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The Journal of Genetic Psychology: Research and Theory on Human Development Publication details, including instructions for authors and subscription information: http://www.tandfonline.com/loi/vgnt20
Learning and Behavioral Correlates in Learning-Disabled Pupils Prone to Heterozygous Thalassemia and Sicklemia Alvin B. Reing
a b
a
Brooklyn College, City University of New York, School of Education , USA b
School of Education, Brooklyn College , City University of New York , Bedford Avenue and Avenue H, Brooklyn , New York , 11210 , USA Published online: 04 Sep 2012.
To cite this article: Alvin B. Reing (1975) Learning and Behavioral Correlates in LearningDisabled Pupils Prone to Heterozygous Thalassemia and Sicklemia, The Journal of Genetic Psychology: Research and Theory on Human Development, 127:2, 305-316, DOI: 10.1080/00221325.1975.10533959 To link to this article: http://dx.doi.org/10.1080/00221325.1975.10533959
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The Journal of Genetic Psychology, 1975, 127, 305-316.
LEARNING AND BEHAVIORAL CORRELATES I N LEARNINGDISABLED PUPILS PRONE T O HETEROZYGOUS THALASSEMIA AND SICKLEMIA*’** Brooklyn College, City University of N e w Y o r k , School of Ediication
ALVIN B. REING
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SUMMARY The pilot study was designed to vindicate planning for large-scale detection and management of two parallel genetic blood disorders occurring in large numbers of Mediterranean- and black Americans: the heterozygous minor traits of thalassemia (Cooley’s anemia) and sicklemia (sickle-cell anemia). Referred pupils (N = 191) matched on schools, age, and learning difficulties associated with learning disabilities were compared as to presumed ethnic origin (n Mediterranean = 80; n black = 64; n “others” = 47) and incidence of trait-related learning and behavioral characteristics. Group mean differences on the study’s criteria were found to be significant. Both minor-trait prone groups equally indicated the associative effects, while the control group without trait-predisposition showed a nonsignificant relationship, thus supporting the hypotheses and the need for large-scale research. A.
INTRODUCTION
The investigation explored the relationship between (a) type, incidence, and interaction of learning disabilities task-analyzed in children (b) activational, attentional, and visual-perceptual characteristics associated with the effects of two heterozygous anemic traits-thalassemia and sicklemia, and (c) the pupils’ ethnic extraction from groups genetically prone to the traits. The purpose was to obtain pilot study data to vindicate large-scale
* Received in the Editorial Office, Provincetown, Massachusetts, on July 2, 1974. Copyright, 1975, by The Journal Press. “Prone to” means here “likely to indicate.” The minor anemic traits studied are genetically transmitted, congenitally present, and do not develop or change in the affected individuals. Proneness implies that if systematic detection programs were standard, the subjects of the study would probably indicate the hemoglobin disorders. Requests for reprints should be sent to the author at the address shown at the end of this article. 305
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research, by showing that Mediterranean- and black Americans, susceptible to the major morbid forms (i.e., Cooley’s anemia, or sickle cell anemia, respectively) indicate in the corresponding, widely prevalent minor forms trait-related learning disorders common to both groups. The genetic theory outlined by Rossi (15) directly links genetics to learning disabilities, and attributes dyslexia, dysgraphia, and other pupil behaviors to gene-determined neurochemical lag. Throne (18), advocate of the opposing radical-behaviorist viewpoint, considers genes and all other intraorganismic variables as contingencies, not causes of dependentvariable responses. A compromising geneticist’s position, supported by Gottesman (6), is that genes determine the reaction range at a molecular level, by affecting phenotypic variations. In the present study, Rossi’s theory of the genetic causation of learning disabilities was tested on two ethnic school populations predisposed to similar inheritable minor blood traits, and on pupils matched except for blood-trait proneness. Of the two variant blood disorders, much more has been written, and many more programs funded, for sicklemia (i.e., sickle or crescent-cell anemia, or drepanocythemia), a genetic disease transmitted mostly by individuals from the black equatorial populations of the world (10). Such cells contain inadequate hemoglobin, the blood’s oxygen-carrying component, and have one-third to one-half the normal life-span, thus protecting the victim from malarial reinfection, since the parasites require normal cell-life to reproduce themselves. In an estimated 2 5 million black Americans, the major (homozygous) trait severely debilitates about one-intwenty, while the minor (heterozygous) trait affects one-in-ten or more, depending upon the sample studied and criteria applied (2). In contrast to sicklemia, considerably less public attention has been paid to thalassemia, its equally important counterpart in a similar sizable number of Americans of Mediterranean extraction (3). A complex group of genetic blood disorders, thalassemia is transmitted mostly by individuals from countries bordering the Mediterranean Sea, North Africa, and the Middle East. As in sicklemia, a deficient synthesis of hemoglobin, and shortened cell-life result in varying degrees of anemia and associative disorders. When the hereditary trait is heterozygous (i.e., thalassemia minor) mild subclinical anemia may occur, hence usually not reported and detected. In the United States, where 23 million people claim Italian ancestry, and over five million Greek descent, the major form is estimated to exist in at
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least 200,000 children. Spot surveys of Mediterranean-area descendants (5) reveal a 1:25 incidence of the major trait and from a 1 : 1 0 to as high as 1:s incidence of the minor trait. Equally affecting both sexes, thalassemia parallels sickle-cell anemia in general cause and effects. Similarly, it is believed to be a built-in defense against malaria, since the disease reduces the formation of red blood cells; those produced have a one-third to one-half life span, preventing the malaria parasite from further reproducing itself and disabling the host. The sickle-cell is sickle-shaped, while the equally flattened thalassemic cell resembles a sombrero; both contain considerably reduced oxygen-bearing hemoglobin. The incidence of the two debilitating blood disorders, sicklemia in black Americans, and thalassemia in Italo- and Greek-Americans, in both minor and major forms, is approximately the same, affecting almost an equally large number of individuals in a similar manner. Thalassemia major usually is evidenced during the first year of life. The earliest signs are pallor, listlessness, loss of appetite, and irritability. Subsequent symptoms are more pronounced: fatigue; weakness; slight degree of jaundice of the whites of the eyes; muddy-yellow skin color; fragile, undersized bones; advanced splenomegaly and hepatomegaly; enlargement of the heart; and, thinning of the skull. In the literature (1) the disease is also alluded to as: Cooley’s anemia; Mediterranean anemia; hereditary leptocytosis; erythroblastic anemia; target cell anemia; or familial microcytic anemia. The more common heterozygous form, thalassemia minor, is usually recognized in adults when the homozygous disorder occurs and generates familial studies ( 1 6 ) . In routine blood examinations, the mild form is manifested as leptocytosis, minimal hypochromic microcutosis (4), reduced hemoglobin level, and a lower hemotocrit reading associated with a slightly increased red blood cell count. Further analysis usually reveals an increased osmotic resistance of erythrocytes and abnormal hemoglobins. According to Lichtman (1I ) , minor-trait thalassemic individuals have a normal life span without significant physical handicaps, enjoy normal health, and never develop the severe form. Nonetheless, resulting mild leptocytosis and its sequelae produce neurochemical imbalance and lag (IS), thereby reducing energy le\rel, cortico-reticular activation (12), and learning efficiency in pupils, and depending upon psychoneural mediation, associative learning deficits or disabilities. But, since the condition is most usually undetected in such children, except when the heterozygous condi-
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tion is found in a family member a n d a systematic laboratory study is undertaken, these behavioral and learning correlates are attributed instead to phenotypic (i.e., psychogenic o r sociogenic) causes (18). T h e investigator, in reviewing the medical and educational literature, found no previous research study relating the effects of thalassemia minor to classroom performance o r adjustment. For that matter, only medical but not psychometric research of the terminal major trait has been reported. As urged by Reing (13), large scale research is needed to assess the psychoeducational consequences traceable to the widely prevalent minor trait of thalassemia, as well as sicklemia, both of which reportedly affect nearly 55 million black and white Americans in the incidence noted. T h e present effort, therefore, was to conduct a limited pilot study to correlate and compare the learning and behavioral characteristics of both minor anemic traits, in order to show the importance of the effects of the heterozygous forms and thus justify the need for large-scale screening and geneticcounseling programs. I t was hypothesized that children with Mediterranean surnames, and black children ( a ) differ from other pupils also referred for psychoeducational evaluation, and ( h ) encounter similar incidence and prevalence of learning difficulties (i.e., reading, writing, and/or mathematics) associated with minor-trait anemic behaviors: i.e., fatigue-weakness, short attention span, and/or HDSA (hyperopic-diplopic-prestrabismicamblyopia). T h e HDSA pre-syndrome, first identified and studied by Reing ( 1 2 , 14), is a progressive binocular visual-perceptual dysfunction found in one-tenth of the normal school population, a n d in larger numbers in handicapped classes. T h e incidence, in the same population from which the present study’s subjects were obtained. is reported to be high because hyperopes continue to be neglected ( 7 ) , since the schools continue to use the Snellen Chart for vision-screening without the convex lens correction required for detecting farsightedness. T h e HDSA criterion was selected for this study because ( a ) it has been linked causatively to specific learning disabilities affecting reading, writing, and mathematics; ( h ) it increases in severity under conditions of lowered vitality, attention, and visual-neural efficiency; and ( c ) it is readily detected by a strabometric-synoroptic or task-analytic procedure ( 1 2 ) . T h e other behavioral criteria, fatigue-weakness and short attention span due to irritability and reduced energy level, are characteristic of heterozygous anemia (9). T h e learning criteria were those commonly reported for pupils
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with academic difficulties: reading, writing, and/or mathematics. Although the referred pupils were task-analyzed (20) for specific learning disabilities (i.e., visual dyslexia or bradylexia, dysgraphia, and/or dyscalculia, respectively), these variables were listed as areas of characteristic learning difficulties, in the absence of neurophysiological clinical data to establish them as “soft signs” (17).
B.
METHOD
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1. Subjects Over a period of three years, in three urban elementary schools, the investigator, using behavior-analytic and psychoeducational procedures, task-analyzed pupils referred by teachers, counselors, and supervisors for suspected learning and behavioral problems. All three schools serviced ethnically similar populations: 40 percent of the pupils of Mediterranean descent (Italian, Greek, Spanish, Israeli, Turkish); 35 percent black; and 25 percent of other ancestry. School policy, curricula, pupils’ age and sex, housing, family, and socioeconomic patterns were similar-most white children were from middle-income families, nearly all the black children were on public assistance. A total of 191 pupils were referred, more or less equally distributed from grades 1-6: 80 white with Mediterranean surnames (experimental group M); 64 black (experimental group B); and 47 “others” (control group 0).Compared to the ethnic populations of the schools, the referred pupils were approximately representative of their respective groups. In compliance with administrative policy, only referred pupils were observed and assessed psychoeducationally on the criteria of the study. 2.
Procedure
The children, identified only by their first names, were task-analyzed by the investigator, by means of psychoeducational tasks suggested by Valett (20), and sensory-perceptual procedures developed by Reing (12). Each child, individually, was given simple, curriculum-related reading, writing, and mathematics tasks, for the assessment of possible learning disabilities, and a synoptic task for the evaluation of intersensory coordination and visual-perceptual efficiency. Positive observational criteria were applied for judging as follows the child’s functioning on near and distant tasks (14),to confirm suspected HDSA: (a) distant vision-comparative alignment of the visual axes in binocular focus, photopic-scotopic pupillary adjustments for
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both eyes, orthophoria, flexible vergence, and eye-preference and ( 6 ) near vision-orthophoric binocular vision, binocular pupillary adjustments, flexible vergence, correct posture at 18 inches from the reading or writing task, less than 30 degree rotation, corresponding eye-hand laterality, and no symbol distortions, omissions, reversals, or fusion errors. After task-analysis and educational-diagnostic data were recorded, the children’s surnames were revealed, and specific classroom behaviors discussed in detail with the teacher. The reason for not identifying the surnames was to reduce if possible the investigator’s bias. Knowing beforehand the Mediterranean ancestry of the pupils, he inadvertently would associate the suspected learning disabilities and thalassemia minorsymptoms together. However, since the black, group 0, and many group M pupils indicated obvious ethnic features, such a procedure could not effectively control for selection bias. 3.
Statistical Analysis
In this ex post fucto correlational study, the prevalence and incidence of pupils in the three groups, indicating the main and interactive criteria of characteristic learning difficulties linked to specific learning difficulties, were first matched to the behavioral characteristics assumed common to heterozygous sicklemia and thalassemia. In Table 1, both the number of pupils so categorized and proportion (percentage) of their total ethnic group (M, B, or 0) are listed. Single-factor and interactive incidences are noted, along with cumulative frequencies. Visual inspection reveals the similar proportionate incidence of the learninglbehavioral correlates in groups M and B, and their marked difference from group 0’s data. Next, a one-way analysis of variance was performed on the findings for groups M , B, and 0 (i.e., the control group of matched pupils neither of black nor Mediterranean extraction). As shown in Table 2 , an F ratio of 12.0 (df = 2/18) was computed, significant at the stated .01 confidence points. Thus, as analyzed, the means of the populations, from which groups M , B, and 0 were obtained, were different with respect to the learning x behavioral correlates of the study associated with the minor traits of sicklemia and thalassemia. In order to test whether the mean differences between groups M , B, and 0 were significant, Scheff6 tests were performed (we Table 3). While the difference in means between the two minor-trait prone groups (M and B) was nonsignificant, their difference from the referred nonanemic prone group (0)was significant (t = 4.618 and t = 3.754, respectively) a t the .01 and .05 (for M), and .05 (for B) points.
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When the means from the Scheff6 tests were arranged in descending order, in Table 4, adjusted by error terms, both groups (M and B) yielded significant means (13.9 and 11.3) compared to control group 0, while groups M and B were not significantly different from each other.
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C. RESULTS The findings supported the two hypotheses of the study. Children with Mediterranean surnames, mostly of Italian or Greek extraction, as well as black pupils, differ from other pupils, also referred for psychoeducational evaluation, on common learning criteria (i.e., reading, writing, a n d o r mathematics) and behavioral criteria assumed associated with heterozygous anemia(i.e., fatigue-weakness, short attention span, and/or HDSA). Secondly, both groups encounter a similar incidence of difficulties on these criteria, significantly different from the norm population, unlike the control groups of otherwise matched, similarly referred pupils.
D.
DISCUSSION
As reported, children of Mediterranean descent (presumed from their surnames) are prone to thalassemia minor, and black children are genetically predisposed to sicklemia minor, both heterozygous traits with similar incidence and prevalence in the United States. The problem of the research was to study the differences or similarities between the two sizable anemia-prone groups of pupils, the norm population through an analysis of variance, and a matched referred group predisposed to neither anemia trait. The results indicated that the learnindbehavioral criteria of the study differentiated the three groups, equating both anemia-prone pupil populations, and underscoring their common need for large-scale detection and genetic-counseling services, since no treatment for remission has yet been found. The important limitation of the study was that it employed psychoeducational assessment procedures on statistically prone subjects, when direct blood examinations by medical personnel (i.e., hemoglobin electrophoresis) should have been conducted to confirm the traits’ probable causation of the criteria1 effects. Instead, the genetic relationships could only be claimed to be presumed. The investigator’s lack of authority, imposed by school regulations, to interview parents to determine family incidence of the minor trait, and to refer pupils for blood tests, greatly reduced the impact of the findings. However, in the case of six pupils from group M, and eight from group B, school referrals were later completed, revealing that five M and
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TABLE 1 INCIDENCEOF LEARNINGAND BEHAVIORAL CHARACTERISTICS OF REFERREDPUPILS Behavioral characteristics related to traits Characteristic learning difficulties Reading M"
Ba 0" Writing M B
0 Math M B
0 Reading x writing M B 0 Reading x math M B 0
Fatigueweakness
(F)
Short attention span (A)
n
%
n
%
8 6 3
10 9 6
9 6 4
11 9
6 4 2
8
I
6
11 1 3
HDSAa (H) n %
F x H
F x A
A x H
F x A x H n %
LI
Total
0
n Y i
n
%
n
%
n
%
1 3 2
1 5 4
5 4 3
6 6 6
1 2 2
1 3 4
39 33 20
49 52 43
1 2 1
1
4 6 6
1 3 1
1
3 2
3 4 3
2
21 26 16
34 41 34
2 2 2
3 3 4
3 4 2
4 6 4
2 2
3 3 4
36 35 21
45 55 45
>
8
8
10
11
5
8
9
6 7 4
9
2
4
4
4 4 3
5 6 6
5 3 2
6
6 4
9 9 9
14 11 6
8 10 5
10 16 11
4 4 4
5 6 9
6 6 3
l-
g 41
M
5 4 8
9 6
2
5
3
2 3
3 z
0
r 0
6 6 2
8 9 4
4 3
5 4 2
6 6
5
4
3
I
6
5
9 6 6
4
8 8 6
4
2
5 3
6 6 4
5 3 2
6 5 4
6 6 6
5 4 1
6 6
2
1
3 1
1 3 1
1 5 2
3 4 2
4 6 4
1 3 1
1 5 2
28 21 13
35 42 28
3 3 4
1 2
1 3 2
25 24 13
31 38 28
1
2
5
2
2
2
1
2
5 2 2
6 3 2
4
6 3
8 5 4
5 2
5
6 6 3
4 3 2
4
4 3 2
6 8
8 9 6
4
5 5
5 5 4
1
2
5
5 3 2
2
6 3
4
6 5
1 1
1
1 2 1 1
2 2
1
3 2
2 3 2
2 3 2
3 5 4
3 5 4
0 1 0
1 2 0
0 2 0
1 3 0
22 17 10
12
22
25
28 27 21
34 26
31
a N = 191. M = experimental group (n = 80) of white pupils with Mediterranean surnames. B = experimental group (n = 64) of black pupils. 0 = control group (n = 47) of “others” (neither M nor B). HDSA = Hyperopic-diplopic-prestrabismic-amblyopia (ex anopsia).
M B 0
Reading x writing x math
B 0
Writing x math M
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cc, c cc,
314
JOURNAL O F GENETIC PSYCHOLOGY TABLE 2 ONE-WAYANALYSIS OF VARIANCE Source
~~
~
-
ss
&
-
~
Between M , B , 0 Within M , B, 0 Total
~~
2 18
760 7 570 3
20
1331 0
F-
_ _ _M S
~
380 35 3 1 60
12 0-
N o t e ; M = experimental group of white pupils with Mediterranean surnames. B = experimental group of black pupils. 0 = control group of “others.” :’: 0 i.01. TABLE 3 MATRIXO F MEANDIFFERENCES W I T H SCHEFFETESTS _ _
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-~
~
~
-
~~~
TABLE 4 MEANSI N DESCENDING ORDER( S C H E F FTESTS) ~ Means
0
Group
_ _ _ ~
~
15.0
0
B
B
-
26.3 11.3“‘
M ~
28.9 13.9“‘ 2.6
M ~~
*
Error term t.O1 = 10.43
six B pupils did indicate the relevant minor trait after subsequent blood examinations, as reported by teachers. T h e assumed correlation between the study’s learning and behavioral criteria was supported by the data. The medical literature provides varying reports as to the effects of the minor traits, from mild to none. T h e present research, though correlational, found statistically significant main and interactive effects on specific pupil-and-trait related criteria. In view of the large number of Americans prone to the heterozygous anemic traits of thalassemia and sicklemia, and the preliminary findings of this pilot study, the planning and funding of large-scale screening and genetic counseling programs seem justified. T h e findings, in addition, appeared to support the genetic theory of Rossi ( 1 5 ) that links the etiology of specific learning disabilities to genotypic
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cause, mediated by neurochemical lag. Gottesman’s (6) position, that the reaction range of behaviors is gene-determined but affected also by phenotypic variables, was partly defended by the data, since HDSA, a trait-related behavioral criterion of the study, is a progressive (developmental) syndrome of measurable visual-perceptual dysfunction, as revealed in recent investigations (12, 13, 14). Throne’s (18, 19) approach emphasizes the potential for learning of any individual under extraordinary environmental conditions, regardless of inner status, and so could not be supported by the findings. The study’s results, furthermore, may serve to clarify the current unabated phenotype vs. genotype controversy. By correlating specific learning disabilities to a genetically transmitted intervening variable (i.e., susceptibility to minor blood traits and their effects), traced to comparable ecological adaptations in extensive Mediterranean and equatorial malarial regions in the world, the investigator was able to link criterion pupil behaviors to detectable and measurable ontogenetic factors, rather than to presumed ethnically differentiated intellect (8) or to poverty-related environmental and educational deprivation (18). For, in the present pilot research, matched black and white children affected by parallel genetic tendencies indicated similar learning/behavioral effects regardless of ethnicity, sociocultural criteria, or intellectual differences that could be possibly predicted.
REFERENCES ASIMOV,I., et al. Stedman’s Medical Dictionary. Baltimore, Md.: William & Wilkins, 1961. Pp. 1518-1519. 2 . FINCH,C. A. Pathophysiologic aspectsofsickle cellanemia Amer. J. M e d . , 1972,32,1-6. 3. FREEDMAN,A. The origin of Cooley’s anemia. Cooley’s Anemia Blood and Research Foundation for Children, New Hyde Park, New York, 1973. 4. GELLIS,S. S., FEINGOLD,M., & RUTMEN,J. Y. Atlas of Mental Retardation Syndromes: Visual Diagnosis of Facies and Physical Findings. Washington, D.C.: U. S. Dept. Health, Educ., & Welf., 1968. P. 115. 5. GERSHUNY, A. Ethnic diseases series: Cooley’s anemia. Queens Tribune-Flushing, North Shore, January 10, 1972, p. 22. 6. GOTTESMAN, I. I. Biogenetics of race and class. In M. Deutsch, I. Katz, & A. R. Jensen (Eds.), Social Class, Race and Psychological Development. New York: Holt, Rinehart & Winston, 1968. 7. GROSVENOR, T. The neglected hyperope. Amer. J . Optom. 6. Arch. Amer. Optom., 1971, 48, 376-382. 8. JENSEN,A. R . How much can we boost ZQ and scholastic achievement? Haruard Educ. R e v . , 1969, 39, 1-123. 9. KEETON,W. T . Biological Science. New York: Norton, 1967. Pp. 542-543, 575. 1.
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10. KOGAN,B. A. Health: Man in a Changing Environment. New York Harcourt, Brace & World, 1970. Pp. 29, 541. 11. LICHTMAN,H. C . Cooky’s anemia. Cooky’s Anemia Blood and Research Foundation for Children, New Hyde Park, New York, 1972. 12. REING, A. B. The effects of oculomotor conditioning and perceptual training on impulsivity, mode of imagery, vocational aspiration, and educational achievement. (Doctoral dissertation, New York University) Ann Arbor, Mich.: University Microfilms, 1970. No. 70-15, 976. . Thalassemia major and minor: Health and educational aspects. Unpublished 13. paper presented at Brooklyn College Center for Inter-American Studies, December 11, 1972. 14. . In defense of the MR-LD model: Specific, reversible visual-perceptual dysfunction (HDSA). J S A S Cat. Select. Doc. in Psyckol., 1974, 4, 54. 15. ROSSI,A. 0.Genetics and learning disabilities. Bekav. Nezrvopsyckiat. 1972, 4,2-7. 16. SCHWARTZ, E . Control of globin synthesis in thalassemia. Cooky’s Anemia Newsletr, 1973, 4(7), 1. 17. SMALL,L. Neuropsychodiagnosis in Psychotherapy. New York: Bruner/Mazel, 1973. Pp. 122-136. 18. THRONE, J. M. Genetic factors in mental retardation: A radical behaviorist point of view. Ment. Retavd., 1972, 10(6),32-35. 19. _ _ . Learning disabilities: A radical behaviorist point of view. J. Learn. Disabil., 1973, 6(9), 543-546. 20. VALETT, R.E. The Remediation of Learning Disabilities: A Handbook of Psychoeducational Resource Programs. Belmont, Calif.: Fearon, 1967. ~
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~
School of Education Brooklyn College C i t y University of N e w York Bedford Avenue and Avenue H Brooklyn, N e w York 11210
The Journal of Genetic Psychology: Research and Theory on Human Development Publication details, including instructions for authors and subscription information: http://www.tandfonline.com/loi/vgnt20
Learning and Behavioral Correlates in Learning-Disabled Pupils Prone to Heterozygous Thalassemia and Sicklemia Alvin B. Reing
a b
a
Brooklyn College, City University of New York, School of Education , USA b
School of Education, Brooklyn College , City University of New York , Bedford Avenue and Avenue H, Brooklyn , New York , 11210 , USA Published online: 04 Sep 2012.
To cite this article: Alvin B. Reing (1975) Learning and Behavioral Correlates in LearningDisabled Pupils Prone to Heterozygous Thalassemia and Sicklemia, The Journal of Genetic Psychology: Research and Theory on Human Development, 127:2, 305-316, DOI: 10.1080/00221325.1975.10533959 To link to this article: http://dx.doi.org/10.1080/00221325.1975.10533959
PLEASE SCROLL DOWN FOR ARTICLE Taylor & Francis makes every effort to ensure the accuracy of all the information (the “Content”) contained in the publications on our platform. However, Taylor & Francis, our agents, and our licensors make no representations or warranties whatsoever as to the accuracy, completeness, or suitability for any purpose of the Content. Any opinions and views expressed in this publication are the opinions and views of the authors, and are not the views of or endorsed by Taylor & Francis. The accuracy of the Content should not be relied upon and should be independently verified with primary sources of information. Taylor and Francis shall not be liable for any losses, actions, claims, proceedings, demands, costs, expenses, damages, and other liabilities whatsoever or howsoever caused arising directly or indirectly in connection with, in relation to or arising out of the use of the Content.
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This article may be used for research, teaching, and private study purposes. Any substantial or systematic reproduction, redistribution, reselling, loan, sub-licensing, systematic supply, or distribution in any form to anyone is expressly forbidden. Terms & Conditions of access and use can be found at http://www.tandfonline.com/ page/terms-and-conditions
The Journal of Genetic Psychology, 1975, 127, 305-316.
LEARNING AND BEHAVIORAL CORRELATES I N LEARNINGDISABLED PUPILS PRONE T O HETEROZYGOUS THALASSEMIA AND SICKLEMIA*’** Brooklyn College, City University of N e w Y o r k , School of Ediication
ALVIN B. REING
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SUMMARY The pilot study was designed to vindicate planning for large-scale detection and management of two parallel genetic blood disorders occurring in large numbers of Mediterranean- and black Americans: the heterozygous minor traits of thalassemia (Cooley’s anemia) and sicklemia (sickle-cell anemia). Referred pupils (N = 191) matched on schools, age, and learning difficulties associated with learning disabilities were compared as to presumed ethnic origin (n Mediterranean = 80; n black = 64; n “others” = 47) and incidence of trait-related learning and behavioral characteristics. Group mean differences on the study’s criteria were found to be significant. Both minor-trait prone groups equally indicated the associative effects, while the control group without trait-predisposition showed a nonsignificant relationship, thus supporting the hypotheses and the need for large-scale research. A.
INTRODUCTION
The investigation explored the relationship between (a) type, incidence, and interaction of learning disabilities task-analyzed in children (b) activational, attentional, and visual-perceptual characteristics associated with the effects of two heterozygous anemic traits-thalassemia and sicklemia, and (c) the pupils’ ethnic extraction from groups genetically prone to the traits. The purpose was to obtain pilot study data to vindicate large-scale
* Received in the Editorial Office, Provincetown, Massachusetts, on July 2, 1974. Copyright, 1975, by The Journal Press. “Prone to” means here “likely to indicate.” The minor anemic traits studied are genetically transmitted, congenitally present, and do not develop or change in the affected individuals. Proneness implies that if systematic detection programs were standard, the subjects of the study would probably indicate the hemoglobin disorders. Requests for reprints should be sent to the author at the address shown at the end of this article. 305
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research, by showing that Mediterranean- and black Americans, susceptible to the major morbid forms (i.e., Cooley’s anemia, or sickle cell anemia, respectively) indicate in the corresponding, widely prevalent minor forms trait-related learning disorders common to both groups. The genetic theory outlined by Rossi (15) directly links genetics to learning disabilities, and attributes dyslexia, dysgraphia, and other pupil behaviors to gene-determined neurochemical lag. Throne (18), advocate of the opposing radical-behaviorist viewpoint, considers genes and all other intraorganismic variables as contingencies, not causes of dependentvariable responses. A compromising geneticist’s position, supported by Gottesman (6), is that genes determine the reaction range at a molecular level, by affecting phenotypic variations. In the present study, Rossi’s theory of the genetic causation of learning disabilities was tested on two ethnic school populations predisposed to similar inheritable minor blood traits, and on pupils matched except for blood-trait proneness. Of the two variant blood disorders, much more has been written, and many more programs funded, for sicklemia (i.e., sickle or crescent-cell anemia, or drepanocythemia), a genetic disease transmitted mostly by individuals from the black equatorial populations of the world (10). Such cells contain inadequate hemoglobin, the blood’s oxygen-carrying component, and have one-third to one-half the normal life-span, thus protecting the victim from malarial reinfection, since the parasites require normal cell-life to reproduce themselves. In an estimated 2 5 million black Americans, the major (homozygous) trait severely debilitates about one-intwenty, while the minor (heterozygous) trait affects one-in-ten or more, depending upon the sample studied and criteria applied (2). In contrast to sicklemia, considerably less public attention has been paid to thalassemia, its equally important counterpart in a similar sizable number of Americans of Mediterranean extraction (3). A complex group of genetic blood disorders, thalassemia is transmitted mostly by individuals from countries bordering the Mediterranean Sea, North Africa, and the Middle East. As in sicklemia, a deficient synthesis of hemoglobin, and shortened cell-life result in varying degrees of anemia and associative disorders. When the hereditary trait is heterozygous (i.e., thalassemia minor) mild subclinical anemia may occur, hence usually not reported and detected. In the United States, where 23 million people claim Italian ancestry, and over five million Greek descent, the major form is estimated to exist in at
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least 200,000 children. Spot surveys of Mediterranean-area descendants (5) reveal a 1:25 incidence of the major trait and from a 1 : 1 0 to as high as 1:s incidence of the minor trait. Equally affecting both sexes, thalassemia parallels sickle-cell anemia in general cause and effects. Similarly, it is believed to be a built-in defense against malaria, since the disease reduces the formation of red blood cells; those produced have a one-third to one-half life span, preventing the malaria parasite from further reproducing itself and disabling the host. The sickle-cell is sickle-shaped, while the equally flattened thalassemic cell resembles a sombrero; both contain considerably reduced oxygen-bearing hemoglobin. The incidence of the two debilitating blood disorders, sicklemia in black Americans, and thalassemia in Italo- and Greek-Americans, in both minor and major forms, is approximately the same, affecting almost an equally large number of individuals in a similar manner. Thalassemia major usually is evidenced during the first year of life. The earliest signs are pallor, listlessness, loss of appetite, and irritability. Subsequent symptoms are more pronounced: fatigue; weakness; slight degree of jaundice of the whites of the eyes; muddy-yellow skin color; fragile, undersized bones; advanced splenomegaly and hepatomegaly; enlargement of the heart; and, thinning of the skull. In the literature (1) the disease is also alluded to as: Cooley’s anemia; Mediterranean anemia; hereditary leptocytosis; erythroblastic anemia; target cell anemia; or familial microcytic anemia. The more common heterozygous form, thalassemia minor, is usually recognized in adults when the homozygous disorder occurs and generates familial studies ( 1 6 ) . In routine blood examinations, the mild form is manifested as leptocytosis, minimal hypochromic microcutosis (4), reduced hemoglobin level, and a lower hemotocrit reading associated with a slightly increased red blood cell count. Further analysis usually reveals an increased osmotic resistance of erythrocytes and abnormal hemoglobins. According to Lichtman (1I ) , minor-trait thalassemic individuals have a normal life span without significant physical handicaps, enjoy normal health, and never develop the severe form. Nonetheless, resulting mild leptocytosis and its sequelae produce neurochemical imbalance and lag (IS), thereby reducing energy le\rel, cortico-reticular activation (12), and learning efficiency in pupils, and depending upon psychoneural mediation, associative learning deficits or disabilities. But, since the condition is most usually undetected in such children, except when the heterozygous condi-
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tion is found in a family member a n d a systematic laboratory study is undertaken, these behavioral and learning correlates are attributed instead to phenotypic (i.e., psychogenic o r sociogenic) causes (18). T h e investigator, in reviewing the medical and educational literature, found no previous research study relating the effects of thalassemia minor to classroom performance o r adjustment. For that matter, only medical but not psychometric research of the terminal major trait has been reported. As urged by Reing (13), large scale research is needed to assess the psychoeducational consequences traceable to the widely prevalent minor trait of thalassemia, as well as sicklemia, both of which reportedly affect nearly 55 million black and white Americans in the incidence noted. T h e present effort, therefore, was to conduct a limited pilot study to correlate and compare the learning and behavioral characteristics of both minor anemic traits, in order to show the importance of the effects of the heterozygous forms and thus justify the need for large-scale screening and geneticcounseling programs. I t was hypothesized that children with Mediterranean surnames, and black children ( a ) differ from other pupils also referred for psychoeducational evaluation, and ( h ) encounter similar incidence and prevalence of learning difficulties (i.e., reading, writing, and/or mathematics) associated with minor-trait anemic behaviors: i.e., fatigue-weakness, short attention span, and/or HDSA (hyperopic-diplopic-prestrabismicamblyopia). T h e HDSA pre-syndrome, first identified and studied by Reing ( 1 2 , 14), is a progressive binocular visual-perceptual dysfunction found in one-tenth of the normal school population, a n d in larger numbers in handicapped classes. T h e incidence, in the same population from which the present study’s subjects were obtained. is reported to be high because hyperopes continue to be neglected ( 7 ) , since the schools continue to use the Snellen Chart for vision-screening without the convex lens correction required for detecting farsightedness. T h e HDSA criterion was selected for this study because ( a ) it has been linked causatively to specific learning disabilities affecting reading, writing, and mathematics; ( h ) it increases in severity under conditions of lowered vitality, attention, and visual-neural efficiency; and ( c ) it is readily detected by a strabometric-synoroptic or task-analytic procedure ( 1 2 ) . T h e other behavioral criteria, fatigue-weakness and short attention span due to irritability and reduced energy level, are characteristic of heterozygous anemia (9). T h e learning criteria were those commonly reported for pupils
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with academic difficulties: reading, writing, and/or mathematics. Although the referred pupils were task-analyzed (20) for specific learning disabilities (i.e., visual dyslexia or bradylexia, dysgraphia, and/or dyscalculia, respectively), these variables were listed as areas of characteristic learning difficulties, in the absence of neurophysiological clinical data to establish them as “soft signs” (17).
B.
METHOD
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1. Subjects Over a period of three years, in three urban elementary schools, the investigator, using behavior-analytic and psychoeducational procedures, task-analyzed pupils referred by teachers, counselors, and supervisors for suspected learning and behavioral problems. All three schools serviced ethnically similar populations: 40 percent of the pupils of Mediterranean descent (Italian, Greek, Spanish, Israeli, Turkish); 35 percent black; and 25 percent of other ancestry. School policy, curricula, pupils’ age and sex, housing, family, and socioeconomic patterns were similar-most white children were from middle-income families, nearly all the black children were on public assistance. A total of 191 pupils were referred, more or less equally distributed from grades 1-6: 80 white with Mediterranean surnames (experimental group M); 64 black (experimental group B); and 47 “others” (control group 0).Compared to the ethnic populations of the schools, the referred pupils were approximately representative of their respective groups. In compliance with administrative policy, only referred pupils were observed and assessed psychoeducationally on the criteria of the study. 2.
Procedure
The children, identified only by their first names, were task-analyzed by the investigator, by means of psychoeducational tasks suggested by Valett (20), and sensory-perceptual procedures developed by Reing (12). Each child, individually, was given simple, curriculum-related reading, writing, and mathematics tasks, for the assessment of possible learning disabilities, and a synoptic task for the evaluation of intersensory coordination and visual-perceptual efficiency. Positive observational criteria were applied for judging as follows the child’s functioning on near and distant tasks (14),to confirm suspected HDSA: (a) distant vision-comparative alignment of the visual axes in binocular focus, photopic-scotopic pupillary adjustments for
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both eyes, orthophoria, flexible vergence, and eye-preference and ( 6 ) near vision-orthophoric binocular vision, binocular pupillary adjustments, flexible vergence, correct posture at 18 inches from the reading or writing task, less than 30 degree rotation, corresponding eye-hand laterality, and no symbol distortions, omissions, reversals, or fusion errors. After task-analysis and educational-diagnostic data were recorded, the children’s surnames were revealed, and specific classroom behaviors discussed in detail with the teacher. The reason for not identifying the surnames was to reduce if possible the investigator’s bias. Knowing beforehand the Mediterranean ancestry of the pupils, he inadvertently would associate the suspected learning disabilities and thalassemia minorsymptoms together. However, since the black, group 0, and many group M pupils indicated obvious ethnic features, such a procedure could not effectively control for selection bias. 3.
Statistical Analysis
In this ex post fucto correlational study, the prevalence and incidence of pupils in the three groups, indicating the main and interactive criteria of characteristic learning difficulties linked to specific learning difficulties, were first matched to the behavioral characteristics assumed common to heterozygous sicklemia and thalassemia. In Table 1, both the number of pupils so categorized and proportion (percentage) of their total ethnic group (M, B, or 0) are listed. Single-factor and interactive incidences are noted, along with cumulative frequencies. Visual inspection reveals the similar proportionate incidence of the learninglbehavioral correlates in groups M and B, and their marked difference from group 0’s data. Next, a one-way analysis of variance was performed on the findings for groups M , B, and 0 (i.e., the control group of matched pupils neither of black nor Mediterranean extraction). As shown in Table 2 , an F ratio of 12.0 (df = 2/18) was computed, significant at the stated .01 confidence points. Thus, as analyzed, the means of the populations, from which groups M , B, and 0 were obtained, were different with respect to the learning x behavioral correlates of the study associated with the minor traits of sicklemia and thalassemia. In order to test whether the mean differences between groups M , B, and 0 were significant, Scheff6 tests were performed (we Table 3). While the difference in means between the two minor-trait prone groups (M and B) was nonsignificant, their difference from the referred nonanemic prone group (0)was significant (t = 4.618 and t = 3.754, respectively) a t the .01 and .05 (for M), and .05 (for B) points.
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When the means from the Scheff6 tests were arranged in descending order, in Table 4, adjusted by error terms, both groups (M and B) yielded significant means (13.9 and 11.3) compared to control group 0, while groups M and B were not significantly different from each other.
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C. RESULTS The findings supported the two hypotheses of the study. Children with Mediterranean surnames, mostly of Italian or Greek extraction, as well as black pupils, differ from other pupils, also referred for psychoeducational evaluation, on common learning criteria (i.e., reading, writing, a n d o r mathematics) and behavioral criteria assumed associated with heterozygous anemia(i.e., fatigue-weakness, short attention span, and/or HDSA). Secondly, both groups encounter a similar incidence of difficulties on these criteria, significantly different from the norm population, unlike the control groups of otherwise matched, similarly referred pupils.
D.
DISCUSSION
As reported, children of Mediterranean descent (presumed from their surnames) are prone to thalassemia minor, and black children are genetically predisposed to sicklemia minor, both heterozygous traits with similar incidence and prevalence in the United States. The problem of the research was to study the differences or similarities between the two sizable anemia-prone groups of pupils, the norm population through an analysis of variance, and a matched referred group predisposed to neither anemia trait. The results indicated that the learnindbehavioral criteria of the study differentiated the three groups, equating both anemia-prone pupil populations, and underscoring their common need for large-scale detection and genetic-counseling services, since no treatment for remission has yet been found. The important limitation of the study was that it employed psychoeducational assessment procedures on statistically prone subjects, when direct blood examinations by medical personnel (i.e., hemoglobin electrophoresis) should have been conducted to confirm the traits’ probable causation of the criteria1 effects. Instead, the genetic relationships could only be claimed to be presumed. The investigator’s lack of authority, imposed by school regulations, to interview parents to determine family incidence of the minor trait, and to refer pupils for blood tests, greatly reduced the impact of the findings. However, in the case of six pupils from group M, and eight from group B, school referrals were later completed, revealing that five M and
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TABLE 1 INCIDENCEOF LEARNINGAND BEHAVIORAL CHARACTERISTICS OF REFERREDPUPILS Behavioral characteristics related to traits Characteristic learning difficulties Reading M"
Ba 0" Writing M B
0 Math M B
0 Reading x writing M B 0 Reading x math M B 0
Fatigueweakness
(F)
Short attention span (A)
n
%
n
%
8 6 3
10 9 6
9 6 4
11 9
6 4 2
8
I
6
11 1 3
HDSAa (H) n %
F x H
F x A
A x H
F x A x H n %
LI
Total
0
n Y i
n
%
n
%
n
%
1 3 2
1 5 4
5 4 3
6 6 6
1 2 2
1 3 4
39 33 20
49 52 43
1 2 1
1
4 6 6
1 3 1
1
3 2
3 4 3
2
21 26 16
34 41 34
2 2 2
3 3 4
3 4 2
4 6 4
2 2
3 3 4
36 35 21
45 55 45
>
8
8
10
11
5
8
9
6 7 4
9
2
4
4
4 4 3
5 6 6
5 3 2
6
6 4
9 9 9
14 11 6
8 10 5
10 16 11
4 4 4
5 6 9
6 6 3
l-
g 41
M
5 4 8
9 6
2
5
3
2 3
3 z
0
r 0
6 6 2
8 9 4
4 3
5 4 2
6 6
5
4
3
I
6
5
9 6 6
4
8 8 6
4
2
5 3
6 6 4
5 3 2
6 5 4
6 6 6
5 4 1
6 6
2
1
3 1
1 3 1
1 5 2
3 4 2
4 6 4
1 3 1
1 5 2
28 21 13
35 42 28
3 3 4
1 2
1 3 2
25 24 13
31 38 28
1
2
5
2
2
2
1
2
5 2 2
6 3 2
4
6 3
8 5 4
5 2
5
6 6 3
4 3 2
4
4 3 2
6 8
8 9 6
4
5 5
5 5 4
1
2
5
5 3 2
2
6 3
4
6 5
1 1
1
1 2 1 1
2 2
1
3 2
2 3 2
2 3 2
3 5 4
3 5 4
0 1 0
1 2 0
0 2 0
1 3 0
22 17 10
12
22
25
28 27 21
34 26
31
a N = 191. M = experimental group (n = 80) of white pupils with Mediterranean surnames. B = experimental group (n = 64) of black pupils. 0 = control group (n = 47) of “others” (neither M nor B). HDSA = Hyperopic-diplopic-prestrabismic-amblyopia (ex anopsia).
M B 0
Reading x writing x math
B 0
Writing x math M
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cc, c cc,
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~~
~
-
ss
&
-
~
Between M , B , 0 Within M , B, 0 Total
~~
2 18
760 7 570 3
20
1331 0
F-
_ _ _M S
~
380 35 3 1 60
12 0-
N o t e ; M = experimental group of white pupils with Mediterranean surnames. B = experimental group of black pupils. 0 = control group of “others.” :’: 0 i.01. TABLE 3 MATRIXO F MEANDIFFERENCES W I T H SCHEFFETESTS _ _
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-~
~
~
-
~~~
TABLE 4 MEANSI N DESCENDING ORDER( S C H E F FTESTS) ~ Means
0
Group
_ _ _ ~
~
15.0
0
B
B
-
26.3 11.3“‘
M ~
28.9 13.9“‘ 2.6
M ~~
*
Error term t.O1 = 10.43
six B pupils did indicate the relevant minor trait after subsequent blood examinations, as reported by teachers. T h e assumed correlation between the study’s learning and behavioral criteria was supported by the data. The medical literature provides varying reports as to the effects of the minor traits, from mild to none. T h e present research, though correlational, found statistically significant main and interactive effects on specific pupil-and-trait related criteria. In view of the large number of Americans prone to the heterozygous anemic traits of thalassemia and sicklemia, and the preliminary findings of this pilot study, the planning and funding of large-scale screening and genetic counseling programs seem justified. T h e findings, in addition, appeared to support the genetic theory of Rossi ( 1 5 ) that links the etiology of specific learning disabilities to genotypic
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cause, mediated by neurochemical lag. Gottesman’s (6) position, that the reaction range of behaviors is gene-determined but affected also by phenotypic variables, was partly defended by the data, since HDSA, a trait-related behavioral criterion of the study, is a progressive (developmental) syndrome of measurable visual-perceptual dysfunction, as revealed in recent investigations (12, 13, 14). Throne’s (18, 19) approach emphasizes the potential for learning of any individual under extraordinary environmental conditions, regardless of inner status, and so could not be supported by the findings. The study’s results, furthermore, may serve to clarify the current unabated phenotype vs. genotype controversy. By correlating specific learning disabilities to a genetically transmitted intervening variable (i.e., susceptibility to minor blood traits and their effects), traced to comparable ecological adaptations in extensive Mediterranean and equatorial malarial regions in the world, the investigator was able to link criterion pupil behaviors to detectable and measurable ontogenetic factors, rather than to presumed ethnically differentiated intellect (8) or to poverty-related environmental and educational deprivation (18). For, in the present pilot research, matched black and white children affected by parallel genetic tendencies indicated similar learning/behavioral effects regardless of ethnicity, sociocultural criteria, or intellectual differences that could be possibly predicted.
REFERENCES ASIMOV,I., et al. Stedman’s Medical Dictionary. Baltimore, Md.: William & Wilkins, 1961. Pp. 1518-1519. 2 . FINCH,C. A. Pathophysiologic aspectsofsickle cellanemia Amer. J. M e d . , 1972,32,1-6. 3. FREEDMAN,A. The origin of Cooley’s anemia. Cooley’s Anemia Blood and Research Foundation for Children, New Hyde Park, New York, 1973. 4. GELLIS,S. S., FEINGOLD,M., & RUTMEN,J. Y. Atlas of Mental Retardation Syndromes: Visual Diagnosis of Facies and Physical Findings. Washington, D.C.: U. S. Dept. Health, Educ., & Welf., 1968. P. 115. 5. GERSHUNY, A. Ethnic diseases series: Cooley’s anemia. Queens Tribune-Flushing, North Shore, January 10, 1972, p. 22. 6. GOTTESMAN, I. I. Biogenetics of race and class. In M. Deutsch, I. Katz, & A. R. Jensen (Eds.), Social Class, Race and Psychological Development. New York: Holt, Rinehart & Winston, 1968. 7. GROSVENOR, T. The neglected hyperope. Amer. J . Optom. 6. Arch. Amer. Optom., 1971, 48, 376-382. 8. JENSEN,A. R . How much can we boost ZQ and scholastic achievement? Haruard Educ. R e v . , 1969, 39, 1-123. 9. KEETON,W. T . Biological Science. New York: Norton, 1967. Pp. 542-543, 575. 1.
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10. KOGAN,B. A. Health: Man in a Changing Environment. New York Harcourt, Brace & World, 1970. Pp. 29, 541. 11. LICHTMAN,H. C . Cooky’s anemia. Cooky’s Anemia Blood and Research Foundation for Children, New Hyde Park, New York, 1972. 12. REING, A. B. The effects of oculomotor conditioning and perceptual training on impulsivity, mode of imagery, vocational aspiration, and educational achievement. (Doctoral dissertation, New York University) Ann Arbor, Mich.: University Microfilms, 1970. No. 70-15, 976. . Thalassemia major and minor: Health and educational aspects. Unpublished 13. paper presented at Brooklyn College Center for Inter-American Studies, December 11, 1972. 14. . In defense of the MR-LD model: Specific, reversible visual-perceptual dysfunction (HDSA). J S A S Cat. Select. Doc. in Psyckol., 1974, 4, 54. 15. ROSSI,A. 0.Genetics and learning disabilities. Bekav. Nezrvopsyckiat. 1972, 4,2-7. 16. SCHWARTZ, E . Control of globin synthesis in thalassemia. Cooky’s Anemia Newsletr, 1973, 4(7), 1. 17. SMALL,L. Neuropsychodiagnosis in Psychotherapy. New York: Bruner/Mazel, 1973. Pp. 122-136. 18. THRONE, J. M. Genetic factors in mental retardation: A radical behaviorist point of view. Ment. Retavd., 1972, 10(6),32-35. 19. _ _ . Learning disabilities: A radical behaviorist point of view. J. Learn. Disabil., 1973, 6(9), 543-546. 20. VALETT, R.E. The Remediation of Learning Disabilities: A Handbook of Psychoeducational Resource Programs. Belmont, Calif.: Fearon, 1967. ~
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School of Education Brooklyn College C i t y University of N e w York Bedford Avenue and Avenue H Brooklyn, N e w York 11210
