopulation of individuals with WS. In short, while Lenhoff et al. (2001a, 2001b) proposedthat the incidence of AP in individuals with WS is greater than that in the typically devel-oping population, we conservatively suggest that, as in the typically developing population,this ability is also rare in WS.

The question now arising would be why only a few individuals with WS possessAP, whereas most do not. The same has been asked for the typically developing popula-tion. However, despite extended research on this topic, the answer is still unclear (Chin,2003; Levitin & Rogers, 2005; Takeuchi & Hulse, 1993). Thus, the way AP is acquiredhas not been fully understood so far (Levitin & Rogers, 2005; Levitin & Zatorre, 2003;Zatorre, 2003). Early musical training seems to be necessary for the development of AP(Baharloo et al., 1998). The type of musical training received may also have an impact(Gregersen, Kowalsky, Kohn, & West Marvin, 2000). As previously mentioned, only amusical training focused on making associations between particular auditory stimuli andtheir respective category labels appears to lead to the acquisition of AP (Levitin & Zatorre,2003). However, although necessary, musical training seems to be not sufficient for thedevelopment of this musical skill (Baharloo et al., 1998; Levitin & Zatorre, 2003). Most ofthe people who receive early musical training do not develop AP (Gregersen et al., 2000;Zatorre, 2003). Apart from musical training, genetics may also play a role. Thus, the obser-vation that AP aggregates in families suggests a genetic basis for AP (Baharloo et al., 1998,2000; Gregersen, Kowalsky, & Li, 2007). Nevertheless, it remains unknown which ones arethe possible genes involved in AP, if they exist, and what they would code for (Levitin &Rogers, 2005; Zatorre, 2003). These genes might be related to the neuroanatomical dif-ferences found between AP possessors and nonpossessors (Keenan et al., 2001; Zatorre,2003). Even so, it is uncertain whether these neuroanatomical differences are a cause oran effect of AP (Levitin & Rogers, 2005). Therefore, there is controversy over the originsof AP. Yet, this musical ability has been considered to provide an opportunity to under-stand how the interaction between genes and environment results in the development of acognitive skill (Baharloo et al., 1998; Gregersen et al., 2000; Zatorre, 2003).

WS offers a privileged setting to study genes-environment interactions in thedevelopment of musical skills. In light of results of the two studies reported here we con-clude that AP is not a remarkable ability in WS. This conclusion contrasts with previousclaims that individuals with WS possess exceptional AP abilities (Lenhoff et al., 2001a,2001b). It is also important to consider that this claim is one of the bases for conceptualiz-ing individuals with WS as a population with outstanding musical skills. In turn, the idea

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that individuals with WS present preserved or enhanced musical abilities in the face of seri-ous impairments in other cognitive domains has led to supporting claims of modularity notonly in WS but also in the typically developing population (Lenhoff et al., 2001a, 2001b;Levitin & Bellugi, 1998; Sacks, 1995). As previously mentioned, data from the currentstudy do not support that individuals with WS possess excellent AP abilities. Therefore,importantly, in view of our results, both the idea of preserved or exceptional musical skillsin WS and the claims regarding the modularity of the musical domain should be broughtinto question.

Original manuscript received May 16, 2011Revised manuscript accepted October 29, 2011

First published online December 6, 2011

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