Case Report How Long Is the Recovery of Global Aphasia? Twenty-Five Years of Follow-up in a Patient With Left Hemisphere Stroke Neurorehabilitation and Neural Repair 24(9) 871­–875 © The Author(s) 2010 Reprints and permission: http://www. sagepub.com/journalsPermissions.nav DOI: 10.1177/1545968310368962 http://nnr.sagepub.com Nicola Smania, MD1, Marialuisa Gandolfi, MD1, Salvatore Maria Aglioti, MD2, Paolo Girardi, PhD1, Antonio Fiaschi, MD1, and Flavia Girardi, SP3 Abstract Background. Current knowledge regarding the time course of aphasia recovery is based on observations limited to the first years after stroke. Objective. The authors studied long-term outcome (25 years) of language in a patient with global aphasia. Methods. A 37-year-old man with global aphasia from a large ischemic lesion in the left middle cerebral artery territory was tested 9 times between 3 weeks and 25 years poststroke by means of the Milan Language Examination, Token Test, Raven Test, and apraxia tests. Results. Three main periods of recovery were identified. The first year after stroke was characterized by recovery of verbal comprehension and word repetition. From 1 to 3 years, naming and reading improved. From 3 to 25 years, progressive improvement of previously emerged functions was found, as well as the appearance of spontaneous speech. Conclusions. This unique long-term follow-up shows that the time span for recovery of language functions in global aphasia after stroke may be much longer than previously documented. Keywords language, outcome, rehabilitation, cerebrovascular disorders Introduction Studies on aphasia recovery report that the greatest degree of language recovery takes place in the first months after stroke.1 Although the rate of recovery drops after 3 to 6 months, evidence of language improvement even 1 or 2 years after stroke have been reported in the literature.2,3 Only a small number of studies have followed patients with aphasia longer than 2 years poststroke.1,4-6 Therefore, current knowledge on the potential recovery of chronic aphasia is not sufficient. In this article, we report the case study of a patient with global aphasia who was tested in a wide range of language, intelligence, and praxic abilities for 25 years after stroke. Information drawn from this study may have important implications for aphasia therapy. Hospital, Verona, Italy. The computed tomography scan of his brain revealed a large ischemic cortical and subcortical lesion in the left middle cerebral artery territory. The patient had no risk factors for cerebrovascular disease and no previous history of cerebrovascular attacks. He was right handed and had 13 years of education. He underwent language rehabilitation for 2 years, 5 times per week in the first 6 months and then 3 times per week until the end of the second year. For many years, his only speech production was the nonsense word “musi.” Details of his brain lesion are presented in Figure 1. He was tested with the Milan Language Examination (MLE), the Token Test,8 the Raven Test,9 and tests for oral, ideational, and ideomotor apraxia10 at 3 weeks, 2 and 6 months, and 1, 2, 3, 10, 21, and 1 CRRNC, University of Verona, Italy Fondazione Santa Lucia IRCCS and Sapienza, Università di Roma, Italy 3 “C Santi” Rehabilitation Centre, Verona, Italy 2 Case Report In 1983, a 37-year-old man presented with acute onset of right hemiplegia, hemianesthesia, and language disturbances. He was admitted to the Neurology Unit of the GB Rossi University Corresponding Author: Nicola Smania, MD, Rehabilitation Unit, “GB Rossi” University Hospital, Via LA Scuro, 10, 37134 Verona, Italy Email: nicola.smania@univr.it 872 Neurorehabilitation and Neural Repair 24(9) description 15%). Significant correlations were found between time changes in these language functions: Comprehension– Repetition (r = .928, P < .001), Naming–Reading (r = .994, P < .001), Repetition–Naming (r = .867, P < .001), and Repetition–Reading (r = .874, P < .001). Discussion Figure 1. Brain magnetic resonance imaging performed 21 years after stroke. Lesion mapping showed involvement of Brodmann’s areas 22, 21, 20, 37, 44, 6, 6s, 4, 40, 39, 3, 1, 2, 5, and 7 (MRIcron software, see http://www.mricro.com/mricron) 25 years poststroke. An additional examination performed 3 years after stroke showed that spatial memory (Corsi BlockTapping test,11 span = 5, supraspan = 8) and selective attention (Attentional Matrices Test,12 total score = 42/60) were within normal ranges. Verbal memory was not testable because of the severity of his aphasia. MLE data were analyzed by means of descriptive analysis and a linear regression model (score = a + β log(time)) to estimate the trend of the MLE data over time. Spearman’s test between the main language functions (comprehension, repetition, naming, event description, reading) was performed by computing the raw scores of speech naming and event description and the mean scores of comprehension, repetition, and reading items of the MLE (P < .05). Statistical analysis was performed with R Package 2.8.1. Results MLE and other neuropsychological test data are given in Table 1. The MLE linear regression model showed a significant improvement in each language function (Comprehension R2 = 81.85, Repetition R2 = 96.67, Naming R2 = 79.13, Reading R2 = 78.68, Event description R2 = 60.36) over time. The rate of improvement was estimated at 1 year (Comprehension 70%, Repetition 50%, Naming 0%, Reading 0%, Event description 0%), 3 years (Comprehension 84%, Repetition 62%, Naming 24%, Reading 25%), and 25 years after stroke (Comprehension 100%, Repetition 85%, Naming 45%, Reading 46%, Event Results showed that recovery of global aphasia after stroke is not limited to just the first years following onset but can extend for even more than 1 decade. Overall, 3 main periods of recovery could be identified (Figure 2). The first period (first year after stroke) was characterized by recovery of verbal comprehension and word repetition, the second (1-3 years) by emergence of naming and reading, and the third (3-25 years) by a progressive improvement of previously emerged functions, as well as the appearance of spontaneous speech (event description). To illustrate the time course of recovery found in our patient, we will discuss each main language function tested by the MLE separately (Figure 2). Auditory comprehension was the first function to show recovery, in particular in the first years.2 However, different patterns of comprehension recovery were evident, depending on the contextual content of the tasks used. In the MLE comprehension subtests, in which the patient was challenged with simple and contextualized tasks (eg, to indicate which figure represents the everyday object verbalized by the speech therapist), the patient showed a rapid recovery, approaching normal performance in a 1-year time frame. On the contrary, a slower and more progressive recovery of performance occurred in the Token Test,3,13 which consists of very decontextualized and unusual verbal comprehension tasks (eg, to touch 1 or more tokens of different sizes and/ or colors; Table 1). Repetition also had its greatest rate of recovery in the first year, paralleling the trend of comprehension improvement. After the first year, repetition continued to slowly improve, reaching its highest level after 10 years. Naming of a visual item, which implies the ability to autonomously transform a visual–structural representation into a phonological output,14 emerged between 1 and 3 years after stroke and progressively improved, reaching 45% of performance in 25 years. It should be noted that reading had a trend of recovery equivalent to that of naming (Figure 2), suggesting that reading and naming may share elements of their neural substrates.15 Spontaneous speech (“Event description” in Table 1) emerged many years after stroke, first documented at 10 years, and then slightly improved over time. Although spontaneous speech production was limited to a few high-frequency words (house, bread, beard, etc), this complex language task implies that the patient reached the skill of autonomously selecting a lexical representation from 873 Smania et al Table 1. Results of Neuropsychological Examination Time From Stroke Milan Language Examination 3 Weeks 2 Months 6 Months 1 Year 2 Years 3 Years 10 Years 21 Years Speech Event description 0 0 0 0 0 0 1 2 0 0 0 0 10 20 35 55 Naming Comprehension Words 40 55 80 95 100 100 100 100 WSR 10 40 80 80 90 90 100 100 Sentences 10 10 45 90 75 100 100 100 Repetition Letters 0 80 90 100 100 100 100 100 80 80 70 90 100 100 100 100 Syllable Words 0 0 35 35 50 70 80 95 Neologism 0 0 20 50 30 30 50 80 0 0 0 20 0 0 30 60 Sentences Writing Text (letter) Unable Unable Unable Unable Unable Unable Unable Unable Naming 0 0 10 15 25 25 25 30 Reading comprehension Words 0 75 100 100 100 100 100 100 SWR 0 70 70 75 90 95 100 100 Sentences 0 0 80 80 80 80 80 80 Reading Letters 0 0 10 20 20 20 40 75 Syllable 0 0 0 0 15 50 50 70 Words 0 0 0 0 0 15 50 70 Neologism 0 0 0 0 0 0 20 20 Sentences 0 0 0 0 0 0 20 40 Dictation Letters 0 90 80 100 100 80 90 100 Syllable 0 20 0 70 90 60 80 100 Words 0 0 0 10 10 10 30 50 Neologism 0 0 0 0 0 10 10 10 Sentences 0 0 0 0 0 0 10 10 Copying Words 0 70 100 100 100 100 100 100 M-m 0 10 50 95 100 95 100 100 Other neuropsychological tests 7 7 10 14 18 22 26 29,5 Token Test Raven’s Test ND ND ND 17 ND 18 ND 26 Oral apraxia ND 15 15 20 16 20 20 20 Ideational apraxia ND 6 12 14 14 14 14 14 Ideomotor apraxia ND 63 60 69 65 52 ND 70 25 Years 2 50 100 100 100 100 100 95 80 40 Unable 40 100 100 100 60 70 70 30 40 100 100 35 10 10 100 100 30 28 20 14 72 Abbreviations: SRW, semantically related word; ND, no data. semantics, accessing the phonological word form, motor programming, and planning of articulation to say the word.14 The most challenging question is to understand why this patient demonstrated such improvements in the long-term. In the first years after stroke, we hypothesize that improvements may have been influenced by language rehabilitation, which took place 5 days per week in the first 6 months and then 3 times per week until the end of the second year.16 With regard to the progressive recovery seen in the long term, other factors, such as the patient’s strong motivation and active social participation,17 may also have played an important role. Indeed, motivation may have continuously stimulated the patient to 874 Neurorehabilitation and Neural Repair 24(9) Figure 2. Time course of recovery of language function over 25 years. MLE indicates Milan Language Examination express himself and to practice his linguistic abilities outside of a rehabilitative context.17 Further important factors possibly contributing to the recovery process may be the patient’s young age1,18 and the presence of relatively spared residual cognitive functions, such as memory and attention.19,20 This condition may have produced a favorable foundation for continued acquisition of new language abilities. Evidence emerging from the present case report may have implications for rehabilitation of language disorders after stroke. First, the results suggest that the time window for possible improvement in global aphasia may be much wider than previously believed. Based on this, it could be hypothesized that patients may benefit from language stimulation over the long term. Moreover, the behavioral changes seen in our patient suggest that possible care programs in chronic aphasia should take into account not only conventional aphasia training approaches but also other kinds of stimulation such as the stimulation of various nonlinguistic cognitive abilities19,20 and the creation of a context within a person’s everyday life that provides a sense of motivation and permits social interaction.17 The main limit of the present case report is that because this study was based only on clinical examination, it is not possible to identify how functional brain changes were involved in the process of long-term language recovery. In conclusion, the present study highlights that in patients with global aphasia there may be some potential for the emergence and improvement of linguistic functions even many years after stroke. Declaration of Conflicting Interests The author(s) declared no conflicts of interest with respect to the authorship and/or publication of this. 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