Crossed aphasia: An analysis of the symptoms, their frequency, and a comparison with left-hemisphere aphasia symptomatology
Introduction
The term crossed aphasia (CA) was coined by Bramwell (1899) to designate an acquired language disturbance caused by a lesion in the cerebral hemisphere ipsilateral to the dominant hand. Bramwell further stated that he was only aware of CA in a few rare cases of left-handed individuals. We now know that Bramwell was wrong on both counts: CA is not rare in left-handers, it is instead the rule; and CA cases in right-handers do exist. So, his term “crossed aphasia” survived the test of time but with a different meaning: Nowadays, it applies exclusively to aphasia following a right-hemisphere lesion in right-handed individuals. Crossed aphasia is rare and prevalence or incidence numbers are particularly difficult to pinpoint with a high degree of accuracy. The estimations of CA have varied significantly depending on the source: from 0% (Zangwill, 1960) to 18% (Mohr et al., 1980). To be sure, the latter prevalence figure stands as an outlier and was derived from a sample of patients with nonvascular etiologies. In the vast majority of estimates, however, CA prevalence is below 3% (Borod, Carper, Naeser, & Goodglass, 1985; Carr, Jacobson, & Boller, 1981; Gloning, Gloning, Haub, & Quatember, 1969; Roberts, 1969; Yinhua, 1996; Zangwill, 1979). Recently, Hojo (1999) reported a 1.1% prevalence rate for CA among 717 aphasia cases in a large Japanese rehabilitation hospital.
The fact that handedness and language lateralization are associated in a clearly nonrandom fashion prompted researchers to look for a genetic explanation for CA. Both traits were linked to neighboring alleles (Habib, Joanette, Ali-Cherif, & Poncet, 1983; Pillon, Desi, & Lhermitte, 1979) which could only be dissociated in very rare and unusual cases, such as in CA. Alexander and Annett (1996) applied Annett’s right shift theory to CA. The authors argued that the theory could predict CA by random lateralization in a small subset of the population in whom the right shift gene was absent (i.e., the “rs–” group). The genetic mechanism would exact early structural (i.e., anatomical) preferences that would set the stage for the development of certain abilities (or clusters of skills) in randomly assigned cerebral hemispheres. According to Alexander and Annett (1996), some language skills could therefore be dissociated. For example, phonological and semantic skills could lateralize differently. This hypothesis predicts, for instance, the existence of CA cases in which comprehension abilities are unexpectedly high because the lexical-semantic system remained in the left hemisphere (e.g., Osmon, Panos, Kautz, & Ganghavadi, 1998).
Bramwell (1899) had already hypothesized that CA must be due to heredity. He stated that the presence of left-handed ancestors would tend to favor language lateralization to the right hemisphere of the brain. This theory was later labeled “stock-brainedness” by Kennedy (1916). This theory stemmed from the fact that these early authors wrongly believed that left-handers had language lateralized in the right hemisphere. To be acceptable, such a theory should currently imply that the left-handed relatives of the CA patient are among the minority of left-handers whose language is lateralized in the right hemisphere. This hypothesis is still very much alive today as the “familial sinistrality” theory. However, more recent authors have cast doubt as to whether familial sinistrality is a variable powerful enough to modify the lateralization pattern of such a major skill as language (e.g., Bishop, 1990; Zangwill, 1979).
“Cultural factors” (Demeurisse, Hublet, Coekaerts, Derouck, & Capon, 1986) have also been invoked as triggers for CA. Bilingualism, for example, could cause a purported language lateralization to the right hemisphere according to the crowding hypothesis. Also, the use of a tone, ideographic, or sign language could allegedly recruit the right hemisphere for language processing because of that hemisphere’s known role in prosody, holistic processing capabilities, and visual processing abilities respectively. Finally, illiteracy could cause a lack of strong language lateralization because of the absence of written language processing. All these variables are naturally unable to explain the majority of CA cases. Furthermore, taken individually, these tentative explanations do not necessarily hold up to scrutiny (see Coppens & Hungerford, 1998, for discussion).
In addition to the search for a causative factor, researchers have also focused on pinpointing specific symptoms in CA. Essentially, the emphasis has been on three issues: first, finding a core set of symptoms that would characterize the syndrome of CA; second, identifying the frequency of specific characteristics and symptoms; and thirdly, finding symptomatology differences between CA and left-hemisphere aphasia. Naturally, these issues are related. The first issue focuses on defining CA as a set of specific symptoms circumscribing a separate clinical entity. One of the goals of this approach was to reach a differential diagnosis of CA by identifying a pathognomonic sign or a combination of defining symptoms. As a result, some researchers in the 1970s and early 1980s (Barroche, Marchal, Ascaillas, Rivail, & Lepoire, 1981; Barroche, Presles, Ramel, Weber, & Arnould, 1979; Brown and Wilson, 1976; Pillon et al., 1979; Urbain et al., 1978) argued that CA was typically characterized by acute mutism, followed by nonfluent aphasia with agrammatism regardless of lesion location in the right hemisphere. In addition, these authors also argued that a CA with jargon was never observed. Since then, many cases have been reported that contradict this characterization of CA. It was even noticed that the distribution of aphasia types was comparable between CA and left-hemisphere aphasia (Carr et al., 1981; Castro-Caldas & Confraria, 1984). It became progressively clear that CA could not be distinguished from left-hemisphere aphasia simply on the basis of an individual patient’s symptomatology. In other words, CA is not a separate clinical entity semeiologically in that a significant number of CA patients’ symptomatologies can be identical to those of patients with left-hemisphere lesions. On the other hand, some CA patients do display unusual or unexpected clinical characteristics. This observation led Alexander, Fischette, and Fischer (1989) to articulate the hypothesis that there are two types of CAs: the mirror-image CA, characterized by a symptom complex that could be expected from a similar lesion in the left hemisphere, and the anomalous CA in which a similar lesion in the right hemisphere would not give rise to the same symptoms. Implied in this distinction is that patients with a mirror-image CA have a language organization in their right hemisphere that is identical to that usually found in the language-dominant left hemisphere, whereas patients with an anomalous CA have either a different language organization (e.g., fluent aphasia with anterior lesion), or possibly an unexpected (i.e., incomplete) language lateralization, or both.
The second issue, namely attempting to quantify features of CA has produced very discrepant results. For example, the gender distribution was questioned. Urbain et al. (1978) reported 67% of females in their sample of CA patients, thereby contradicting contemporary publications (e.g., Joanette, Puel, Nespoulous, Rascol, & Lecours, 1982). These features have systematically led researchers to the third issue: identifying potential differences between CA and left- hemisphere aphasia. For example, CA patients’ mean age was debated. Some researchers reported that CA patients were younger than left-hemisphere aphasics (Donoso, Vergara, & Santander, 1980) whereas other researchers maintained that the ages of the two groups of patients was comparable (Castro-Caldas & Confraria, 1984). Three main potential clinical differences have also been described: a discrepancy between oral and written language impairments, the frequency of apraxia signs, and the presence of typical right-hemisphere disorders. Several authors argued that a discrepancy between written and oral language is a frequent characteristic of CA (e.g., Joanette et al., 1982). In most cases, written language is worse than oral language, but a number of cases have also been described with the opposite pattern. However, it is not clear whether this discrepancy appears more often than in left-hemisphere aphasia (Coppens & Hungerford, 1998). The same uncertainty applies to the presence of apraxic characteristics. As far as typical right-hemisphere signs are concerned, researchers have usually concentrated on constructional apraxia and neglect. The more recent description of two types of CA (Alexander et al., 1989) generates new and interesting questions: Is the frequency of these signs different for mirror-image and anomalous CA? Is one of these types more likely to contain patients with familial sinistrality? Only a large sample of CA patients will be able to generate reliable data to answer these questions. Such is the purpose of the present study.
Section snippets
Methods
A number of review articles have attempted to compile CA cases to highlight the frequency of specific symptoms in CA (Alexander et al., 1989; Castro-Caldas & Confraria, 1984; Castro-Caldas, Confraria, & Poppe, 1987; Coppens & Hungerford, 1998; Faglia, Rottoli, & Vignolo, 1990; Joanette et al., 1982; Urbain et al., 1978). Some authors have used all the potential explanations for CA, such as familial sinistrality or cultural factors, as exclusion criteria (e.g., Habib et al., 1983; Joanette et
Results
Table 1 presents the data in chronological order for the 167 case studies reviewed. Descriptive statistics revealed that the average age for the total sample was 57.34 years (n=164). Male subjects accounted for 66.1% of the cases and females for 33.9% (n=165).
The aphasia type distribution is as follows: 54 Broca (32.2%), 22 Wernicke (13.2%), 17 global (10.2%), 17 conduction (10.2%), 9 mixed transcortical (5.4%), 8 anomic (4.8%), 4 transcortical sensory (2.4%), and 3 transcortical motor (1.8%).
Discussion
This analysis of CA cases is the most exhaustive to date. It also includes for the first time a significant number of cases published in Japanese. The first set of analyses in this review focused on the frequency of appearance of certain characteristics and symptoms in the entire sample of CA patients. The mean age of the CA patients is similar to that of typical left-hemisphere aphasia (LHA). In fact, the average of 57.34 years is virtually identical to the CA average age (57.4 years) and the
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