Eur opean Rev iew for Med ical and Pharmacol ogical Sci ences 2015; 19: 81-85 Foreign accent syndrome: two case reports and literature review

H.-E. LIU 1,2 , P. QI 3, Y.-L. LIU 4, H.-X. LIU 5, G. LI 1

1Department of Neurosurgery, Qilu Hospital of Shandong University, Jinan, China 2Department of Neurosurgery, People’s Hospital of Binzhou City, Binzhou Medical University, Binzhou, Shandong, China 3Department of Rehabilitation Medicine, People’s Hospital of Binzhou City, Binzhou Medical University, Binzhou, Shandong, China 4Department of Neurosurgery,Binzhou Medical University Hospital, Binzhou, Shandong, China 5Department of Cardiovascular Medicine, Binzhou Medical University Hospital, Binzhou, Shandong, China

Abstract. Foreign accent syndrome is a lobe and haematoma was under left temporal changed accent syndrome mainly caused by subdural. After expectant treatment with drug, brain injury. This study is to report two cases she gradually turned to be conscious and speak - with foreign accent syndrome and their clinical able. But her accent was turned to be Mandarin. features. And we also point some lasted arti - cles to understand and discuss this disease in And with mild psychiatric symptom she can clinical manifestation, imaging, speak clearly and repartee. With recovery and pathology, pathogenesis, diagnosis and treat - language functional exercise, her accent was re - ment for the sake of further study. turned after 10 days. And MRI showed that left temporal lobe and lateral fissure was contusion Key words: and haematoma (Figure 1). After 3 months fol - low up, patient did not reappear different accent. Foreign accent syndrome (FAS), Hemisphere damage. A 25 year-old male was admitted to the hospital with head injury. He was . GC score is 6. Di - ameter of right and left pupil was 5 mm and 2 mm. Introduction Light reaction was disappeared in right and dull in left. Stimulating limb is able to flex and muscle ten - Foreign accent syndrome (FAS) is a brain injury sion high. Bilateral Pap levy was positive. CT caused accent syndrome. The patient changed the showed that contusion was in left temporal lobe , usual accent to another language or accent. It was and haematoma was in right side of the dura mater , firstly reported by Pierre Marie in 1907 that a Paris as well as intracranial pneumatocele (Figure 2). He patient was changed to London Se Shen regional was diagnosed with diffuse axonal injury and multi - accent due to right hemiparesis. In 1919, Pick re - ple brain contusion. After expectant treatment with ported a Czech became Polish accent after the left drug, he gradually turned to be conscious and hemisphere . So far, there were 70 patients speakable. His accent was turned to be Mandarin. with FAS has been recorded. The main causes in - He was able to speak clearly, repartee and self-care clude cerebrovascular disease, traumatic brain in - in daily life. After nerve function strengthen and jury, multiple sclerosis, neurodegenerative dis - verbal guidance, foreign accent was lasting 1 month eases, mental disorders, brain tumors, deep brain and gradually returned to the local accent. After 6 stimulation and some unknown reasons 1,2 . months follow up, foreign accent was disappeared. Due to disagree, he was not performed MRI.

Case report Discussion A 41 year-old female was admitted to the hos - pital with head injury. She was coma. GC score Clinical manifestation is 9. Eye Open: to pain; Verbal Response: none; FAS usually occurs in dominant hemisphere Motor Response: decerebrate extension. Brain damage. And its severity is not associated with in - CT showed that contusion was in left temporal cidence , onset time , duration and recovery. Tran

Corresponding Author: Gang Li, MD; e-mail: [email protected] 81 H.-E. Liu, P. Qi, Y.-L. Liu, H.-X. Liu, G. Li

Figure 1. Contusion and haematoma in left temporal lobe and lateral fissure was. and Mills 3 reported that a female case with severe onous, intermittent, not clear, and is similar to hypertension and toxic nodular goiter was admit - of verbal apraxia symptoms. FAS, ver - ted to the hospital with speech pattern change, bal apraxia and dysarthria ataxia may share a lower extremity weakness and mild headache. Di - common pathophysiological mechanism 7. agnosed with recurrent laryngeal nerve damage, FAS is a long range of duration . Most FAS pa - she left the hospital and came back after 3 tients never knew the changed accent or language months. Imaging shows subacute the left pontine before invasion. The condition of FAS in these infarction. So she was diagnosed as typical FAS. two cases did not last very long. With recovery of After a month, she appeared more severe infarc - the disease , they gradually turned to the local ac - tion, lower extremity weakness and slurred cent. Luzzi et al 8 reported a 64 year-old right- speech. This indicated that FAS, an atypical clini - handed Italian female. She appeared Spanish ac - cal symptom of cerebrovascular disease, can oc - cent for 3 years. Before that, she could only cur before the invasion. It can be among the first speak Italian, who never went abroad and learnt symptoms of multiple sclerosis 4. Abel et al 5 re - other . After 12 months follow up, she ported a 60 year-old female with left parietal lobe hesitated during speak, was occasionally voice was diagnosed with breast cancer metastasis. Be - paraphasia,can understand, and was diagnosed fore treatment, she was with FAS and dysarthria, with the variant of left frontotemporal lobar de - whilst after that, she was merely with FAS. generation. Bhandari 9 reported a 55 year-old It is still controversial that FAS is a kind of male ischemia and FAS for one day. There mild verbal apraxia or subtype. Roy et al 6 ana - are no abnormality in physical examinations and lyzed acoustic sound of two FAS francophone imaging. After recovery of epilepsy, FAS disap - cases. He pointed that their language feature is peared. similar to mild speech apraxia . Dronkers tracked the verbal apraxia site in central front of the Neuroimaging guide groove of dominant hemisphere language The damage in left frontal lobe, lower pari - areas insula . Cerebellar damage can cause lan - etal lobe, basal ganglia and frontal white matter guage disorder. It mainly manifests slow, monot - can be usually found in FAS patients. These

82 Two FAS case reports and literature review

Figure 2. Contusion was in left temporal lobe, and haematoma was in right side of the dura mater, as well as intracranial pneumatocele.

brain tissues are connected as an extensive bilat - volved in somatosensory area damage other eral cortical and subcortical networks, which than non -motor area cortex. The fact of a case plays an important role in language production. with left temporal lobe contusion caused FAS So the damage in these can cause the change of indicated that language center damage is the verbal order and disorder of speech rhythm, ren - main cause of FAS. But another case with dif - dering foreign accent 10 . It was also reported that fuse axonal injury had no language center dam - was involved in the formation of age, indicating that the damage in language FAS. Cohen et al 11 stated that a right-handed pa - sports network could lead to FAS. Further, tient with right frontal parietal lobe infarction Dronkers et al 13,14 used voxel-based lesion- was FAS for 3 years. After right cerebellar hem - symptom mapping (VLSM), which is a voxel- orrhage again, FAS was disappeared and speak - by-voxel brain–behavior mapping technique, to ing was turned to normal. This indicated that identify the associated brain areas. He suggest - the neural mechanism of language generation ed that middle temporal gyrus might particular - was complex and fineness. It needs to balance ly play an important role in comprehension at between bilateral cerebral and cerebellar. Every the word level 14 . Whilst, underlying white mat - change can influence on the whole movement ter, the anterior superior temporal gyrus, the su - language network. This also provided the neural perior temporal sulcus and angular gyrus, mid- basis for FAS intervention therapy and neuro - frontal cortex, inferior frontal gyrus is of impor - modulation. Kate et al 12 performed a study of tance at the level of the sentence 14 . Another MR structural imaging, which proved that FAS study 15 also pointed that articulatory planning patients always had some abnormal structure, deficits were associated with a cortical area be - including left superior temporal gyrus , middle neath the frontal and temporal lobes. However, frontal gyrus, bilateral cortical structures , hypo - other areas involved in speech movements also thalamus, left cerebellum . A cortical stimulation have the ability to regulate this complex ma - study 5, however, considered that FAS was in - chine 16 .

83 H.-E. Liu, P. Qi, Y.-L. Liu, H.-X. Liu, G. Li

Language pathology were diagnosed with craniocerebral trauma caused Patient with FAS is under a normal range of speech and accent memory disorders. But there IQ, short-term and long-term memory, naming, were two FAS cases without psychiatric history reading and spelling ability. Most of studies show and secondary brain injury existing FAS during an that FAS is due to language damage early stage of language development. So FAS was (Rhythm and tone ). Segmental and suprasegmen - reckoned as a developmental motor speech disor - tal features of language are closely associated der other than a clinical syndrome 20 . Some re - with FAS . In the segment level , production of searchers pointed that FAS was not caused by ver - vowels and consonants was affected . Vowel er - bal apraxia but basal ganglia movement language rors consist of vowel prolonged vowels , shorten - disorder and compensation of other sports cortical ing , taut . Consonant errors include changes in ar - language center network 21 . ticulation positions and ways , voiceless and dull - ness of plosive . In the superasegmental level, change of stress, rhythm and tone can cause high Diagnosis and treatment pitch, abnormal loud guttural friction and pro - nunciationavoidance 17 . We can evaluate phonolo - Diagnosis of FAS is based primarily on clini - gy and voice feature to get detailed data of tone, cal manifestations. Its features include rhythm which can also provide language pathology basis and sound segments damages, which is similar to for FAS treatment 18 . normal sound but different in accent before brain Some researchers studied about acoustical damage . FAS is now no specific effective treat - measurement , voice onset time (VOT), vowel du - ment . But multiple methods, such as DWI, PWI ration, fundamental frequency, duration conso - and MRI, may help us to understand the recov - nant in FAS patients. They pointed that the vocal ery, functional location and reorganization fur - contraction error , tense or relaxed vowels, vowel ther during rehabilitation 16 . With recovery, the formant variation were popular. Consonant accent usually can be returned to the original ac - changes included wrong articulation position and cent. During treatment of primary disease, it is way . Rhythm abnormalities included a variety of beneficial for language functional recovery to main acoustics, such as longer speech pause, stac - strengthen the language functional exercise and cato speech rhythm, etc. The most evident error psychological implications . was sound or silent reversed VOT of American English 19 . In normal condition, American English expresses long labial VOT when the audio pauses ––––––––––––– –––– ––– ––––– and short pause VOT while the silent pause . Declaration of Interest The authors report no declarations of interest . Pathogenesis Though the machine of FAS is still unknown, there are two hypotheses. One is that inhibiting the original neural circuits can cause “trace con - References ditioned reflex” in Pavlov doctrine. In other 1) POLAK AR, W ITTEVEEN AB, M ANTIONE M, F IGEE M, DE words, inhibited foreign accent nerve center KONING P, O LFF M, VAN DEN MUNCKHOF P, S CHUURMAN worked after local accent nerve center disorder. PR, D ENYS D. Deep brain stimulation for obses - The other is that local accent nerve center is de - sive-compulsive disorder affects language: a case stroyed but foreign accent nerve center is intact . report. Neurosurgery 2013; 73: E907-910. FAS is seen as a language, accent and memory 2) MENDIS D, H ASELDEN K, C OSTELLO D. Short case re - port: 'Speaking in tongues'--foreign accent syn - dysfunction syndrome . Formation and integrity of drome. Logoped Phoniatr Vocol 2013; 38: 79-81. language require an intact motor speech network. 3) TRAN AX, M ILLS LD. A case of foreign accent syn - After local accent memory loop is damaged, rela - drome. J Emerg Med 2013; 45: 26-29. tively static foreign accent loop continues to be ac - 4) CHANSON JB, K REMER S, B LANC F, M ARESCAUX C, N AMER tive and contacts with Broca’s centre, rendering IJ, DE SEZE J. Foreign accent syndrome as a first FAS 1. Damaged loop can be re -established during sign of multiple sclerosis. Mult Scler 2009; 15: rehabilitation so that the accent can be returned to 1123-1125. 5) ABEL TJ, H EBB AO, S ILBERGELD DL. Cortical stimula - local. This also explains the reason that the pa - tion mapping in a patient with foreign accent syn - tients with undamaged or minor damaged Broca's drome: case report. Clin Neurol Neurosurg 2009; centre can appear FAS. In our study, two cases 111: 97-101.

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