BRAIN AND LANGUAGE 3, 88-l 13 (1976) The “Pure Form” of the Phonetic Disintegration Syndrome (Pure Anarthria); Anatomo-Clinical Report of a Historical Case ANDRE ROCH LECOURS' Centre de Recherches en Sciences Neurologiques, C. R.M.C., UniversitP de Mont&al AND FRANCOIS LHERMITTE Service de Neurologie et Neuropsychologie, Hbpital Groupe de Recherches Neuropsychologiques, de la Salpitrit+e, Paris and I. N.S. E. R.M. U-84. The relationships of simple aphasia (Baillarger), pure anarthria (Marie), pure motor aphasia (Dejerine), subcortical motor aphasia (Lichteim, Charcot) and phonetic disintegration (Alajouanine, Ombredane, & Durand) are outlined. Alajouanine et al.‘s pioneering neurolinguistic studies of phonetic disintegration, both in the semiological context of Broca’s aphasia and as an isolated clinical entity, are briefly discussed. One of their neurological and linguistic observations is reviewed in detail, including Marguerite Durand’s comparative phonetic description of a small corpus of French and English segments (bilingual patient). Anatomical examination of the brain, in this case, is reported: It revealed, essentially, a cortico-subcortical softening of the inferior half of the left precentral gyrus, with sparing of Broca’s area proper. Comments are made on Durand’s conclusion about the possibility of a lesser involvement of English vs. French phonetics in this patient. The notion of pure anarthria is redefined, both from a theoretical and from an operational point of view. The phonetic characteristics of the dystonic phase of phonetic disintegration are discussed. The discussion also bears on the concept of anarthria vs. that of aphasia, and on the possible localizations of cerebral lesions leading to pure phonetic disintegration. The localization of the lesion in the reported case is compared to that in Bernheim’s and Ladame’s cases, on which Dejerine grounded his theory of motor aphasia. The clinical existence of a form of aphasia-designated as such or otherwise-consisting of isolated or nearly isolated involvement of the The authors are indebted to Madame Blanche Ducame and to Professors Raymond Escourolle and Laurent Santerre with whom they discussed, respectively, the clinical, pathological, and linguistic aspects of their manuscript. They also wish to express their gratitude to Professor Jean-Claude Gautier, who brought to their attention the possiblity of completing Alajouanine’s and Durand’s historical neurolinguistic case report by an anatomical study. 1 A. R. L. is a Scholar of the Medical Research Council of Canada. His work is supported by M. R. C. grant MA-4210. 88 Copyright All rights 0 1976 by Academic Press, Inc. of reproduction in any form reserved. PURE ANARTHRIA 89 purely expressive aspect of spoken language has been recognized early in the course of the history of aphasiology. Baillarger’s classification, for instance, as presented at 1’AcadCmie de Medecine, in 1865, is unequivocal in this respect: “It is well known that a number of aphasics suffer of nothing but a more or less complete deprivation of speech (la parole). Theirs’ is simple aphasia (I’aphasie simple), that is, aphasia in the strict sense of the word. Besides these patients, one can observe others whose disorder is quite different, i.e., those who pronounce words unrelated to their thoughts. Their lesions result in a more or less severe perversion of the faculty of avec perversion de la facultk du langage articulk). articulated language (I’aphasie I will limit myself to comments on the two main forms [of simple aphasia]. The first one groups all cases of aphasia with loss of both spoken and written expression: the second, all those in which there is loss of spoken expression only. . . [In the latter], the patient’s memory for words is normal since they can translate all their thoughts into written language.” In Baillarger’s opinion, Broca’s interpretation of such isolated involvement of the motor moment of spoken language, i.e., an amnesia specific to patterns of movements in the bucco-phonatory apparatus (Broca, 1861), was inadequate: in view of the occasional preservation of a certain level of automatic speech production in this condition, he suggested that it might testify, at least in some cases, to specific loss of the capacity of voluntary speech production.’ (Baillarger, 1865, as reprinted in 1890.) An aphasic disorder limited to spoken expression was thus well enough defined, in Broca’s time to generate pertinent theoretical discussions. Since most of the latter took place in France, terminological quarrels were bound to develope in parallel. Clearly, the most significant was to oppose Pierre Marie and Dejerine a few decades later. To the former, “nosological sanity” required simple aphasia limited to spoken expression to be named otherwise; he suggested anarthria, a label still widely in use within the French school. “It [anarthria] is characterized by the fact that the patient’s utterances are so distorted and incomprehensible that one might, in this respect, confuse anarthria and Broca’s aphasia; but differential characteristics between the two are numerous and definite. As opposed to aphasics, anarthrics understand perfectly what is told to them, including complex sentences; they can read and write, and are even able to indicate . . the number of syllables or letters in the words they cannot utter” (Marie, 1906a). On the other hand, “true aphasia is not characterized by absence or distortion of elocution but by a comprehension disorder, i.e., a particular type of intellectual deterioration,” and by anomalies in reading and writing (Marie, 1906b). Such considerations led Pierre Marie to the formulation of his famous tquation asserting the oneness of aphasia and the essentially different natures of anarthria and aphasia: “Broca’s Aphasia = Wernicke’s Aphasia + Anarthria” (Marie, 1907). ’ “L’analyse des phinomGnes conduit ir reconnaitre, dans pertains cas de ce genre, que /‘incitation verbale involontairepersiste, mais quel’incitation verbale volontajre est abo/ie.” 90 LECOURS AND LHERMITTE Pierre Marie also had very definite- but, curiously enough, limited to horizontal dimensions (Fig. I)-conceptions about the site of brain lesions leading to anarthria and of those leading to aphasia. He attributed the former to damage within a region that was soon to be known as Pierre Marie’s quadrilateral (Fig. 1): “With regard to anarthria, there are no difficulties whatsoever, and everybody agrees that lesions are localized in the region and in the surroundings of the lenticular nucleus, that is, in this structure itself, or in the anterior limb and in the knee of the internal capsule, or else in the external capsule. It should be noted that anarthria does not necessarily result of a left hemisphere lesion, i.e., that it can occur as a result of damage to the zone of the lenticular nucleus on the right side” (Marie, 1906a). Marie and Dejerine were indeed in complete agreement as to the clinical characteristics of isolated arthric disorder in hemispheric lesions. “In summary, ” wrote the latter, “the single morbid phenomenon consists of FIG. 1. Pierre Marie’s quadrilateral (l!%a). The comment is translated from the original: “Horizontal section, through a normal left hemisphere, showing the topography of territories lesions of which lead to language disorders or, on the contrary, which are in no way related to production of such disorders. Lesions involving portions of the hemisphere (frontal lobe and, particularly, third frontal convolution) located rostra1 to transverse line A, which originates at the sulcus separating the third frontal from the insular convolutions, lead neither to anarthria nor to aphasia. Lesions involving the structures between lines A and B cause anarthria. Line B is traced transversally, from the posterior aspects of the insula and of the lenticular nucleus. Lesions occurring behind line B give rise to aphasia. In I is localized the isthmus of white matter which interconnects the temporo-parietal lobe and the lenticular nucleus region: in lesions leading to a combination of Wemicke’s aphasia and anarthria, that is. to Broca’s aphasia, this isthmus is most often among the involved structures.” PURE ANARTHRIA 91 an anomaly in the articulation of the sounds of language. . . . All other language modalities are intact and internal language is normal” (Dejerine, 1914). But Dejerine considered this clinical picture as a variety of aphasia proper, which he called pure motor aphasia or, with Charcot (1883) and Lichteim (1885) subcortical motor aphasia. Furthermore, he agreed that the arthric disorder in subcortical motor aphasia and that in cortical motor or Broca’s aphasia are identical, but he forcefully denied that Broca’s aphasia represents a mere clinical admixture of arthric disorder and Wemicke’s aphasia, and he rejected Marie’s assertion defining aphasia, whether of the Broca or of the Wernicke type, as a variety of intellectual deterioration (Dejerine, 1906, 1914). He also insisted-and the fact is of obvious importance- that pure motor aphasia, although it does occur straight off in a few cases, usually emerges at an earlier or a later stage in the evolution of Broca’s aphasia, sometimes even of global aphasia. Dejerine’s opinion as to the site of brain damage in pure motor aphasia and Marie’s opinion as to the site of brain lesion in pure anarthria (cf. unteu) were, at least apparently, quite different. In his earlier writings, Dejerine unhesitantly affirmed that damage did not involve Broca’s area itself but, subcortically, “the association fibers connecting Broca’s area to the rolandic operculum” (Dejerine, 1901) or to lower structures, i.e., to bulbar nuclei (Dejerine as quoted in Marie, 1906b). However, new anatomo-clinical observations later led Dejerine to a more circumspect formulation of his doctrine: “The localization of the lesion responsible for pure motor aphasia . . . appears to be less certain than a few years ago. It was thought to be always subcortical to Broca’s area, isolating it or its efferent fibers from the motor centers governing lingual, pharyngeal and laryngeal muscles” in the process of articulated linguistic expression. But, in view of new observations in which lesions of the foot of the left third frontal convolution were found to be both cortical and subcortical, pure motor aphasia is now best thought of in terms of a clinical entity related to the presence of either a subcortical or a corticosubcortical lesion (Dejerine, 1914). In any event, there are no reasons to believe that isolated lesions of the lenticular nucleus lead to arthric disorder (Dejerine, 1906). Until the late 193Os, students of aphasia kept applying their descriptive abilities to the brains of aphasics, and very little to their language. Aside from Freud’s comments on the structures of paraphasias (1891), neurolinguistics was yet to be born. The event took place in 1939, with the publication by Theophile Alajouanine, Andre Ombredane and Marguerite Durand, the latter a linguist, of a systematic study of the phonetic disorder in Broca’s aphasia (Le syndrome de dtsinttgrution phonktique duns l’uphusie). In another description princeps, now nearly forgotten, Alajouanine and Durand, this time with Pierre Pichot, later 92 LECOURS AND LHERMITTE published a detailed neurological, psychological and linguistic report on their patient, E. Fr., a bilinguial subject who suddenly developed pure anarthria as the result of a stroke, presumably by embolism (Alajouanine et al., 1949). In the present paper, we plan: (a) to review E. Fr.‘s case, paying particular attention to Durand’s phonetic study and taking into account a few additional documents given us by Madame Blanche Ducarne, who acted for a while as E. Fr.‘s therapist; (b) to complete the observation by a description of the brain lesion responsible, in this particular case, of pure anarthria of sudden onset; and (c) to discuss the problem of “pure” phonetic disintegration from the triple point of view of clinical neurology, pathological anatomy, and linguistics. CASE REPORT Monsieur E. Fr. (1884-1957) was born in France of an English mother and a French father, Although both were fluent in French and English, near exclusive use of the latter characterized intrafamilial linguistic exchange. Thus, E. Fr. spoke mainly English until the age of four; indeed, his knowledge of French was said to be-at best-rudimentary when he began primary school at the age of six. He attended French schools from 1890 to 1900, which eventually led him to nativelike mastery of the French language. He obtained a Certificat d’Etudes Primaires at the age of twelve and a Brevet ElCmentaire at the age of sixteen. The latter is said to have pre-supposed higher than average proficiency and correctness in written as well as in spoken French. Had he so oriented his career, this diploma would have entitled E. Fr. to work as an instituteur (primary school) in certain parts of France. As things turned out, he became a successful commercial agent. He spent all of his life in France but, due to his familial, social, and professional relationships, he kept speaking English as well as French. It was indeed his and his family’s very definite opinion that he was “perfectly bilingual”; comments to this effect explicitly stated that he could speak either language “saris accent.” As were all members of his immediate family, E. Fr. was right-handed. On December 10, 1947, E. Fr .-then 63 years old-was admitted to the Service de Cardiologie of I’Hapital LariboisiPre, in Paris. The hospitalization aimed at elective study and treatment of a long-standing (approximately 10 years) cardiac ailment manifested by episodes of paroxystic tachycardia. On December 12, while walking in the hospital, the patient suddenly fell to the floor. His state of consciousness remained normal. Immediate examination revealed the presence of a right hemiplegia and hemihypesthesia; it also showed that comprehension of spoken language was normal although the only vocal production possible was an undifferentiated raucous sound emitted whenever the patient at- PURE ANARTHRIA 93 tempted to talk. Near complete regression of the sensori-motor deficit rapidly took place at limbs level: on December 13, the patient could move his right arm and leg and the hemihypesthesia had improved considerably; in early January, 1948, he was able to walk unassisted. From December 12 to early January, the patient’s speech impairment remained maximal. Rapid improvement occurred thereafter over a period of 4 weeks: at the end of January, E. Fr. could talk rather efficiently although in a slowed, syllabical, dysprosodic, and dysarthric manner. In the following translated excerpts of a letter written in January, the patient describes, for the benefit of his physician, the course of his partial recovery: Doctor, I will try to explain to you the reason I have to syllabicate. In the normal state, thought is expressed, through speech [/a parole], automatically. One gives very little attention, if any, to articulation; by the strength of habit, articulation has become mechanical [machinale]. Four weeks ago, all that came out of my mouth when I wanted to say a word was a raucous sound. Then, one day, I succeeded to pronounce “a,” ‘3” and, later, ‘I a, ,. ‘6e, 7. “.I, 11‘1 ” “u”; during the following days, I got a few syllables and words 0, beginning with a vowel. It has been only ten days since I have been able to utter “oui”. . . . [In the past few days,], I have nearly recovered the ‘j,” “g” and “I.” Three days ago, I was unable to say the name of my son, “Jean”; neither was I able to say “le, ” “la, ” “les.” I can only talk syllabically because my articulation is sluggard [paresseuse]. It is no longer automatic but has to be commanded, directed. I have to think of the word I am going to utter, and of the way in which to utter it. If I want I can no longer do so out of habit; it is no longer automatic. If I to say “bonjour,” am not very attentive, I would say “beuseu.” If I wish to say “le,” “la,” without being very attentive, I would say “de, ” “da.” For this reason, I must articulate each vowel, each consonant, in short each syllable.2 The patient wrote this text, and earlier or later ones, with his right hand. We will come back to the conspicuous absence of paragraphic transformations, both literal and verbal, and to the remarkable normalcy in choice of words, graphism proper, and written language automatisms (linear disposition, regularity in spatial distribution of letters, words, and lines, alineas, etc.) (Fig. 2). In view of what was considered to be a clear-cut indication for speech therapy, the patient was transfered to Alajouanine’s Centre du Langage, at l’H6pital de la Salpbtrikre, on January 24, 1948. A neurological examination, on admission day, showed the existence of a residual sensori-motor deficit, face included, with brisker osteo-tendinous reflexes and diminished cutaneo-abdominal reactions on the right side, and a right Babinski sign; a minimal right hemihypesthesia was thought to be present but it was no longer to be found a week or so later. From * The two errors in tense concordance, both involving use of a conditional predicate, would not come as a surprise in a text written by a normal subject of comparable educational level. 94 LECOURS AND LHERMITTE FIG. 2. Case E. Fr. Sample of handwriting in early evolution (right hand). January 24, that is, from 6 weeks after the onset of the neurological disease, clinical examinations yielded no evidence of bucco-facial apraxia nor of any neurological anomaly other than the residual right hemideficit and the speech disorder. Detailed psychological and aphasiological evaluations were made. Oral Expression Although at a much slower pace than during the 4 previous weeks, spontaneous and/or reeducation-determined regression of the speech disorder kept occurring until mid-February; but for imperceptibly progressive gain in fluency, very little change was observed thereafter. At this point of stabilization, a very striking dissociation was manifest in conversational exchange between lexical, semantic, and syntactic adequacy (indeed, at times, sophistication), on the one hand, and gross arthric disorder, on the other. The speech flow was laborious, syllabical, PURE ANARTHRIA 95 explosive, with marked anomalies in scansion and rhythm. Hesitations were numerous, usually in the form of repetitions of initial phonemes. In order to provide a likely impression, the following conversational sample should be read aloud, slowly and syllabically, with exaggeration in the strength and amplitude of peribuccal and other bucco-phonatory movements, particularly in the case of occlusive phonemes. E. Fr. is answering a question concerning his work: /m>/J. /m>m/J. Momentanement, je /sl/& /sl/J je suis /Rm:/&/Roe/J representant en vins. C’estJ. cei ce n’est pas mon m&tier en temps normal. MaisJ. mais& J’letill j’ttais reprtsentant, mais deJ de /Ii/l lleii linoleums. /dJ l&/i Avant /Ii/& la guerre, j’etais agent d’une /y/J. iyll usine de . . dune /pa/$ /pafi/l /fabikm/ de linoleums et /twa/ cirees3 Whether or not transformations of the type /f-b/ + /p-f/ in the approach to /fabikce/ Cfubrique) should be considered phonemic paraphasias rather than phonetic distortions will be discussed below. Except for a large number of phonetic approaches and distortions (cf. Durand’s phonetic study), repetition and reading aloud were normal. The arthric disorder was manifest both when the patient spoke English and when he spoke French. Nonetheless-and common agreement existed on this point between the patient, his family, the neurologists Alajouanine and Ombredane and the linguist Durand-arthric anomalies were thought to be grosser and more frequent when E. Fr. conversed, narrated, repeated, and read in French than when he conversed, narrated, repeated, and read in English. Indeed, it was mainly this fact that prompted Alajouanine’s et al. original report. We shall return to Durand’s attempt at objectifying this dissociation. Written Expression In obvious contrast with the arthric disorder distorting the patient’s oral expression, no anomalies of graphism were observed in his written productions (Fig. 2). Most likely in partial if not complete dependence of the residual sensori-motor deficit in the right-hand, redaction time was 3 Phonetic approaches and distortions appear between oblique slashes and are-approximately-transcribed in symbols of the International Phonetic Association as listed in Harrap’s New Shorter French & English Dictionary, J. E. Mansion, (Ed.), 1%7. Toronto and Vancouver: Clarke, Irwin & Co. Vertical arrows (4) indicate the end of phonetic approaches; they are followed by the patient’s attempts at correction of his utterances. Without the approaches and distortions, the transcript would read: “Momentantment, je suis reprksentant en vins. Ce n’est pas mon metier en temps normal. J’ttais representant, mais de linolbums. Avant la guerre, j’btais agent d’une usine de . d’une fabrique de linolkums et toiles cirges.” In translation: “Temporarily, I work as a salesman for vine-growers. This is not my usual trade. I was a salesman, but in the linoleum business. Before the war, I was the agent of a linoleum and oilcloth factory.” (The idiomatic English equivalent of both “usine” and “fabrique” being “factory,” we were not able to translate the patient’s lexically sophisticated hesitation between the two words.) 96 LECOURS AND LHERMITTE somewhat longer than expected although quite fluent (19 min for the written description below, including stimulus exploration time). Spelling was normal, aloud as well as in writing. As in conversational exchange, lexicon, syntax, and semantics were normal, or nearly so, in spontaneous written expression. The fact is well illustrated by the patient’s essay, at the end of January 1948, on Caran d’Ache’s histoire suns paroles (“Dipart pour la revue”; Fig. 3): Un officier de hussard a manifest6 le de’sir de se rendre ti la revue, month’,je veux dire ti cheval. Depuis des annees il n’avait pas fait d’tquitation et avait pris un embonpoint remarquable. Son dksir (est) exauck, c’est’ci-dire qu’on lui am&e un superbe pur sang en vue de I’exaucer. Mais pour monter ri cheval, c’est une affaire qui s’avke impossible saris le secours d’un aide. L’aide qui avait (ameni) le cheval est ii proximite. L’officier I’appelle, mais malgrk tous ses efforts, c’est en vain que I’opkration est tentie. II se trouve 6 passer un camarade de notre homme de corvte, il I’appelle, et toux deux au prix d’efforts consid&ables, arrivent b mettre I’officier sur le cheval. Mais patatras, le be1officier pique une t&e et son uniforme de grande tenue, le plumet, le casque qui lui est descendu plus bas que les oreilles, son bedon a fait &later sa veste, ses genoux qui ont marquP sur la culotte. Le r&e n’est plus rkalisable. Le bel of$cier est ramen& par le jeune homme de cotvie, ir qui I’aventure provoque une hilarite’ bien compre’hensive.4 FIG. 3. From Ombredane (1950), Caran d’Ache’s histoire saris paroles (“Dkpartpour revue”). E. Fr.‘s written description in the text. la 4 In translation: “A hussard officer has expressed the desire to go to the revue, mounted, I mean on a horse. For years he had not been horseback riding and had acquired notable stoutness. His desire (is) granted, that is, he is given a superb thoroughbred to grant it. But to mount a horse turns out to be an impossible venture without the succour of an aidedecamp. The aide-de-camp who had (brought) the horse is near by. The officer calls him, but in spite of all his efforts, it is in vain that the operation is attempted. It so happens that a comrade of our chore man passes by, he calls him, and the both of them at the price of PURE ANARTHRIA 97 Both the spontaneous corrections or modifications in this text (parentheses) and the questionable use of “comprPhensive” instead of “comprthensible” illustrate phenomena not infrequently observed in the written productions of normal subjects. Whether or not such is also the case of syntactical incorrections in long sentences (see the segment beginning with “Mais patatras”) is more difficult to assess. Be that as it may, E. Fr.‘s reader is left with a global impression of-at worst-near normalcy in writing. It should be said, however, that the patient himself long considered his neurological illness to have interfered, in some minor fashion, with his premorbid fluency in writing. Copying and writing on dictation were normal; given the patient’s educational level, near absence of spelling errors in the latter activity testified to its normalcy. Comprehension of Language Comprehension of spoken and written language remained normal throughout the evolution of E. Fr.‘s disease. This was true of all types of linguistic stimuli, including intricate and abstract ones. In January 1948, for instance, the 25 progressively more complex orders suggested by Weisenburg and McBride (1935) were executed without delays and without errors. Psychological Evaluation During the first trimester of 1948, E. Fr. was submitted to an impressive array of perceptive-motor and/or psychometric tests. All results were found to be within normal limits, including those in: Fould-Raven’s Progressive Matrices 38 (90th percentile); Weigl- Goldstein-Scheerer’s Color Form Sorting Test and Trist-Hargreaves’ Progressive Shapes Test [thus, in Goldstein’s (1948) terms, there existed no evidence of “disturbance of language by impairment of abstract attitude”]; Bender’s Visual Motor Gestalt Test; Rey’s Complex Design Test; Thurstone’s S Factor Test; Thurstone’s W Factor Test and Binois-Pichot’s Vocabulary Test (90th percentile); Wechsler’s Scale Similarities Subtest (90th percentile); etc. Essentially comparable results were obtained during the first trimester of 1949.5 considerable efforts, succeed in lifting the officer on the horse. But pafarras, the handsome officer falls on his head and his ceremonial uniform, the head ornament, the helmet that has fallen below the ears, his belly has popped out of his jacket, his knees that have stamped on the breeches. The dream is no longer realizable. The handsome officer is brought back by the young chore man, in whom the adventure causes an easily comprehensive hilarity.” 5 Details are discussed in Alajouanine’s et al. original report (1949). 98 LECOURS AND LHERMITTE DURAND’S PHONETIC STUDY OF E. Fr.‘S ARTHRIC DISORDER In the original E. Fr. case report, Marguerite Durand gives a systematic description of the recordings she made, on the Rousselot cylinder, of 14 French and six English words and short phrases repeated by the patient. She compares these segments to those produced, in the same conditions, by three students of the Znstitut Phonitique (“un franc&, un an&is d’oxford et un australien”). Durand’s chosen stimuli were the following (numbers in parentheses will be used for reference further in the text): (1): papa /papa/; (2): baba /baba/; (3): assis /asi/; (4): musique /myzik/; (5): anneau /ano/; (6): un agneau / &nar)o/; (7): la lampe llal&pl; (8): on coupe tout /3kuptu/; (9): baisse-toi /bestwa/; (10): 1’0s dur /l>sdyR/; (11): barre trop longue /baRtRolSg/; (12): un arbre /&naRbR/; (13): on ose /%oz/; (14): la bague /labag/; (15): a top /at>p/; (16): abate /abeit/; (17): too bad /tu:baed/; (18): easy /i:zi/; (19): easter /i:star/; (20): a plough /a plau/. Durand’s main findings on French segments [(l) to (14)] can be summarized as follows: (A) On the whole, the duration of phonemes is greater than normal (average duration 2.5 times that in the corresponding utterances of the French control). (B) Partial or complete devoicing of voiced consonants is frequent, e.g.: /b/ in (9) and /d/ in (10) are totally deprived of recordable vocal cord vibrations, i.e., lb/ += lpl and Id/ +z /t/; voicing of the initial /b/ in (2) is delayed until the second half of the emission, i.e., /b/-,= /pb/; cessation of the laryngeal vibrations occurs too soon in lzi of (13), i.e. (z( *= /zs/; etc. (C) Partial or complete denasalization of nasal phonemes is frequent, e.g.: /&/ in (6) and (12) and /T/ in (11) are totally deprived of nasal resonance, i.e., l&l += Ice/ and I??,/*= /I/; nasal resonance occurs abnormally late in /m/ of (4) and in /zr/ of (7), i.e., /m/ += /bm/ and /&/ += IaiiP; etc. (D) As in the case of the initial /p/ in (1) and in that of lpi in (7), the articulation of occlusive consonants is often abnormally strong. Complete defricativation of fricative consonants is also observed, e.g.: /z/ in (4) is transformed into an approximately corresponding occlusive, i.e., /zJ -1: /d/. Although typical of such cases, partial defricativation of fricative consonants, that is, abnormal production of affricates (e.g. /s/ += /ts/, 6 In Durand’s terminology, production of a ps~udodiphthong PURE ANARTHRIA 99 /zJ += /dz/, etc.), does not occur in E. Fr.‘s repetition of Durand’s 14 French segments.’ (E) Delateralization of laterals is also observed, e.g.: /l/ in (10) and /l/ in (11) are transformed into an approximately corresponding occlusive, i.e., /l/+=/d/. The articulation of/R/, as in (lo), is often abnormally strong. (F) Because of an instability in aperture and/or site of articulation, usually with some irregularity and lack of suppleness in palatal and/or laryngeal movements, vowels-such as, lyl in (4), /&/ in (7) /I/ and /y/ in (lo), etc.-are often transformed into pseudodiphthongs or pseudopolyphthongs. (G) The initial vowels of normal segments-such as /a/ in (3) and (5) l&i in (6), lY in (8), etc.-are often preceded by an additional (usually glottal) consonant. (H) Epentheses and kindred phenomena are observed, e.g., two explosions instead of a single one are recorded in relation with /pt/ of (8), i.e., the cluster is dissociated; two additional vowels are uttered in relation with /RbR/ of (12), i.e., /&naRbR/ *= /oenaRpaeRa/; etc. Although very few occurred in the course of the Rousselot sampling, other types of transformations were observed, e.g.: addition by reduplication and postpositioning of a phoneme, as in /papa/ + /papap/, substitution by retrograde assimilation, as in /pkeRe/ --+ /pRceRe/, substitution by dissimilation of the second phoneme of a pair, as in /tZtapopRe/ + &kapopRd, etc. As mentioned above, it was clear, to the ears of E. Fr. and of those who attentively listened to him, that his phonetic disintegration was more immediately apparent in his French than in his English utterances. Durand attempted to document the fact through Rousselot recordings. Besides observing several anomalies shared by both French and English segments -e.g., an added glottal consonant appears before the initial vowel in (15), (18), and (19); the diphthong in (16), /ei/, is transformed into a polyphthong; etc.-she found that English diphthongs, on the whole, appeared to be closer to English norm than French vowels were to French norm; that /z/ in English (18) appeared to be closer to norm than lzi in French (13); that intramorphemic /st/ in English (19) appeared to be closer to norm than intermorphemic /st/ in French (9); that final lri in English (19) appeared to be closer to norm than final /R/ in French (10). EVOLUTION E. Fr. was followed at la SalpPtri&e until 1952. In later years, he often complained that he was becoming absentminded and that his memory was declining. Although his speech flow became somewhat more ’ /s/ is represented represented. three times in these segments and lzi twice; lfi. /vi, is/ and /3/ are not 100 LECOURS AND LHERMITTE rapid, no dramatic change occurred with regard to his arthric disorder. An electroencephalogram recorded in 1952 was normal. The patient died of cardiac disease in or around 1957. An autopsy was performed and the cerebrum was kept in formalin. In 1963, the specimen was brought to la SalpZtrih-e and routinely processed and described by Professor JeanClaude Gautier. ANATOMICAL STUDY Artifactual dorso-ventral flattening of the cerebral hemispheres is manifest. A single old infarct is seen on inspection of the uncut brain; it destroys (macroscopically, to the exception of the opercular fringe) the inferior half of the left precentral gyrus. The lesion appears to impinge on the foot of the second but not on that of the third frontal convolution (Fig. 4). But for a very small ancient infarct (cortex and a few superficial U-fibers) in the depth of the left cingulate sulcus (arrow), coronal sections rostral to (Fig. 5A) and through Broca’s area (Fig. 5B) show no evidence of ischemic lesion, either macroscopically or miscroscopically. Microscopic examination at several levels confirms the foot of F3 to be intact; rostrally, the infarct destroys the cortex and immediate subcortex of the caudalmost few millimeters of the foot of F2. A section through the left precentral gyrus (Fig. 5C) shows the ischemic necrotic lesion to destroy the cortex of this structure, caudal to the third & second frontal gyri, as well as its immediate subcortex, including the U-fibers to and from the third & second frontal and postcentral gyri; sparing of a narrow opercular fringe is confirmed microscopically but, deep to it, the dorsalmost aspect of the insular cortex is destroyed. The latter part of the lesion spares the extreme capsule and deeper structures. Sections through the anterior (Fig. 5 D) and posterior (Fig. 5E) halves of the first temporal convolution show two small patches of ischemic cortical destruction in the insula (arrows); these appear to represent the caudalmost part of the infarct. The temporal gyri, including the planum temporale, are not involved; neither are the supramarginal and angular gyri (Fig. 5F). Some neuronal rarefaction is manifest throughout the cortical mantle; it does not predominate in any particular area. SUMMARY AND DISCUSSION After a single, probably embolic, stroke leading to ischemic necrotic softening of the inferior three fifths of the left precentral gyrus, sparing Broca’s area, and to patchy cortical damage in the left insula, a right-handed bilingual-English and French-adult developed pure anarthriu, that is, a temporary speech suppression followed by a phonetic disintegration syndrome without other evidence of spoken or written speech or language disturbance. Stabilization of the disorder occurred a few weeks after the initial suppression and period of improvement; at PURE ANARTHRIA B FIG. 4. Surface localization of E. Fr.‘s infarct. (4A) macroscopical specimen: (4B) reproduction of a drawing made by Professor Jean-Claude Gautier prior to brain cutting. Comments in the text. this point, it was thought that the patient’s arthric anomalies were more manifest when he spoke French than when he spoke English. When first looked for and recorded in the patient’s hospital dossier, i.e., 6 weeks after the onset of the arthric disorder, no bucco-facial apraxia was observed. A thorough neurological, psychological, and linguistic study of the case was done by Th. Alajouanine, P. Pichot, and M. Durand; as far as we know, their publication in l’Enci?phale (1949) constitutes the first neuro- FIG. 5. Coronal sections through E. Fr.‘s left hemisphere. Loyez preparations. Comments in the text. 102 PURE ANARTHRIA 103 linguistic report on pure anarthria. Several questions are raised by their and our analysis of their data and by our anatomical study of the case. These questions concern, mainly, the significance of the observed dissociation between French and English utterances, the notion ofpureness in socalled pure anarthria, the phonetic characteristics of the dystonic phase of phonetic disintegration, the nosological situation of pure anarthria (within or without the realm of aphasia), the localization of cerebral lesions in patients with pure anarthria. We shall briefly discuss each of these points in turn. The English-French Dissociation On account of technical as well as theoretical drawbacks that have become apparent since the day’s of Durand’s experimental study of E. Fr.‘s phonetic anomalies -such as the relative inadequacy of the Rousselot recording device, the quantitative insufficiency of the studied sample, the functional incomparability of at least some of the very few specifically compared segments [e.g., /R/ in French (10) compared to /r/ in English (19)]-one might be tempted to give due credit to the work of a pioneer and to discard the English- French issue altogether. Among those who knew the patient, there was however unanimous by-ear agreement that his English phonetics, although quite abnormal, were less so than his French phonetics. Of course, a speech disorder a major characteristic of which is the production of diphthong-like segments is bound to be-at least superficially-more apparent to the listener’s ears in a language that prohibits diphthongs than in one that prescribes their frequent occurrence.* Yet, this was probably not the only reason of the by-ear agreement on E. Fr.‘s English vs. French phonetics. Although always resorting to a comparison between lesser vs. greater anomaly, and never to a comparison between normalcy vs. anomaly, such aphasic interlanguage dissociations do occur -involving phonetic and/or phonemic and/or syntactic and/or lexical aspects of speech and language-and are still in need of a satisfactory explanation. When suggested, this explanation will have to take into account the fact that the dissociation usually if not always becomes apparent only after a certain period of evolution and that, typically, it is more manifest in spontaneous productions, such as attempts at conversational exchange, than in more constraining linguistic activities. Although it is insufficient to say that earlier acquisitions are less “fragile,” an eventual theory of the problem might, at least partially, be based on date, mode, tempo, and intensity of apprenticeship. 8 After all, the ultimate sequella of phonetic disintegration, in certain cases where it stik.=-sFrench locutors, has long been known under the name of English pseudoaccent (pseudoaccent britanique). 104 LECOURS AND LHERMITTE To Alajouanine et al. (1949), E. Fr.‘s and similar cases indicate that phonetic disintegration, although exclusively disturbing the arthric aspect of spoken expression, occurs or can occur at a relatively “high level”s of cerebral organization. More frequently observed in phonetic disintegration, dissociations between more vs. less propositional language productions (e.g., conversation and narration vs. rote enumeration of the days of the week or prayer recitation) also support this point of view. At all events, it is unlikely that the study of interlanguage dissociations in aphasia, whether of the E. Fr. or of any other type, will ever provide ground for suggesting the possibility of anatomical indepp;ldence of the structures governing linguistic production in different languages. The “Pureness” of Pure Anarthria From a linguistic point of view, a triple dissociation (theoretically) characterizes the pure form of phonetic disintegration: Comprehension of language is normal but linguistic expression is not; within lingu’stic expression, written production is normal but spoken expression is lot; within spoken expression, the morphosyntactic and phonemic levels are normal, but the phonetic level is not (see Insert 1). Insert 1 PHONETIC LEVEL(or third articulation): the exclusive choice of a certain number of features-discrete actions of muscles of the bucco-phonatory apparatus-and their largely cotempoml integration into a more complex unit, the phoneme. PHONEMIC LEVEL (or second articulation): the exclusive choice of a certain number of phonemes and their serial integration into more complex segments the simplest of which being the morpheme. MORPHOSYNTACTIC LEVEL (or first articulafion): the exclusive choice of a certain number of morphemes and equivalent units and their serial integration into progressively more complex segments, the syntagms. (Buyssens, l%7; Martinet, 1%7; Lecours et al., 1970.) E. Fr.‘s behavior in comprehension tests testified to the existence of the first dissociation (expression vs. comprehension) from the very onset of his neurological disorder; once past the acute phase, its completeness was demonstrated when the patient was shown capable of 9 “C’est done dans la cohPsion et dans i’assemblage dynamique que constirue I’Clocution que reside le trouble. Aurremenr dir, il a lieu h un niveau beaucoup plus PlevP que la simple re’alisation morrice. II se produit done ti un niveau fort 6levk de l’impulsion articulatoire, ef c’est saris doute ce qui explique, dans une certaine mesure, la possibilite d’une dissociation qui serait incomprt?hensible b un niveau purement moteur” (Alajouanine et al., 1949). PURE ANARTHRIA 105 understanding, without delay, abstract, complex, and rapidly presented linguistic material. Because of the initial sensori-motor hemideficit and speech suppression, the existence of the other two dissociations could only be verified after a few weeks of evolution (early documents report no attempts at testing E. Fr.‘s ability to write with his left hand). In our opinion, the existence of the second dissociation (spoken vs. written expression) is unquestionable in this case: graphism proper was normal; no dysorthographic transformations were found in his numerous and lengthy productions, an argument of great weight in view of the high frequency of such transformations in nearly all other forms of aphasia; but for a few infelicities of style, nearly all of which were spontaneously rectified as in normal subjects of comparable educational level, choice and serial integration of written words into phrases and sentences were normal and, indeed, at times elegant (see Figs. 2 and 3 and corresponding sections in the text). The patient’s relative slowness in composing written essays is shared by a great number of brain-damaged subjects, whatever the site of their lesions, and by a nonnegligible proportion of normal persons as well. Discussion of the last dissociation (third vs. second and first articulations) comprises two aspects. On the one hand, it is certain that first articulation, i.e., choice and serial arrangement of the sense-making units of language, was entirely normal in E. Fr.‘s conversations and narrations; whether one considers his few lexical hesitations (e.g., “usine” vs. ‘fabrique,” c.f. antea) as belonging with the paraphasic component of normal language production (Freud, 1901,as translated and published in 1960; Fromkin, 1972) or as trivial semantic approximations does not modify the assertion. On the other hand, it is no less certain that E. Fr. produced a small number of transformations-e.g., /p-p/ -+ /p-p-p/ in /papap/ instead of /papa/, /l-RI -+ /R-RI in /pRceRe/ instead of /pkeRe/, cannot be /f-b/ + /p-f/ in /pafikae/ instead of IfabRiW, etc.‘O-that formally distinguished from the phonemic paraphasias found in patients having no arthric difficulties whatsoever, for instance, certain conduction aphasics (Lecours & Lhermitte, 1969; Blumstein, 1973; Lecours et al., 1973; Lecours & Caplan, 1974). In our opinion, most if not all patients with phonetic disintegration produce at least a small number of such transformations (Poncet et al., 1972). They probably correspond, at least partially, to what Alajouanine et al. (1939) termed the dyspraxic component of phonetic disintegration. Their presence in so-called pure anarthria leads to two questions: Are they less numerous in the language samples of normal locutors than in those of such patients as E. Fr.? If lo At least two of the transformations interpreted as phonetic distortions in Durand’s Rousselot sampling-in (IO), /I/ + /d/ in the presenceof id/, in (I I), /I/ -+ /d/ in the presence of /g/-could have, in other cases, been considered as typical phonemic paraphasias. 106 LECOURS AND LHERMITTE so, do they necessarily indicate the existence of a primary disorganization of the second articulation of language? The first question could only be answered through analysis of a sufficient control corpus. In the present stage of our knowledge, one’s suggested answer to the second question is mainly a matter of preconceptions; our own would lead us to a negative answer in cases such as E. Fr.‘s, where the production of a very small number of phonemic paraphasias would be the only argument against labeling the disorder as pure anarthria. Even if phonemic pat-aphasias were to be found more frequent in such patients than in normal speakers, it would remain possible or probable that any unspecific stress -for instance, that related to the existence of any brain disease, or that inherent to testing one’s linguistic abilities in a hospital situation-enhances any normal or abnormal locutor’s tendency to a limited paraphasic production. Be that as it may, the study of E. Fr.‘s speech inevitably leads to concluding either that he presented pure anarthria or that he presented nearly pure anarthria. Both conclusions imply that the neuronal nets governing the third articulation in spoken linguistic expression are endowed with at least relative anatomo-physiological autonomy and that their localization in the brain, although not precisely known, is discrete enough that focal damage to brain tissue can result in their isolated or nearly isolated dysfunction. The Phonetic Characteristics of the Dystonic Phase of Phonetic Disintegration From an aphasiological point of view, a phoneme is the audible result of the largely synchronous realization of a specific combination of dynamic states in the bucco-phonatory muscles. Such dynamic states -corresponding to muscle contractions of different degrees or to absence of contraction-are known as traits or features; the choice of specific features and their integration into phonemes are known as the third articulation of language (see Insert 1). Phonetic disintegration, whether occurring in isolation as in E. Fr.‘s case (Alajouanine et al., 1949), or representing but one semiological aspect of a more complex speech-language disorder, as is the rule in Broca’s aphasia (Alajouanine et al., 1939), can thus be conceived as an acquired disorganization of the third articulation of spoken language. In their 1939 monograph, Alajouanine et al. suggested that phonetic disintegration is the result of a triple motor dysfunction, paretic, dystonic, and dyspraxic (“un aspect paralytique, un aspect dystonique, un aspect apraxique”). What they meant by the last of these labels is not quite clear (it probably designates any aspect of the patient’s phonetic disorder that cannot be explained by lack of strength or hypertonia in bucco-phonatory muscles). Since they implicitly recognized, by PURE ANARTHRIA 107 reference to single phonemes, the variability in the phonetic performances of single patients, the meanings of the first two labels is apparently not to be taken in as absolute a manner as when used to qualify certain other neurological disorders. For instance, theparetic transformation of orals into nasals is certainly not as constant and clear-cut in most cases of (early) phonetic disintegration as in the bulbar forms of Charcot’s disease. Nevertheless, the frequent-indeed, characteristicevolution from a paretic to a dystonic phase, following a course of events not unlike that described by Twitchell (1951) in hemiplegia, validates the terminology and leads one to consider, in typical cases, the dystonic phase of phonetic disintegration as one of postparalytic reorganization. In such cases, this phase can thus be conceived as the phase d’ttat of phonetic disintegration. We will now try to define this phase d’ktat in terms of bucco-phonatory dynamics. Whether or not E. Fr.‘s utterances had the characteristics of the paretic phase at one time or another prior to Durand’s study is not known. At the time of this study, however, he certainly produced phonetic transformatiohs ‘pathognomonic of the dystonic phase. In our opinion, the most frequent and typical of these transformations can be understood as primarily related to a single dynamic dysfunction: a tendency to initiate bucco-phonatory movements in a state of exagerated muscular contraction followed by an anomaly in the dynamics of intermediary contraction corresponding, in the normal, to the distorted features. This anomaly can be an impossibility to attain the state of intermediary contraction, a delay in attaining it, an intermittence in realizing it, an early return to the initial stage of exagerated contraction.” In all cases, the latter tendency to keep or resume the initial abnormal dynamic state is constant. Thus, on the one hand, dynamic states in which relatively intense muscular contraction is needed for normal feature production are either normal or, as it were, “abnormally normal”: oral phonemes remain oral; voiceless ones remain voiceless; occlusive consonants remain occlusive, often exageratedly so, i.e., with greater than normal plosion [e.g., /p/ in (7)]. On the other hand, dynamic states in which muscular contraction of intermediate intensity is needed for normal feature production are very often (but not always) involved. Fricatives are a good example: they are frequently transformed into their approximate corresponding occlusive, indicating an impossibility to attain intermediate contraction that would permit, for instance, apico-alveolar frication [e.g., /zl in (4)], or else into affricates, witnessing to a delay in attaining intermediate contraction [e.g., /yza3/ -+ = /ydzaM] or to an early return I’ This lack of suppleness in the execution of phono-articulatory movements. and the patients’ typical attempts at on-the-spot corrections, probably explain, at least in part, the greater than normal length of the utterances in the dystonic phase of phonetic disintegration. 108 LECOURS AND LHERMITTE to the initial stage of dystonic occlusion. Although the involved agonists and antagonists are not exactly the same, the mechanism of transformation of apical laterals into approximate occlusives is probably of the same nature [e.g., /Y in (1 l)]. Likewise, absence of voicing in normally voiced phonemes [e.g., /b/ in (9)], or else its delay [e.g., /b/ in (2)], intermittence or abnormally short duration [e.g., /z/ in (13)] indicates similar dystonic defects in laryngeal muscle dynamics; and absence of nasal resonance in normally nasal phonemes [e.g., /g/ in (ll)], or its delay [e.g., /&/ in (7)], intermittence or abnormally short duration indicates similar disorder in velar activity. Comparable to the affricates, the production of pseudodiphthongs and pseudopolyphthongs often corresponds to a difficulty in reaching and maintaining or to intermittence in, the appropriate level of contraction in the muscles that determine the degree of buccal aperture; or else it is due to similar phenomena in labial, lingual, velar, and/or laryngeal muscles. Several features of a single phoneme can be concomitantly involved in such transformations, leading to a quantitatively more severe distortion of speech. Although the manifestation of the dysfunction is essentially motor, i.e., arthric, it is entirely conceivable that partial or complete deprivation of proprioceptive feedback from the bucco-phonatory apparatus, as presumably determined by site and extent of brain lesion in certain patients, plays a role in its genesis (Luria, 1964). If correct, the above analysis of the dystonic phase of phonetic disintegration would provide partial explanation for other characteristic phenomena, for instance, epenthesis and (usually glottal) occlusion prior to the emission of an initial vowel, but it would not, at least as a primary mechanism, account for the occasional production of phonemic-paraphasia-like transformations. Anarthria vs. Aphasia Pierre Marie’s intelligence and prestige were such, and his argumentation as to the necessity of excluding anarthria from the physio-pathological concept of aphasia was so forceful (Marie, 1906a, 1906b, 1907) that no general agreement on the problem-definitely not a primordial one-has been reached to this day. Our own position is the following: Since acquired disorders in comprehension of language and expressive disorders affecting the first and second articulations of spoken and written language, whether occurring more or less in isolation or in different combinations, are considered as aphasia, and since disorganization of the third articulation of spoken language, i.e., phonetic disintegration, or anarthria, is also recognized-or at least currently designated-as aphasia when it occurs in the semiologically more intricate context of Broca’s aphasia, we see no reason why isolated or nearly isolated disorganization of the third articulation, i.e., phonetic disintegra- PURE ANARTHRIA 109 tion in its pure form, or pure anarthria, should not be empirically considered as a relatively infrequent form of aphasia.12 Otherwise, one might be led to exclude several other clinical entities, for instance pure or nearly pure conduction aphasia, from the concept of aphasia, a step of doubtful value. In this domain, abiding by Baillarger’s (1865) and Dejerine’s (1901, 1906, 1914) views appears preferable. The Localization of Lesions in Pure Phonetic Disintegration Both Marie and Dejerine, the latter in early writings only, maintained that pure anarthria, or pure motor aphasia, is always the result of a subcortical lesion. For Marie (1906a, b, c), this lesion involved one part or another of the “zone lenticufaire”; for Dejerine (1901), it was immediately subcortical to Broca’s area, disconnecting it from “the rolandic operculum” and, therefore, from more peripheral motor white and gray. It is not entirely clear why Pierre Marie kept insisting that Broca’s area had nothing to do with speech production, and consequently, that capsule-lenticular lesions leading to anarthria had nothing to do, either directly or indirectly, with the outputs of Broca’s area. And it is not clear why Dejerine failed, at least for a while, to recognize that lesions other than the ones immediately subcortical to Broca’s area could interrupt the left hemisphere pathway giving Broca’s area access to more peripheral motor centers. In our opinion, it is possible that not all aspects of the quarrel had to do with brain structures and lesions. In any event, thinking of pure anarthria in terms of functional isolation of Broca’s area from more peripheral motor structures apparently remains a sensible-if not necessarily complete-approach. In order to isolate Broca’s area from both the left and the right precentral gyri a lesion probably has to involve either most of its cortex or most of its subcortex. Now, lesions strictly limited to the cortex of a neatly circumscribed area-that is, lesions leaving even the U-fibers intact-are excessively rare, in ischemic as well as in other diseases. Furthermore, even if the anatomical existence of such lesions was unquestionable, the quality of the information transmitted from damaged neurones into their supposedly intact axonal prolongations would raise serious questions. Thus, from a practical standpoint, one is left with two possibilities: either the lesion is cortico-subcorticol and it presumably leads to Broca’s aphasia, arthric disorder included; or it is subcortical only and, according to Dejerine (1901), it leads to pure motor aphasia. One might suggest that the latter is perhaps the case in some of the patients whose pure motor aphasia gradually emerges with evolution rather than existing I2 One in which, as in E. Fr.‘s case, there is usually no associated intellectual deterioration, a fact less frequently observed in other varieties of aphasia (Lhermitte et al., 1974). 110 LECOURS AND LHERMITTE straight off, in the absence of a comprehension disorder, and given contemporary neurological nosology, the existence of reduction in such cases would probably not change the diagnostic label (Alajouanine & Lhermitte, 1960). On the other hand, lesions of several other localizations can theoretically act to isolate Broca’s area from more peripheral structures in or through the left hemisphere. l3 Such are, among others, cortico-subcortical lesions of the inferior half of the left precentral gyrus and lesions immediately subcortical to this area, as well as lesions of some parts of the capsular components of Pierre Marie’s quadrilateral. In E. Fr.‘s case, isolation of Broca’s area from the left precentral gyrus was presumably realized by destruction of most of their common U-fibers in their caudal ends and, unquestionably, by ischemic destruction of the cortex itself in the larynx-mouth portion of the left precentral gyrus. It is likely that these parts of the lesion played a role in determining the anomalies in the patient’s arthric behavior. It is not obvious, however, that they were the only determinant: it is possible, for instance, that involvement of the U-fibers between precentral and postcentral gyri-one role of whose in the normal presumably includes feeding proprioceptive information from the phono-articulatory apparatus back to motor cortical structures-took part, or could have taken part, in determining the characteristics of E. Fr.‘s abnormal behavior in speech production; furthermore, we know of no argument permitting to assert or deny that the cortical lesions in the insula were of some significance as to the patient’s phonetic disintegration. [Had the patient been Pierre Marie’s, the latter might have decided to extend lines A and B of his famous drawing to the cortical surface of the insula (Fig. l).] In view of the importance of the precentral gyrus lesion in E. Fr.‘s case14, one might raise a question as to how often such a lesion exists, associated with that of the third frontal in Broca’s aphasia with typical phonetic disintegration. In reviewing Dejerine’s anatomoclinical writings on motor aphasia, we were struck by the fact that both of the cases (Bernheim’s and Ladame’s, as discussed in Dejerine, 1906) on which he built his argument on Broca’s aphasia had, besides partial lesions of the foot of the third frontal, left precentral lesions almost identical to E. Fr.‘s (compare Figs. 4 and 6). Were similar lesions found to be the rule rather than the exception, new questions conI3 Cases like E. Fr.‘s suggest that, in spite of the “bilaterality” of the primary cortical representation of phono-articulatory muscles, lesions of the left precentral gyrus can lead to permanent arthric disorder. I4 In the context of the 1972 meeting of the W. F. N. Research Group on Aphasia, Professor Eberhard Bay reported an anatomo-clinical case in which, as in E. Fr.‘s, pure anarthria appeared to be the result of an isolated cortico-subcortical lesion of the left precentral gyrus. PURE ANARTHRIA 111 FIG. 6. Dejerine’s representations of the surface lesions in Bemheim’s (6A) and Ladame’s (6B) cases, the latter after Von Monakow. From La Presse MPdicale, July 18, 1906. cerning the physiopathological mechanism of phonetic disintegration in Broca’s aphasia might be raised.15 In summary, one might suggest that damage to different levels of a complex although discrete neuro-axonal net can lead to phonetic disintegration, pure or otherwise. Whether or not careful linguistic studies could permit clinical distinctions between the effects on speech of lesions at different levels of this net remains, in our opinion, to be demonstrated. REFERENCES Alajouanine, Th., Ombredane, A., & Durand, M. 1939. Le Syndrome de De’sintPgrufion PhonPtique dam I’Aphasie. Paris: Masson. Alajouanine, Th., Pichot, P., & Durand, M. 1949. Dissociation des alterations phonetiques avec conservation relative de la langue la plus ancienne dans un cas d’anarthrie pure chez un sujet francais bilingue. .&c&hale, 28, 245-265. I5 It should be noted that E. Fr.‘s written expression (right hand), including graphism and orthography, remained normal or nearly so in the presence of cortico-subcortical destruction of most of the portion of the left precentral gyrus juxtaposed to the foot of the second frontal, and of a small caudal fringe of the latter structure itself. 112 LECOURS AND LHERMITTE Alajouanine, Th., & Lhermitte, F. 1960. Les troubles des activit6s expressives du langage dans I’aphasie. Leurs relations avec les apraxies. Revue Neurologique, 102, 66-91. Baillarger, J. G. F. 186.5,as reprinted in 1890. De I’aphasie au point de vue psychologique. Aphasie simple. Aphasie avec perversion de la faculte du langage. In J. Cl. F. Baillarger, Recherches sur les Maladies Mentales, Paris: Masson. Vol. I, Pp. 584-601. Blumstein, S. E. 1973. A Phonological Investigation of Aphasic Speech. The Hague: Mouton. Broca, P. 1861. Remarques sur le siege de la faculte du langage articule, suivies d’une observations d’aphtmie (perte de la parole). Bulletin de la Socihte d’dnthropologie, 2 (VI), 330-357. Buyssens, E. l%7. La Communication et I’Articulation Linguistique. Paris: Presses Universitaires de France. Charcot, J. M. 1883, as translated in 1884. Differente Formi d’Afazia; Lezioni Fatte nella Salpetriere nel Semestre d’Estate de1 I’anno 1883. Milano: Antica Casa Editrice Dr. Francesco Vallardi. Dejerine, J. 1901.Anatomie des Centres Nerveux. Paris: Rueff. Dejerine, J. 1906. L’aphasie motrice; sa localisation et son anatomie pathologique. Presse Medicale, 14,453-457. Dejerine, J. 1914. Semiologie des Affections du SystPme Nerveux. Paris: Masson. Freud, S. 1891, as translated in 1953. On Aphasia; a Critical Study, International Universities Press, New York. Freud, S. 1901, as translated in l%O. Slips of the tongue. In S. Freud, Psychopathology of Everyday Life. London: The Hogarth Press and The Institute of Psychoanalysis. Pp. 53- 105. Fromkin, V. A. 1971. The non-anomalous nature of anomalous utterances. Language, 47, 27-52. Goldstein, K. 1948. Language and Language Disturbances, New York: Grune & Stratton. Lecours, A. R., & Lhermitte, F. 1969. Phonemic paraphasias; linguistic structures and tentative hypotheses. Cortex, 5, 193-228. Lecours, A. R., Dotdain, Cl., & Lhermitte, F. 1970. Recherches sur le langage des aphasiques: I. Terminologie neurolinguistique. Encephale, 59, 520-546. Lecours, A. R., De&he, Cl., & Lhermitte, F. 1973. Paraphasies phonemiques; description et simulation sur ordinateur. In Colloques ZRZA-Znformatique Medicale Institut de Recherche d’lnformatique et d’Automatique, Rocquencourt. Pp. 31I-350. Lecours, A. R., & Caplan, D. 1975. Blumstein’s phonological investigation of aphasic speech. Brain and Language, 2,237-254. Lhermitte, F., Lecours, A. R., Ducame, B., & Escourolle, R. 1974. Unexpected anatomical findings in a case of fluent jargon aphasia. Cortex, 10, 83-96. Lichteim, L. 1885. iiber Aphasie. Deutsches Archivfir klinische Medizin, 36,204-208. Luria, A. R. 1964. Factors and forms of aphasia. In A. V. S. de Reuck & M. O’Connor (Eds.), Disorders of Language. London: J. & A. Churchill. pp. 143-167. Marie, P. 1906a, as reprinted in 1926. La troisieme circonvolution frontale gauche ne joue aucun role special dans la fonction du language. In P. Marie, Travaux et Memoires. Paris: Masson. Vol. I, pp. 3-30. (Originally published in Semaine Medicale, May 23, 1906.) Marie, P. 1906b, as reprinted in 1926. Que faut-il penser des aphasies sous-corticales (aphasies pures)? In P. Marie, Travaux et Memoires. Paris: Masson. Vol. I, pp. 31-64. (Originally published in Semaine Medicale, October 17, 1906.) PURE ANARTHRIA 113 Marie, P. 1906c, as reprinted in 1926. Un cas d’anarthrie transitoire par lesion de la zone lenticulaire. In P. Marie, Travaux et MCmoires. Paris: Masson. Vol. I, pp. 153- 157. (Originally published in Bulletins et MPmoires de la SocitQk MCdicale des H6pituux, December 14, 1906.) Marie, P. 1907, as reprinted in 1926. Sur la fonction du language. In P. Marie, Travaux et MPmoires. Paris: Masson. Vol. I, pp. 93- 113. (Originally published in Revue de Philosophic. 1907.) Martinet, A. 1%7. EIPments de Linguistique GPnCrak. Paris: Collin. Ombredane, A. 1950. L’Aphnsie et I’Elaboration de la Pensee Explicite. Paris: Presses Universitaires de France. Poncet, M., Degos, C., Deloche, G., & Lecours, A. R. 1972. Phonetic and phonemic transformations in aphasia. International Journal of Mental Health, 1,46-54. Twitchell, T. E. 1951.The restoration of motor function following hemiplegia in man. Brain, 74 (part IV), 443-480. Weisenburg, T., & McBride, K. E. 1935. Aphasia: a Clinical and Psychological Study, New York: Commonwealth Fund.