- Original research
- Open Access
Aetiology of auditory dysfunction in amusia: a systematic review
© Casey; licensee BioMed Central Ltd. 2013
Received: 10 December 2012
Accepted: 19 April 2013
Published: 24 April 2013
Amusia, a music-specific agnosia, is a disorder of pitch interval analysis and pitch direction change recognition which results in a deficit in musical ability. The full range of aetiological factors which cause this condition is unknown, as is each cause’s frequency. The objective of this study was to identify all causes of amusia, and to measure each of their frequencies.
Design: systematic review was conducted by search of multiple databases for articles related to the aetiology of amusic auditory dysfunction. The Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines for reporting of systematic reviews were followed, utilizing the PRISMA checklist and PRISMA flowchart methodology. Setting: Retrospective medical database review. Main outcome measures: evidence yielded from the systematic review process.
The initial search protocol identified 5723 articles. Application of a classification review filter and exclusion of irrelevant or duplicates led to the initial identification of 56 relevant studies which detailed 301 patients. However, these studies were of poor quality. Because of this, synthesis and statistical analysis were not appropriate.
Although initially a large number of relevant studies were identified, and might point in future to potential diagnostic categories, it was not appropriate to synthesise and analyse them due to poor quality, considerable heterogeneity and small numbers. This suggests that large, high quality studies focussed directly on understanding the aetiology of amusia are required.
Amusia is a music-specific auditory agnosia consisting of neurological deficit in musical ability . Knoblauch’s definition is ‘the loss of a musical ability, such as the comprehension of music, the production of music, or the ability to read or write musical notation’ .
However, a grey area lies between ‘amusia’ and auditory agnosias which impair auditory function related to pitch processing, interval analysis, timbre, rhythm or the emotional components of music but with sparing of gross musical ability . This study will focus on studies in which there is a clear loss of musical ability and thus satisfy Knoblauch’s definition.
Fry estimates that 4.2% of the UK adult population may be amusic . As music and melody form a fundamental part of human experience, amusia is associated with significant distress, especially for musical professionals. Removal of a stimulus which in some instances can activate neuroanatomical regions associated with intense pleasure can represent a severe handicap .
Thus, a systematic examination of the aetiology of amusia may be of considerable importance in forming a differential diagnosis in the clinic and in informing prevention strategies. Understanding the aetiological factors behind amusia can also shed light on musical auditory function and aid in deficit prediction. The neural basis of music is a matter of intense investigation.
However, no such aetiological categorization has yet been conducted. Here, a systematic review of the causes of amusia is performed.
This systematic review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines for reporting of systematic reviews, utilizing the PRISMA checklist and PRISMA flowchart methodology .
Initial search protocol
Systematic review was conducted by search of multiple databases for articles related to the aetiology of amusic auditory dysfunction (Medline and The Cochrane Library). Key Terms used to identify the concept of amusic auditory dysfunction included amusia (sensory and motor), dysmusia, tone deaf(ness), tune deaf(ness), pitch processing deficit and note deaf(ness). Keywords used to identify aetiological frequency included aetiology, etiology, aetiologic(al) factor(s), aetiol*, etiol*, pathophysiology identification, risk factor(s) and cause(s). Aetiological keywords were combined with those specific to amusic auditory dysfunction. A secondary search was then conducted, and the grey literature was also searched.
Initial search using the amusic key-terms, with and without aetiological key-term modifiers, yielded 5723 articles for assessment. Articles to form part of the systematic review were then filtered based on a classification review filter. Studies to be included had to be directly relevant to amusia, contain a definition of amusia consistent with the current literature (i.e. music specific auditory agnosia), and provide a full aetiological explanation for all subjects with accompanying diagnostic evidence.
After elimination of irrelevant articles and application of the above criteria, results were narrowed to 56 papers containing 301 patients ranging from 1878 to 2012, including articles in English, German, Spanish, French, Italian, Russian and Japanese (non-English language articles were translated into English).
However, 40 of these 56 were single case reports; the remaining studies also contained small patient numbers (and/or had amusia as a secondary concern within the paper). This meant that the quality of evidence found was very poor. Due to this, no studies found were appropriate for synthesis and analysis.
However, none were suitable for synthesis and further analysis because they were poor quality with regards to small numbers. In addition, considerable heterogeneity precluded synthesis.
Although initially a large number of relevant studies were identified, and might point in future to potential diagnostic categories, it was not possible to synthesise them due to poor quality, considerable heterogeneity and small numbers.
40 of the 56 initially identified studies were single case reports: clearly, drawing conclusions from a synthesis of these would not be appropriate. The remaining studies also contained relatively small patient numbers. In addition, these studies were extremely heterogeneous. Studies came from a wide range of time periods, with different reporting methods.
On this basis, to answer the question of amusia aetiology, it is necessary in the future to design and implement large, high quality studies with agreed diagnostic criteria.
Implications for research and future hypotheses
Understanding and quantifying amusia aetiology would be useful for assessment of this condition in a clinical setting. This paper suggests that this will not be possible until relevant high quality evidence is produced.
In conclusion, due to the infrequent and irregular nature of the evidence related to amusia aetiology, all relevant studies suffer from small numbers and considerable heterogeneity. This, in addition to the other problems outlined above, suggests that large, high quality studies focussed directly on understanding the aetiology of amusia are required before a synthesis and analysis can take place.
- Pearce JM: Selected observations on amusia. Eur Neurol 2005,54(3):145–148.PubMedView ArticleGoogle Scholar
- Knoblauch A: Über Störungen der musikalischen Leistungsfähigkeit infolge von Gehirnläsionen. Deutsch Arch Klin Med 1888, 43:331–352.Google Scholar
- Stewart L, von Kriegstein K, Warren JD, Griffiths TD: Music and the brain: disorders of musical listening. Brain 2006,129(Pt 10):2533–2553.PubMedView ArticleGoogle Scholar
- Kalmus H, Fry DB: On tune deafness (Dysmelodia): frequency, development, genetics and musical background. Ann Hum Genet 1980,43(4):369–382.PubMedView ArticleGoogle Scholar
- Blood AJ, Zatorre RJ: Intensely pleasurable responses to music correlate with activity in brain regions implicated in reward and emotion. Proc Natl Acad Sci USA 2001,98(20):11818–11823.PubMedView ArticleGoogle Scholar
- Moher D, Liberati A, Tetzlaff J, Altman DG, PRISMA Group: Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. BMJ 2009, 339:b2535.PubMedView ArticleGoogle Scholar
- Allen G: Note-deafness. Mind 1878, 10:157–167.View ArticleGoogle Scholar
- Anonymous: Amusia. Br Med J 1894,2(1773):1441–1442.View ArticleGoogle Scholar
- Smith PB: Auditory Aphasia with Amusia. Br Med J 1897,1(1892):842–843.PubMedView ArticleGoogle Scholar
- Kohl GF, Tschabitscher H: A case of amusia. Wien Z Nervenheilkd Grenzgeb 1953,6(2–3):219–230.PubMedGoogle Scholar
- Scholer W: A case of pure word deafness with amusia. Monatsschr Ohrenheilkd Laryngorhino 1955,8(2):106–109.Google Scholar
- Fukuda K, Hasegawa T: A case of visual agnosia with amusia (author's transl). Seishin Shinkeigaku Zasshi 1976,78(5):417–430.PubMedGoogle Scholar
- Mazzucchi A, Marchini C, Budai R, Parma M: A case of receptive amusia with prominent timbre perception defect. J Neurol Neurosurg Psychiatry 1982,45(7):644–647.PubMedView ArticleGoogle Scholar
- Botez M, Wertheim N: Expressive aphasia and amusia following right frontal lesion in a right-handed man. Brain 1959, 82:186–202.PubMedView ArticleGoogle Scholar
- Wertheim N, Botez M: Receptive amusia: a clinical analysis. Brain 1961, 84:19–30.PubMedView ArticleGoogle Scholar
- Ulrich G, The syndrome of auditory agnosia: A case report and an attempt of neuropsychological qualification (author's transl). Arch Psychiatr Nervenkr 1977,224(3):221–233.PubMedGoogle Scholar
- Haguenauer JP, Schott B, Michel F, Dubreuil C, Romanet P: Three case histories of cortical and sub-cortical auditory lesions. Audiological and tomodensimetrical confrontations (author’s transl). Ann Otolaryngol Chir Cervicofac 1979,96(3):185–196.PubMedGoogle Scholar
- Traugott NN, Beskadarov AV, Vasserman LI, Galunov VI, Dorofeeva SA: Clinico-experimental study of auditory-speech agnosia (case with anatomo-histologic verification. Zh Nevropatol Psikhiatr Im S S Korsakova 1980,80(12):1790–1798.PubMedGoogle Scholar
- Mavlov L: Amusia due to rhythm agnosia in a musician with left hemisphere damage: a non-auditory supramodal defect. Cortex 1980,16(2):331–338.PubMedGoogle Scholar
- Buscaino GA, Grossi D, Orefice G: Pure verbal deafness with amusia. Apropos of a case. Acta Neurol (Napoli) 1981,3(1):260–265. ItalianGoogle Scholar
- McFarland HR, Fortin D: Amusia due to right temporoparietal infarct. Arch Neurol 1982,39(11):725–727.PubMedView ArticleGoogle Scholar
- Takeda K, Bandou M, Nishimura Y: Motor amusia following a right temporal lobe hemorrhage--a case report. Rinsho Shinkeigaku 1990,30(1):78–83. ReviewPubMedGoogle Scholar
- Sidtis JJ, Feldmann E: Transient ischemic attacks presenting with a loss of pitch perception. Cortex 1990,26(3):469–471.PubMedGoogle Scholar
- Griffiths TD, Rees A, Witton C, Cross PM, Shakir RA, Green GG: Spatial and temporal auditory processing deficits following right hemisphere infarction. A psychophysical study. Brain 1997,120(Pt 5):785–794.PubMedView ArticleGoogle Scholar
- Peretz I, Belleville S, Fontaine S: Dissociations between music and language functions after cerebral resection: A new case of amusia without aphasia. Can J Exp Psychol 1997,51(4):354–368.PubMedView ArticleGoogle Scholar
- Estañol B, Méndez A: A case of cortical amusia in a patient with musical ability. Rev Neurol 1998,26(152):612–615.PubMedGoogle Scholar
- Schön D, Semenza C, Denes G: Naming of musical notes: a selective deficit in one musical clef. Cortex 2001,37(3):407–421.PubMedView ArticleGoogle Scholar
- Kohlmetz C, Altenmüller E, Schuppert M, Wieringa BM, Münte TF: Deficit in automatic sound-change detection may underlie some music perception deficits after acute hemispheric stroke. Neuropsychologia 2001,39(11):1121–1124.PubMedView ArticleGoogle Scholar
- McChesney-Atkins S, Davies KG, Montouris GD, Silver JT, Menkes DL: Amusia after right frontal resection for epilepsy with singing seizures: case report and review of the literature. Epilepsy Behav 2003,4(3):343–347.PubMedView ArticleGoogle Scholar
- Di Pietro M, Laganaro M, Leemann B, Schnider A: Receptive amusia: temporal auditory processing deficit in a professional musician following a left temporo-parietal lesion. Neuropsychologia 2004,42(7):868–877.PubMedView ArticleGoogle Scholar
- Satoh M, Takeda K, Murakami Y, Onouchi K, Inoue K, Kuzuhara S: A case of amusia caused by the infarction of anterior portion of bilateral temporal lobes. Cortex 2005,41(1):77–83.PubMedView ArticleGoogle Scholar
- Satoh M, Takeda K, Kuzuhara S: A case of auditory agnosia with impairment of perception and expression of music: cognitive processing of tonality. Eur Neurol 2007,58(2):70–77.PubMedGoogle Scholar
- Lechevalier B, Rumbach L, Platel H, Lambert J: Pure amusia reavealing an ischaemic lesion of right temporal planum. Participation of the right temporal lobe in perception of music. Bull Acad Natl Med 2006,190(8):1697–1709. discussionPubMedGoogle Scholar
- Peretz I, Cummings S, Dubé MP: The genetics of congenital amusia (tone deafness): a family-aggregation study. Am J Hum Genet 2007,81(3):582–588. Epub 2007 Jul 20PubMedView ArticleGoogle Scholar
- Hyde KL, Lerch JP, Zatorre RJ, Griffiths TD, Evans AC, Peretz I: Cortical thickness in congenital amusia: when less is better than more. J Neurosci 2007,27(47):13028–13032.PubMedView ArticleGoogle Scholar
- Buklina SB, Skvortsova VB: Amusia and its topic specification. Zh Nevrol Psikhiatr Im S S Korsakova 2007,107(9):4–10.PubMedGoogle Scholar
- Särkämö T, Tervaniemi M, Soinila S, Autti T, Silvennoinen HM, Laine M, Hietanen M: Cognitive deficits associated with acquired amusia after stroke: a neuropsychological follow-up study. Neuropsychologia 2009,47(12):2642–2651. Epub 2009 Jun 3PubMedView ArticleGoogle Scholar
- Hyde KL, Zatorre RJ, Peretz I: Functional MRI evidence of an abnormal neural network for pitch processing in congenital amusia. Cereb Cortex 2011,21(2):292–299. Epub 2010 May 21PubMedView ArticleGoogle Scholar
- Barquero S, Gomez-Tortosa E, Baron M, Rabano A, Munoz DG, Jimenez-Escrig A: Amusia as an early manifestation of frontotemporal dementia caused by a novel progranulin mutation. J Neurol 2010,257(3):475–477.PubMedView ArticleGoogle Scholar
- Phillips-Silver J, Toiviainen P, Gosselin N, Piché O, Nozaradan S, Palmer C, Peretz I: Born to dance but beat deaf: a new form of congenital amusia. Neuropsychologia 2011,49(5):961–969. Epub 2011 Feb 21PubMedView ArticleGoogle Scholar
- Reed CL, Cahn SJ, Cory C, Szaflarski JP: Impaired perception of harmonic complexity in congenital amusia: a case study. Cogn Neuropsychol 2011,28(5):305–321. Epub 2012 Jan 17PubMedView ArticleGoogle Scholar
- Confavreux C, Croisile B, Garassus P, Aimard G, Trillet M: Progressive amusia and aprosody. Arch Neurol 1992,49(9):971–976.PubMedView ArticleGoogle Scholar
- Piccirilli M, Sciarma T, Luzzi S: Modularity of music: evidence from a case of pure amusia. J Neurol Neurosurg Psychiatry 2000,69(4):541–545.PubMedView ArticleGoogle Scholar
- Ayotte J, Peretz I, Hyde K: Congenital amusia: a group study of adults afflicted with a music-specific disorder. Brain 2002,125(Pt 2):238–251.PubMedView ArticleGoogle Scholar
- Wilson SJ, Pressing JL, Wales RJ: Modelling rhythmic function in a musician post-stroke. Neuropsychologia 2002,40(8):1494–1505.PubMedView ArticleGoogle Scholar
- Bautista RE, Ciampetti MZ: Expressive aprosody and amusia as a manifestation of right hemisphere seizures. Epilepsia 2003,44(3):466–467.PubMedView ArticleGoogle Scholar
- Russell SM, Golfinos JG: Amusia following resection of a Heschl gyrus glioma. Case report. J Neurosurg 2003,98(5):1109–1112.PubMedView ArticleGoogle Scholar
- Murayama J, Kashiwagi T, Kashiwagi A, Mimura M: Impaired pitch production and preserved rhythm production in a right brain-damaged patient with amusia. Brain Cogn 2004,56(1):36–42.PubMedView ArticleGoogle Scholar
- Terao Y, Mizuno T, Shindoh M, Sakurai Y, Ugawa Y, Kobayashi S, Nagai C, Furubayashi T, Arai N, Okabe S, Mochizuki H, Hanajima R, Tsuji S: Vocal amusia in a professional tango singer due to a right superior temporal cortex infarction. Neuropsychologia 2006,44(3):479–488.PubMedView ArticleGoogle Scholar
- Peretz I, Brattico E, Tervaniemi M: Abnormal electrical brain responses to pitch in congenital amusia. Ann Neurol 2005,58(3):478–482.PubMedView ArticleGoogle Scholar
- Hyde KL, Zatorre RJ, Griffiths TD, Lerch JP, Peretz I: Morphometry of the amusic brain: a two-site study. Brain 2006,129(Pt 10):2562–2570.PubMedView ArticleGoogle Scholar
- Hoffmann M: Isolated right temporal lobe stroke patients present with Geschwind Gastaut syndrome, frontal network syndrome and delusional misidentification syndromes. Behav Neurol 2008,20(3):83–89.PubMedGoogle Scholar
- Vanstone AD, Cuddy LL, Duffin JM, Alexander E: Exceptional preservation of memory for tunes and lyrics: case studies of amusia, profound deafness, and Alzheimer’s disease. Ann N Y Acad Sci 2009, 1169:291–294.PubMedView ArticleGoogle Scholar
- Tillmann B, Burnham D, Nguyen S, Grimault N, Gosselin N, Peretz I: Congenital Amusia (or Tone-Deafness) Interferes with Pitch Processing in Tone Languages. Front Psychol 2011, 2:120.PubMedView ArticleGoogle Scholar
- Hochman MS, Abrams KJ: Amusia for Pitch Caused by Right Middle Cerebral Artery Infarc. J Stroke Cerebrovasc Dis 2012. http://dx.doi.org/10.1016/j.jstrokecerebrovasdis.2012.06.016 Google Scholar
- Geshwind N: The brain of a learning-disabled individual. Ann Dyslexia 1984, 34:319–327.View ArticleGoogle Scholar
- Lebrun MA, Moreau P, McNallt-Gagnon A, Mignault Goulet G, Peretz I: Congenital amusia in childhood: a case study. Cortex 2012,48(6):683–688.PubMedView ArticleGoogle Scholar
- Eustache F, Lechevalier B, Viader F, Lambert J: Identification and discrimination disorders in auditory perception: a report on two cases. Neuropsychologia 1990,28(3):257–270.PubMedView ArticleGoogle Scholar
- Hattiangadi N, Pillion JP, Slomine B, Christensen J, Trovato MK, Speedie LJ: Characteristics of auditory agnosia in a child with severe traumatic brain injury: a case report. Brain Lang 2005,92(1):12–25.PubMedView ArticleGoogle Scholar
- Johannes S, Jöbges ME, Dengler R, Münte TF: Cortical auditory disorders: a case of non-verbal disturbances assessed with event-related brain potentials. Behav Neurol 1998,11(1):55–73.PubMedGoogle Scholar
- Mazzoni M, Moretti P, Pardossi L, Vista M, Muratorio A, Puglioli M: A case of music imperception. J Neurol Neurosurg Psychiatry 1993,56(3):322.PubMedView ArticleGoogle Scholar
- Peretz I, Kolinsky R, Tramo M, Labrecque R, Hublet C, Demeurisse G, Belleville S: Functional dissociations following bilateral lesions of auditory cortex. Brain 1994, 117:1283–1301.PubMedView ArticleGoogle Scholar
- Penagos H, Melcher JR, Oxenham AJ: A neural representation of pitch salience in nonprimary human auditory cortex revealed with functional magnetic resonance imaging. J Neurosci 2004, 24:6810–6815.PubMedView ArticleGoogle Scholar
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