Anatomical Basis for Acquired Fluent Aphasia in Children Hugo R. VanDongen, PhD," M. Christa B. Loonen, MD,' and Krijn J. VanDongen, M D t Three girls aged 9 to 11 years developed fluent aphasia associated with acute brain lesions. As localized by computed tomography, the abnormalities i n all three resided in the posterior part of the left hemisphere, encroaching upon Wernicke's area. VanDongen HR, Loonen MCB, VanDongen KJ: Anatomical basis for acquired fluent aphasia in children. Ann Neurol 17:306-309, 1985 I t has been widely stated that acquired aphasia in childhood is invariably nonfluent, that is, that aphasic children have speech articulation problems, show syntactical errors, and speak in a telegraphic style; however, victims do n o t use neologisms or jargon and logorrhea is absent 12, 3, 9, 10, 14, 151. Exceptions have been observed 116, 20-221, but neither t h e characteristics of t h e aphasia nor the site of the lesion were documented in these cases. O v e r a period of four years, 27 children (15 boys and 12 girls; age range, 3 t o 15 years) referred t o our department for speech problems were classified as aphasic according t o published criteria 18, 18, 191. The most common cause was head trauma (9 patients), followed by vascular disorders (6),infectious diseases (4), brain tumors (4), and convulsive disorders and acquired aphasia (4). From this series we describe the language defect and radiographic findings in 3 righthanded girls, aged 9 t o 11 years, who had fluent aphasia. Before t h e onset of the aphasia, n o n e of t h e children had shown neurological signs or learning problems. suggesting a hematoma (Fig 1A). Seventeen days later the extent of the hematoma evident on C T scanning was grossly reduced, and the hemiplegia gradually resolved. Two months after onset the patient had a series of tonic-clonic seizures. Figure 1B shows the residual CT abnormalities one year after onset. Spontaneous speech was recorded in a conversational setting and rated for the following characteristics proposed for the classification of fluent speech: a phrase length exceeding four words, a rate of speaking of more than ninety words per minute, and normal prosody, articulation, pauses, and effort [ l , 11). The Table shows the speech characteristics of the patient before the onset of the seizures. Although neither paraphasia nor the use of neologisms was present during the interview, both were observed during the picture-naming test (forty items). Indeed, naming difficulties were so severe that the girl stopped the test after 10 trials. O n repetition testing she could repeat only three monosyllabic words. O n auditory comprehension testing using the Token test, she could perform only six of the ten items in the first (easiest) series. She was able to read aloud only four letters. Following the convulsions (mentioned earlier), there was further deterioration in her speech capacities; she spoke effortfully and could produce only simple words, and these in a telegraphic style. By this stage she could understand only simple commands, and detailed formal language examination (repeating, Token test, picture naming) was impossible. During the next two years virtually no improvement was observed. Patient 2 From the Divisions of *Neurology and tRadiology, Academic Hospital Rotterdam-Dijkzigt, 40 Dr Molewaterplein, 3015 G D Rotterdam, The Netherlands. At the age of 10 agirl suffered a CT-demonstrated contusion in the left temporal lobe just underneath a fracture as the result of a traffic accident (Fig 2A). The depressed fracture was corrected surgically six days after the accident, and one week later the patient was discharged home. The residual C T abnormalities are shown in Figure 2B. Detailed language examinations were carried out on the third and thirteenth days after onset. In her spontaneous speech (see the Table), as well as in picture naming, the patient's speech was marked by paraphasia. Repetition of polysyllabic nouns was impossible, as was the repetition of three-word sentences. Comprehension of spoken language during the interview was, however, considered normal, in contrast with the patient's poor score in auditory comprehension on the Token test (28 out of 61). Writing to dictation revealed many mistakes, including paraphasia. After craniotomy there was rapid improvement in spontaneous speech, picture naming, reading aloud, and writing to dictation. Recovery of the ability to repeat polysyllabic nouns and sentences was less pronounced, however, and the Token test still demonstrated a receptive disorder (score, 38 out of 61). Two and one-half months after the trauma, the girl was reexamined; the only deficit was found during writing to dictation, on which she showed some spelling errors. Six months later these errors were still present, as reported by her tutor, but the deficit disappeared within the following year. Received Feb 14, 1984, and in revised form July 13. Accepted for publication July 15, 1984. Patient 3 Address reprint requests to Dr Hugo R. VanDongen, Division of Neurology, Erasmus University Rotterdam, Rm EE 2287, PO Box 1738, 3000 DR Rotterdam, The Netherlands. An 11-year-old girl had an accident while cycling. O n admission neurological examination showed right knee and ankle jerks to be somewhat brisker than left, the only abnormality Case Reports Patient 1 A 9-year-old girl suffering from aplastic anemia suddenly developed language difficulties and a right hemiplegia. Computed tomographic (CT) brain scanning showed a hyperdense, space-occupying lesion in the left temporal region 306 A B F i g 1, Patient 1. (A)Computed tomographic scan at onset shows a hematoma posterior to the sylvian fismre. (Bi Scan one year after onset, demonstrating enlarged ventricles, dilated posterior horn, and possible calc$cation in the left temporo-occipital cortex. words, and her attempts at reading aloud were unintelligible because of her frequent use of neologisms. She could write only simple words. Understanding of verbal commands was considerably limited, as reflected in the Token test score of 13 out of 61. At discharge, twelve days after onset, paraphasia and the use of neologisms no longer occurred, but mild word-finding difficulties were still present. Repetition of single words was normal, although the patient was still unable to repeat five-word sentences. Verbal comprehension had become normal, as reflected in a Token test score of 52 out of 61. By thirty-four days after onset, only very mild word-finding difficulties were present, and one year after the trauma, her tutor reported normal school performance. Analysis of Spontaneous Speech Characteristic Patient 1 Patient 2 Patient 3 Phrase length (mean no. of words) Pauses Prosody Rate of speaking (wordshin) Effort Articulation 7.5 9.5 13.6 Normal Normal 121 Normal Normal Normal Normal 135 None Normal 96 None Mostly normal None Normal noted. C T scanning showed a small area of mixed density in the left posterior temporal region (Fig 3). Detailed language assessments were carried out on the fourth and twelfth days after onset. O n the fourth day the patient's spontaneous speech (see the Table) showed frequent paraphasias but only infrequent neologisms. Clinically her speech was empty of meaning, and the girl was found to be severely anomic. She could repeat only two monosyllabic Discussion The three reported children with fluent paraphasic speech challenge the view that acquired aphasia is always nonfluent and devoid of paraphasia C7, 141. A boy with a neologistic jargon aphasia is also known t o us; his utterances could not be analyzed because of the rapid emission of inappropriate words. Thus, 4 of 27 aphasic children admitted to our department during a period of four years showed a fluent aphasia. This discrepancy can perhaps be explained as follows: 1. In most studies of acquired aphasia in children, a clinical sign that raises the possibility of aphasia has been a right-sided paresis or paralysis; this motor Brief Communication: VanDongen et al: Fluent Childhood Aphasia 307 A Fig 3. Patient 3. Computed tomographic scan jive days after onset, demonstrating a small, mixed density contusion (arrows) in the left teinporal lobe behind the sylvian fi.f.sure. deficit has been caused by an anterior lesion, producing an associated nonfluent aphasia [22]. 2. The ability of the CT scan to demonstrate a left temporal lesion may lead to a more careful search for associated speech disturbances, increasing the recognition of fluent aphasia in children and differentiating it from confusion. 3. If head trauma is the underlying cause of the aphasia, recovery may be observed within a short time [13}, so that the fluent characteristics of the aphasia may be either not recognized or not recorded. B Fig 2. Patient 2. (A) Computed tomographic scan at onset shows a left temporal depressed fracture. a subgaleal hematoma in the vicinity ofthe fracture$anda mixeddensity contusion in the left temporal lobe posterior to the sylvian jssure. (B}Scan two months after onset, showing localatrophy in the posteriorpart of the left temporal lobe. 308 Annals of Neurology Vol 17 No 3 March 1985 The current view that the left temporal hemisphere is already specialized for language at birth { 5 } does not specify the influence of age, Sex, and location of the lesion on the clinical picture of childhood aphasia. The patients described here demonstrate that before 10 years of age, a language deficit resembling fluent aphasia in adults can occur; furthermore, repetition ability during recovery remained disproportionately severely impaired, suggesting a conduction aphasia, which is considered to be one of the fluent aphasias [S}. That the 3 reported patients are all girls raises the question of sex differences in the cerebral organization E S patient 1 k B patient 2 mpatient 3 I overlap 1 precentral gyrus 2 postcentral g y r u s 3 supramarginal g y r u s 4 s u p e r i o r temporal g y r u s 5 middle temporal g y r u s A f r o n t a l lobe €3 p a r i e t a l lobe C temporal lobe D occipital lobe right left right Fig 4. ksions evident on computed tomographic scanning at onset in patients I , 2, and 3 . The sylvianjssure was identified using the anatomical Ptudies of Matsui and Hirano {17} and of Gad0 and associates {6}, and taking into account slight d i f f e r ences in the plane of orientation. of language functions [4}.This is a controversial issue, but our findings, based on an admittedly small sample, do not support the claim that lesions posterior to the sylvian fissure rarely produce aphasia in women 112). The patients reported all have posterior lesions (Fig 4), i.e., lesions in Wernicke’s area. In the case of the 2 patients with head injury, however, the lesions could have been more diffuse. Moreover, we have observed a child with nonfluent aphasia caused by a glioma in the posterior part of the temporal lobe, indicating that posterior lesions do not invariably result in fluent aphasia. The authors thank Dr F. VanHarskamp, Mrs E. Yousef-Bak, and Mrs W. M. E. VandeSandt-Koenderman for their helpful advice, Dr D. N. Brooks for revising the English text, and Mrs J. DoornboschKonijn for preparing the manuscript. References 1. Benson DF: Fluency in aphasia: correlation with radioactive scan localization. Cortex 3:373-394, 1967 2. Brown JW: The neural organization of language: aphasia and lateralization. Brain Lang 3:482-494, 1976 Hecaen H: Lateralization and language representa3. Brown JW, tion: observations on aphasia in children, left-handers, and “anomalous” dextrals. Neurology (Minneap) 26:183-188, 1976 4. Brust JCM, Shafer SQ, Richter RW, et al: Aphasia in acute stroke. Stroke 7:167-174, 1976 5. Carter RL, Hohenegger MK, Satz P: Aphasia and speech organization in children. Science 2 18:797-799, 1982 6. Gad0 M, Hanaway J, Frank R: Functional anatomy of the cere- left bral cortex by computed tomography. J Comput Assist Tomogr 3:1-19, 1979 7. Geschwind N . Disorders of higher cortical function in children. In Cohen RS, Wartofsky MW (eds): Selected Papers on Language and the Brain. Boston Studies in the Philosophy of Science, Vol 16. Dordrecht, Holiand, Reidel, 1974, pp 467-481 8. Goodglass H, Kaplan E: The Assessment of Aphasia and Related Disorders. Philadelphia, Lea & Febiger, 1972 9. H6caen H: Acquired aphasia in children and the ontogenesis of hemispheric functional specialization. Brain Lang 3:114-134, 1976 10. H6caen H: Acquired aphasia in children: revisited. Neuropsychologia 2 1:5 8 1-587, 1983 11. Kerschensteiner M, Poeck K, Brunner E: The fluencynonfluency dimension in the classification of aphasic speech. Cortex 8:233-247, 1972 12. Kmura D: Sex differences in cerebral organization for speech and praxic functions. Can J Psychol 37:19-35, 1983 13. Lange-Cosack H , Tepfner G: Das Hirntrauma im Kmdes- und Jugendalter. Berlin, Springer Verlag, 1973 14. Lenneberg E: Biological Foundations of Language. New York, Wiley, 1967 15. Ludlow C L Children’s language disorders: recent research advances. Ann Neurol 7:497-507, 1980 16. Martins IP, Ferro JM: A case of acquired conduction aphasia in a child. Paper presented at the 9th Annual Meeting of the International Neuropsychological Society, Lisbon, Portugal, June 1983 17. Matsui T, Hirano A: An Atlas of the Human Brain for Computerized Tomography. Stuttgart, Fisher Verlag, 1978 18. VanDongen HR, Loonen MCB: Factors related to prognosis of acquired aphasia in children. Cortex 13:131-136, 1977 19. VanDongen HR, VanHarskamp R, Verhey-Stol FW, et al: Afasie onderzoek met de tokentest. Ned Tijdschr Psychol 28:633-647, 1974 20. VandeSandt-Koenderman WME, Smit IAC, VanDongen HR, et al: A case of acquired aphasia and convulsive disorder: some linguistic aspects of recovery and breakdown. Brain Lang 21:174-183, 1984 21. VanHout A: Importance of paraphasias in acquired aphasia in children. Paper presented at the 9th Annual Meeting of the International Neuropsychological Society, Lisbon, Portugal, June 1983 22. Woods BT, Teuber H-L: Changing patterns of childhood aphasia. Ann Neuroi 3:273-280, 1978 Brief Communication: VanDongen et al: FIuent Childhood Aphasia 309