Defining 'normal': Methodological issues in Aphasia and intelligence research.

Theodore H. Weisenburg (1876-1934) and a series of colleagues embarked on a research program in 1927 to develop standardized tests to investigate the nature of language and intellectual impairments in aphasic and non-aphasic individuals. This project culminated in two significant contributions to neuropsychological testing (Weisenburg & McBride, 1935; Weisenburg, Roe and McBride, 1936). After an initial study demonstrated the problematic aspects of Henry Head's aphasia tests (1926), Weisenburg developed a new battery of tests which were given to individuals with aphasia. The significant innovation of this work was the original concept of a matched control group. This included those with other neurological impairments, and a range of non-neurologically impaired individuals with the aim of providing a characterization of what was 'normal'. They identified many crucial participant variables regarding age, education, and socioeconomic status and used population statistics to ensure their control sample was representative. A detailed critical assessment of each of their successive elaborations is examined, focusing on the methodological innovations they represent. The contribution of this work to contemporaries and successive generations of neuropsychologists is examined regarding ongoing issues in clinical testing and research design.

The Long View of Language Localization.

For British neurologists, one case was considered to represent significant evidence regarding the organization of language in the brain in the second half of the 19th century. The interpretation of its significance was based on repeated standard clinical assessment of behavioral deficits, the use of a psychological model of processing, and lesion localization to inform understanding of clinic-pathological correlation. The aphasic deficits experienced by a single case were observed and recorded by London neurologist Henry Charlton Bastian (1837-1915) over a period of 18 years and used as a demonstration of clinico-pathological reasoning regarding language function. This case was well documented in many of Bastian's publications; presented in teaching demonstrations; included in discussions at medical society meetings and public lectures; and reported widely in the medical press. When this patient died, the autopsy findings were added to the extensive record of his language deficits. Some aspects of the size and site of the lesion were consistent with Bastian's clinical predictions arising from his model of language processing, while others presented more of a paradox. This single case was a significant source of discussion and reflection in the medical community throughout the second half of the 19th century. Examination of various interpretations of this case reveal the assumptions regarding the functional architecture of language processing and more general theoretical considerations of how evidence from cases of acquired neurogenic aphasia can be employed in developing such models. This long view into a historical case sheds light on the challenges of clinic-pathological correlation methods in the understanding of localization of language functions which remain today.

The laryngoscope and nineteenth-century British understanding of laryngeal movements.

The source of the human voice is obscured from view. The development of the laryngoscope in the late 1850s provided the potential to see the action of the vocal folds during speaking for the first time. This new instrument materially contributed to the understanding of vocal fold neuroanatomy, neurophysiology, and neuropathology. The laryngoscope led to elaborated understanding of disorders that previously were determined by changes in sound. The objective of this paper is to detail the consequences of this novel visualization of the larynx, and to trace how it aided in the development of understanding of the movements of the vocal folds. This is demonstrated through an examination of the activities and practices of a group of London clinicians in the second half of the nineteenth century.

Preserved appreciation of aesthetic elements of speech and music prosody in an amusic individual: A holistic approach.

We present a follow-up study on the case of a Greek amusic adult, B.Z., whose impaired performance on scale, contour, interval, and meter was reported by Paraskevopoulos, Tsapkini, and Peretz in 2010, employing a culturally-tailored version of the Montreal Battery of Evaluation of Amusia. In the present study, we administered a novel set of perceptual judgement tasks designed to investigate the ability to appreciate holistic prosodic aspects of 'expressiveness' and emotion in phrase length music and speech stimuli. Our results show that, although diagnosed as a congenital amusic, B.Z. scored as well as healthy controls (N=24) on judging 'expressiveness' and emotional prosody in both speech and music stimuli. These findings suggest that the ability to make perceptual judgements about such prosodic qualities may be preserved in individuals who demonstrate difficulties perceiving basic musical features such as melody or rhythm. B.Z.'s case yields new insights into amusia and the processing of speech and music prosody through a holistic approach. The employment of novel stimuli with relatively fewer non-naturalistic manipulations, as developed for this study, may be a useful tool for revealing unexplored aspects of music and speech cognition and offer the possibility to further the investigation of the perception of acoustic streams in more authentic auditory conditions.

Morell Mackenzie's Contribution to the Description of Spasmodic Dysphonia.

OBJECTIVES: Since the middle of the 20th century, most discussions of spasmodic dysphonia (SD) reference a paper by Ludwig Traube published in1871 as the first historical citation, crediting him with priority for this clinical syndrome. However, our recent research has determined that the original observation by Traube was published in 1864 and does not in fact describe what is currently recognized as SD. It appears that many clinics throughout Europe and North America were investigating and publishing observations on a range of voice disorders. METHODS: The wider context of work on laryngeal disorders in the 1860s-1870s is considered. One of Traube's contemporaries, Morell Mackenzie, made significant contributions to the understanding of laryngeal movement disorder and its consequences for the voice. These will be examined to gain a clearer focus on the characterization of this disorder. RESULTS: The clinical descriptions published by Morrell Mackenzie in the 1860s provide details that conform quite closely to our current-day understanding of SD. CONCLUSIONS: The citation of Traube's "hysterical" patient links to mid 20th-century views of the functional nature of SD and the utility of psychiatric treatment. The description presented by Mackenzie is consistent with current views of SD as a movement disorder.

Tracing Spasmodic Dysphonia: The Source of Ludwig Traube's Priority.

OBJECTIVES: Since the mid-20th century, one citation is given historical priority as the first description of spasmodic dysphonia (SD): Ludwig Traube's 1871 case of the "spastic form of nervous hoarseness." Our objective is to understand how this case serves as the foundation of understanding laryngeal movement disorders. METHODS: The original German paper was located and translated. Bibliographical and bibliometric methods are used to determine the citation history of this original source over the past 140 years. RESULTS: Although secondary citations in contemporary publications typically credit Traube for establishing the clinical entity SD, his case does not conform to currently accepted diagnostic features. Citation patterns indicate the source of Traube's priority is publications by Arnold and Luchsinger, mid-20th-century ENT clinicians, particularly their influential 1965 textbook used to train US and UK clinicians on voice disorders for several generations. CONCLUSIONS: Sometimes secondary citations in medical literature lead to the inadvertent perpetuation of factual misrepresentation. The clinical picture of Traube's original case does not represent what clinicians would recognize as SD today. The rich 19th-century literature on voice disorders is a valuable resource for present day clinicians.

An ecological method for the sampling of nonverbal signalling behaviours of young children with profound and multiple learning disabilities (PMLD).

BACKGROUND: Profound and multiple learning disabilities (PMLD) are a complex range of disabilities that affect the general health and well-being of the individual and their capacity to interact and learn. METHOD: We developed a new methodology to capture the non-symbolic signalling behaviours of children with PMLD within the context of a face-to-face interaction with a caregiver to provide analysis at a micro-level of descriptive detail incorporating the use of the ELAN digital video software. CONCLUSION: The signalling behaviours of participants in a natural, everyday interaction can be better understood with the use of this innovation in methodology, which is predicated on the ecology of communication. Recognition of the developmental ability of the participants is an integral factor within that ecology. The method presented establishes an advanced account of the modalities through which a child affected by PMLD is able to communicate.

The Third Man: Robert Dunn's (1799-1877) Contribution to Aphasia Research in Mid-Nineteenth-Century England.

Throughout his medical career, Robert Dunn (1799-1877) published a number of clinical cases with postmortem reports involving acquired language disorders, with the first noted in 1842. He developed a physiologically informed approach to psychological function during the 1850s along with a group of notable colleagues Benjamin Collins Brodie, Henry Holland, Thomas Laycock, John Daniel Morell, and Daniel Noble. He was also active in ethnographic research on human origins and racial diversity. As such, Dunn represents an interesting player in the developing fields of neurology, psychology, and anthropology in England in the latter part of the nineteenth century. These various strands converged at the meeting of the British Association of the Advancement of Science in 1868, where Dunn shared the program of lectures on the cutting-edge topic of aphasia with Paul Broca (1824-1880) and John Hughlings Jackson (1835-1911). Dunn's ideas developed over a longer time frame than his younger colleagues and as such represent a unique blending of concepts from the earlier work of Franz Josef Gall (1758-1828) and Jean-Baptiste Bouillaud (1798-1881) to the perspectives on language organization in the brain developed after 1861.

Singing by speechless (aphasic) children: Victorian medical observations.

In the second half of the nineteenth century, British clinicians made observations regarding the ability of individuals with impaired language abilities to sing or hum. One notable publication was of two cases of children briefly observed by John Hughlings Jackson (1835-1911) in 1871. These children were speechless but could produce some musical expression. Other such cases attracted the attention of Victorian clinicians who were actively pursuing theoretical questions regarding the organization of brain function and laterality. The presence of musical expression in children who failed to develop spoken language was seen as a notable symptom for early practitioners of pediatric neurology.

The merest Logomachy: The 1868 Norwich discussion of aphasia by Hughlings Jackson and Broca.

This article reconsiders the events that took place at the 1868 meeting of the British Association for the Advancement of Science (BA) in Norwich. Paul Broca and John Hughlings Jackson were invited to speak on the new and controversial subject of aphasia. Over the ensuing decades, there have been repeated references made to a debate between Broca and Jackson. This meeting has been identified as a turning point in favour of Broca's position on the cerebral localization of language. A return to original sources from key witnesses reveals that the opinion of the British practitioners was generally against Broca's views. Close examination of contemporaneous materials suggests that no public debate between Jackson and Broca occurred. However, the public discussion after Broca's presentation records notable concerns over both theoretical issues of localization of function and the status of exceptional clinical cases. A significant stage in the development of current views on the organization of language in the brain is revealed in the accounts of the BA meeting in August 1868 and successive responses to these events in the British press over a period of years.

The validity of Barlow's 1877 case of acquired childhood aphasia: case notes versus published reports.

In 1877, Barlow described a ten-year-old boy with right hemiplegia and aphasia, quick recovery of language function, and subsequent left hemiplegia and aphasia, who was shown to have symmetrical left and right Broca's area lesions at autopsy. The report of this case motivated many writers in the second half of the nineteenth century to develop theories on localization, laterality, equipotentiality and development of specialization, recovery of function, and the role of the right hemisphere (see Finger et al., 2003, for review). This paper presents an analysis of the original archived case notes that have recently come to light. Examination reveals discrepancies in significant details of the history of the case and raises questions about the degree of impairment and recovery throughout his illness as reported in the published article. Consideration of these differences between the presentation of the case in the British Medical Journal publication and the documentation in the original patient records raises issues about the validity of this case as evidence for the many arguments it was to support that have persisted to the present.

Language development in a 3-year-old boy with Prader-Willi syndrome.

Prader-Willi syndrome (PWS) is a genetic disorder which has widespread developmental consequences including motor, cognitive and language delay. Previous research on PWS children has focused primarily on phonological development and dysfluency. In the present study, the lexical development of a boy with PWS was investigated in a series of 18 play sessions recorded over a 4 month period from the ages 3;7 to 3;11. In comparison to the language development of children with Down syndrome this child with PWS appears to display a distinct developmental pattern. The possibility of detailing a behavioural phenotype of genetic disorders affecting language development is discussed.

The unknown source of John Hughlings Jackson's early interest in aphasia and epilepsy.

The National Hospital for Paralysis and Epilepsy in London (founded 1859) was the scene of great discoveries in the new specialty of neurology, carried out in great part by John Hughlings Jackson (1834-1911). The clinicians Jonathan Hutchinson and Charles Edward Brown-Sequard are typically identified as Jackson's mentors. This paper discusses the previously neglected role of Jabez Spence Ramskill (1824-1897), founding physician of the National Hospital. Ramskill appears to have been significant in providing the opportunity and context that led Jackson to develop his theories concerning higher cerebral function disorders. As assistant physician to Ramskill at the National Hospital, Jackson was provided with a vast caseload of epileptic, hemiplegic, and aphasic patients. Ramskill and Jackson both published papers on aphasia in the London Hospital Reports in 1864. Consideration of the similarities and differences between these 2 papers highlight significant issues in the clinical and theoretical development of understanding language organization in the brain. The early writings of Jackson and case notes of Ramskill document a close link between the 2 and indicate the debt that Jackson had to Ramskill for providing him with the opportunities to develop his original ideas on epilepsy and aphasia.

Premature thoughts on writing disorders.

Three papers appeared in the 19th century describing the dissociation between speech and writing: Marce (1856), Ogle (1867), and Pitres (1884). An account of the convincing evidence of dissociations put forward in these papers is presented. Three explanations are proposed as to the reason why the observations reported by these authors were overlooked or rejected by their contemporaries, namely: (a) in the first half of the century it seems that very little knowledge of the processes underlying writing (as opposed to speech) was available, (b) the debates focussed on the independence of speech versus motor control and language versus the intellect, (c) parallelisms between phylogeny, ontogeny and aphasia impeded the application of the principle of double dissociations, including the dissociations between speech and writing. It is argued that this phenomenon in the history of aphasia is best captured by the concept of prematurity in scientific discovery proposed by Stent (1972, 2003).

The history of written language disorders: Reexamining Pitres' case (1884) of pure agraphia.

The first clinical description of pure agraphia was reported by the French neurologist Pitres in 1884. Pitres used the case study evidence to argue for modality-specific memory representations and the localization of writing. This article reviews Pitres's contribution to the study of acquired writing disorders, the components of writing models and the cerebral localization which subserve writing, in light of the views entertained by his contemporaries and current authors. Although numerous cases have been reported throughout this century, the view that writing can be impaired while other language functions and motor activities remain intact is still challenged.