The patient was a 62-year-old, right-handed man who had worked as a chef. He had received 12 years of education. He was urgently admitted to the hospital because of paralysis of the right upper and lower limbs. The patient was diagnosed with left putamen hemorrhage (size: 3 x 2 cm) (Fig. 1) and underwent nonsurgical treatment. In the next hospital, he underwent a physical and language rehabilitation program for about 3.5 months and was discharged home 93 days after onset. At 192 days after onset, the patient visited our hospital to begin language rehabilitation on an outpatient basis. The patient's Functional Independence Measure (FIM) score was 114/126 (motor FIM: 84, cognitive FIM: 30). Neurological findings included lucidity and right hemiplegia, and neuropsychological findings consisted of no nonverbal intellectual disability [Raven's colored progressive matrices (RCPM): 31/36] and subcortical aphasia. Written informed consent was obtained from the patient to publish information in a case report.
In the baseline evaluation, the patient's language and speech abilities were evaluated 1 month before and immediately before the start of CIAT. To evaluate the training effect, language function was assessed immediately after the end of CIAT and at intervals of 1 month, 3 months, and 6 months after CIAT ended. Language function, in which word frequency and imageability were controlled, was assessed by the Japanese version of the Western Aphasia Battery (WAB)) and the single-word-naming task in the Test of Lexical Processing in Aphasia (TLPA).) The Verbal Activity Log (VAL) was used to assess the patient's level of spontaneous real-world spoken language.,) We confirmed that the patient showed VAL scores of 3.0 points or less at 6 months or more after stroke onset and had moderate aphasia. We also confirmed the absence of the following factors: untreated serious illness, severe hearing loss, traumatic brain injury, and neurodegenerative disease.
The WAB Aphasia Quotient (AQ) was 75.6 at 1 month before CIAT and 77.3 immediately before CIAT. The patient's WAB score was 65/80 at 1 month before CIAT and 70/80 immediately before CIAT. The patient's spontaneous speech score declined, and we observed that he mainly uttered only words or two phrases when describing a picture. During word naming, once word finding became difficult for the patient, he was unable to resume speaking or answer the next question. Inconsistent articulation errors in the TLPA confirmed apraxia of speech. Given that some oral gestures, such as mouth opening, tongue protrusion, whistling gesture, blowing up cheeks, throat clearing, and tongue tapping, could be achieved upon verbal instruction, we judged that there was no oral apraxia. The patient showed good performance in yes/no questions and auditory word recognition for auditory verbal comprehension of the WAB, but the number of errors increased as the number of clauses increased in sequential commands. A previous study reported that the average change in AQ was 5.3 in patients with chronic aphasia.) Because the difference in AQ between 1 month before and immediately before CIAT was 1.7, we judged that the spontaneous recovery of language function had reached a plateau. The number of correct answers for the TLPA single-word naming was 138/200 (high frequency, 77; low frequency, 61) 1 month before and 129/200 (high frequency, 72; low frequency, 56) just before CIAT; therefore, we judged there was no recovery.
The learned nonuse of verbal language was confirmed by a reduction in spontaneous speech in daily life and scores below 2.5 on the two VAL scales.)
A Japanese version of CIAT,,) based on CIAT II,) was used in this case. The therapy sessions involved the following activities:
1. Confirmation of homework and review of spontaneous real-world spoken language from the previous day (30 minutes)
2. Word repetition drills (20 minutes)
3. Phrase repetition drills (20 minutes)
4. Language card game (40 minutes)
5. Picture description (20 minutes)
6. Role-play activity (30 minutes)
7. Home skill assignment (10 minutes)
During the word and phrase repetition drills, the patient repeated as much as possible the words and phrases that were familiar to him, such as food and drinks provided at his restaurant and the tools he used at his work. These words and phrases were also used in the later role-play task. When the patient was unable to repeat or name words correctly, we encouraged him to speak the word by: (1) talking about his episodes related to the word, and (2) repeating the word and short sentences using the word many times. The difficulties of the tasks were adjusted by changing the number, type, and position of the syllables. In the language card game, the patient and the therapist were each given about ten picture cards. In an effort to accumulate pairs, the patient and therapist asked each other for a matching card by using the question: "Do you have XX (name)?" This activity required the patient (and therapist) to name the object on the card. If the opponent had the specified card, they provided the card to the questioner and then aimed to make the same pair as soon as possible. In the picture description, the patient explained a landscape painting of daily life drawn in black and white. The therapist urged the patient to explain the details of the picture following his initial overview. If the patient was unable to recall the target word, then the therapist presented the first syllable of the word and the episode associated with the word as a hint for verbal output. In the role-play activity, verbal conversation was practiced while assuming the daily situations of patients who require communication. A "transfer package" (TP)) was also introduced to transfer the therapeutic gain of CIAT to daily communication. As the first step of TP, we clarified what the patient wanted to achieve by improving speech before the start of CIAT. The patient and his wife agreed to communicate verbally as a daily task during the CIAT program instead of using nonverbal communication. When confirming homework, the therapist checked the patient's schedule from the previous day and his use of speech in his daily activities. The therapist provided feedback each time and encouraged the patient to communicate by verbal language, as much as possible, using demonstrative pronouns and circumlocutions. The therapist also instructed the patient's wife not to speak on the patient's behalf.
The CIAT tasks were carried out for 3 hours per day, 5 days per week, for 3 weeks (45 hours). During the training, the patient was instructed not to use compensatory strategies, such as writing and nonverbal gestures, and he was encouraged to use only verbal language.
Immediately after CIAT, the WAB AQ had increased by 4.5 points from 1 month before therapy and by 2.8 points from immediately before therapy (Table 1). Immediately after CIAT and 3 months after therapy, the improvement from 1 month prior to treatment did not reach 5.3 points, which is the average change in WAB AQ in the chronic stage of aphasia.) However, at 6 months after therapy, the WAB AQ score had increased by 6.1 points from 1 month before CIAT. Improvements were noted in auditory word recognition, sequential commands, and word-naming scores.
The number of correct answers for the TLPA immediately after CIAT increased by 10 points from 1 month before treatment and by 19 points from immediately before therapy. In particular, the number of correct answers for high-frequency words tended to increase after treatment. However, these improvements for high-frequency words had decreased at 3 and 6 months after treatment. In addition, the error type of paraphasia and the number of occurrences of apraxia of speech in the TLPA were evaluated. The criteria used to classify the paraphasia error types and apraxia of speech were based on previous reports) as follows: 1) No response: no verbal response; 2) Phonological fragments: phonologically related to the target word; 3) Other phonological fragments: phonologically unrelated to the target word; 4) Formal paraphasia: not semantically similar but phonologically similar to the target word; 5) Mixed paraphasia: phonologically and semantically related to the target word; 6) Semantic paraphasia: semantically related to the target word; 7) Phonemic paraphasia: nonwords that are phonologically incorrect with the target word; 8) Monemic paraphasia: paraphasia consisting of two or more morphemes; 9) Irrelevant paraphasia: paraphasia that is semantically unrelated to the target word; 10) Perseverative paraphasia: paraphasia with verbal perseveration. Apraxia of speech was evaluated by nine characteristics, namely, sound substitutions, imprecise consonants, revisions, repetitions, prolongations, abnormal stress, slow rate, restricted pitch variation, and inconsistent errors.)
After CIAT, there were fewer errors in phonological fragments, semantic paraphasia, monemic paraphasia, irrelevant paraphasia, and apraxia of speech when compared with assessments 1 month before and immediately before therapy (Table 2). However, the number of errors in phonological fragments and semantic paraphasia after CIAT, monemic paraphasia after 3 and 6 months, and irrelevant paraphasia immediately after therapy only showed a one-point difference from that before CIAT; thus, there was no clear difference. Monemic paraphasia was not observed immediately after therapy but was observed again at 3 months after therapy. There was no significant change in errors of no response between before and after CIAT. However, instances of no response tended to become more frequent at 3 months after therapy when compared to immediately after CIAT. Errors of apraxia of speech were not observed immediately after CIAT.
The VAL score at 3 months after CIAT was 2.5 points or higher, which is the criterion for learned nonuse of verbal language. However, the VAL at 6 months after CIAT was obviously below this level. In an interview, the patient's wife revealed that the spread of COVID-19 had reduced the number of opportunities to go out or eat out. She also indicated that they used to spend time at home with friends before the outbreak but that their reduced number of visits had reduced opportunities for conversation.