Abstract
Fragile X syndrome is a recently identified form of mental retardation that is associated with a chromosomal abnormality and inherited in an X-linked manner. Previous studies have suggested that distinctive speech and language characteristics are associated with the syndrome. Twelve adult male residents of an institution for the retarded (aged 23 to 51 years) were compared on a series of speech and language measures to 12 adult males with nonspecific forms of MR who were residents of the same institution and were matched on age and IQ. A second contrast group consisted of similarly matched autistic men. Results revealed that there were no significant differences among the groups' performance, with the exception of increased rates of echolalia in the autistic group. A nonsignificant trend toward poorer performance on expressive measures on the part of the fragile X group was noted. The implications of these findings for further research on the syndrome are discussed.
Similar content being viewed by others
References
Allen, W., Herndon, C., & Dudley, F. (1944). Some examples of the inheritance of mental deficiency: Apparently sex-linked idiocy and microcephaly.American Journal of Mental Deficiency, 48, 325–334.
Arthur, G. (1952).Leiter International Performance Scale-Arthur adaptation. New York: Psychological Services.
Blomquist, H., Bohman, M., Edvinsson, S., Gillberg, C., Gustavson, K., Holmgren, G., & Wahlstrom, J. (1985). Frequency of the fragile X syndrome in infantile autism.Clinical Genetics, 27, 113–117.
Brown, R. (1973).A first language. Cambridge: Harvard University Press.
Brown, W., Friedman, E., Jenkens, E., Brooks, J., Wisniewski, K., Raguthu, S., & French, J. (1982). Association of fragile X syndrome with autism.Lancet, 1, 100.
Carrow, E. (1973).Test of auditory comprehension of language. Hingham, Massachusetts: Teaching Resources.
Daker, M., Chidiac, P., Fear, C., & Berry, A. (1981). Fragile X in a normal male: A cautionary tale.Lancet, 1, 780.
De la Cruz, F. (1985). Fragile X syndrome.American Journal of Mental Deficiency, 90, 119–123.
DeRoover, J., Fryns, J., Parloir, C., & VanDenBerghe, H. (1977). X-linked recessively inherited non-specific mental retardation.Annales de Genetique, 20, 262–268.
Dunn, L., & Dunn, L. (1981).Peabody Picture Vocabulary Test-Revised. Circle Pines, Minnesota: American Guidance Service.
Escalante, J., Grunspun, H., & Frota-Pessoa, O. (1971). Severe sex-linked mental retardation.Journal de Genetique Humaine, 19, 137–140.
Fisher, M., & Zeaman, D. (1970). Growth and decline of retardate intelligence. In N. Ellis (Ed.),International review of research in mental retardation (Vol. 4). New York: Academic Press.
Fryns, J. (1984). Fragile X syndrome.Clinical Genetics, 26, 497–528.
Gardiner, M. (1979).Expressive One-Word Picture Vocabulary Test. Novato, CA: Academic Therapy Publications.
Goldman, R., & Fristoe, M. (1969).Test of Articulation. Circle Pines, Minnesota: American Guidance Service.
Hagerman, R., & McBogg, P. (1983).The fragile X syndrome. Dillon, Colorado: Spectrum.
Herbst, D., Dunn, H., Dill, F., Kalousek, D., & Krywanink, L. (1981). Further delineation of X-linked mental retardation.Human Genetics, 58, 366–372.
Howard-Peebles, P., Stoddard, G., & Mims, M. (1979). Familial X-linked mental retardation, verbal disability, and marker X chromosomes.American Journal of Medical Genetics, 7, 171–189.
Kleinberg, J., & Galligan, B. (1983). Effects of deinstitutionalization on adaptive behavior of mentally retarded adults.American Journal of Mental Deficiency, 88, 21–27.
Leckman, J., & Paul, R. (1985).Behavioral characteristics of the fragile X syndrome. Paper presented at the Symposium on Fragile X syndrome, University of Connecticut Health Sciences Department, Hartford.
Lehrke, R. (1974). X-linked mental retardation and verbal disability. National Foundation March of Dimes. New York: Intercontinental Medical Book Corp.
Levitas, A., Hagerman, R., Braden, M., Rimland, B., McBogg, P., & Matus, I. (1983). Autism and the fragile X syndrome.Developmental and Behavioral Pediatrics, 4, 151–158.
Lubs, H. (1969). A marker X chromosome.American Journal of Human Genetics, 21, 231–244.
Lubs, H., Watson, M., Breg, W. R., & Lujan, E. (1984). Restudy of the original marker X family.American Journal of Medical Genetics, 17, 133–134.
Martin, J., & Bell, J. (1943). A pedigree of mental defect showing sex linkage.Neurological Psychiatry, 6, 154–157.
Meryash, D., Cronk, C., Sachs, P., & Gerald, P. (1984). An anthropometric study of males with the fragile X syndrome.American Journal of Medical Genetics, 17, 159–174.
Meryash, D., Szymanski, L., & Gerald, P. (1982). Infantile autism associated with the fragile X syndrome.Journal of Autism and Developmental Disorders, 12, 294–301.
Miller, J. (1981).Assessing language production in children: Experimental procedures. Baltimore: University Park Press.
Newcomer, P., & Hammill, D. (1977).Test of language development. Austin, Texas: Empiric Press.
Paul, R., Cohen, D., Breg, W. R., Watson, M., & Herman, S. (1984). Fragile X syndrome: Its relation to speech and language disorders.Journal of Speech and Hearing Disorders, 49, 328–332.
Popovich, B., Vekemans, M., Rosenblatt, D., & Monroe, P. (1982). Fragile X.New England Journal of Medicine, 306, 1551–1552.
Renpenning, H., Gerrard, J., Zalewski, W., & Tabata, T. (1962). Familial sex-linked mental retardation.Canadian Medical Association Journal, 37, 954–956.
Snyder, R., & Robinson, A. (1969). Recessive sex-linked mental retardation in the absence of other recognizable abnormalities.Clinical Pediatrics, 8, 669–674.
Sutherland, G. (1977). Fragile sites on human chromosomes: Demonstration of their dependence on the type of tissue culture medium.Science, 197, 265–266.
Terman, L., & Merrill, M. (1973).Stanford-Binet Intelligence Scale (Form L-M). Boston: Houghton Mifflin.
Turner, G., Brookwell, R., Daniel, A., Selikowitz, M., & Zilibowitz, M. (1980). Heterozygous expression of X-linked mental retardation and X-chromosome marker fra(X) (q27).New England Journal of Medicine, 303, 662–664.
Turner, G., & Jacobs, P. (1983). Marker X-linked mental retardation. In H. Harris & K. Hirschhorn (Eds.),Advances in human genetics. New York: Plenum.
Turner, G., & Turner, B. (1974). X-linked mental retardation.Journal of Medical Genetics, 7, 407–415.
Watson, M., Leckman, J., Annex, B., Breg, W., Boles, D., Volkmar, R., Cohen, D., & Carter, C. (1984). Fragile X in a survey of 75 autistic males.New England Journal of Medicine, 310, 1462.
Webb, G., Rogers, J., Pitt, D., Halliday, J., & Theobald, T. (1981). Transmission of fragile (X)-(q27) site from a male.Lancet, 2, 1231–1232.
Yarborough, K., & Howard-Peebles, P. (1976). X-linked nonspecific mental retardation; Report of a large kindred.Clinical Genetics, 9, 125–130.
Author information
Authors and Affiliations
Additional information
We wish to express our most sincere appreciation to the staff of the Southbury Training School for their cheerful cooperation throughout this study, most especially to Edward Benjamin, Richard Witham, and Dr. Jean Gino. To our research nurse, Sharon Ort, research associate Wendy Marans, and research assistants Deborah Soriano, Elaine Algiers, and Karen Beebe, whose help in collecting, coding, and rating the data were invaluable, we also express our thanks. This research was supported by grants from the NIMH Mental Health Clinical Research Center grant no MR30929-09, the Children's Clinical Research Center grant no. RR00125-05, NICHD grant no. HPO3008-18, the MacArthur Foundation, the Merck Foundation and U.S. Public Health Service grant no. HD-11624.
Rights and permissions
About this article
Cite this article
Paul, R., Dykens, E., Leckman, J.F. et al. A comparison of language characteristics of mentally retarded adults with fragile X syndrome and those with nonspecific mental retardation and autism. J Autism Dev Disord 17, 457–468 (1987). https://doi.org/10.1007/BF01486963
Issue Date:
DOI: https://doi.org/10.1007/BF01486963