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A capillaroscopic investigation of mongolism Shalman, Denis Clarence 1957

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A CAPIILAROSCOPIC INVESTIGATION OP MONGOLISM by DENIS CLARENCE SHALMAN A THESIS SUBMITTED IN PARTIAL FULFILMENT OP THE REQUIREMENTS FOR THE DEGREE OP MASTER OP /^^<->^X^»-^^ in the Department of Neurological Research We accept this thesis as conforming to the standard required from candidates for the degree of MASTER OP Members of the Department of THE UNIVERSITY OP BRITISH COLUMBIA. October, 1957. ABSTRACT The purpose of the study was to determine the morphological c a p i l l a r y pattern of the mongoloids and compare t h i s with another c o n s t i t u t i o n a l l y retarded c l i n c i a l group - the phenyl-ketonurias - and the degree of deviation from the "normal" pattern. Photomicrographs were taken with c a p i l l a r y microscope and the c a p i l l a r i e s were analysed into the ten basic morphological categories as set out "by Gibson, Bosley and G r i f f i t h s (1956). It was concluded that the mongoloids and the phenylketon-urias d i f f e r e d s i g n i f i c a n t l y in the amount of primitive immature configuration they displayed. The hypothesis of powdermaker (1929) that structure i s related to function, was found tenable through a capillaroscopic examination of s c h o l a s t i c a l l y retarded school children. In presenting t h i s thesis i n p a r t i a l fulfilment of the requirements fo r an advanced degree at the University of B r i t i s h Columbia, I agree that the Library s h a l l make i t f r e e l y available for reference and study. I further agree that permission for extensive copying of t h i s thesis for scholarly purposes may be granted by the Head of my Department or by his representative. It i s understood that copying or publication of t h i s thesis f o r f i n a n c i a l gain s h a l l not be allowed without my written permission. Department of The University of B r i t i s h Columbia, Vancouver 8, Canada. ACKNOWLEDGEMENTS The writer wishes to thank Dr. ¥.0. .Gibson for h i s s k i l l f u l guidance i n conducting the research project, and also Dr. J . ?/ada for his constructive c r i t i c i s m s of the thesis. Special thanks are extended to the nursing s t a f f at Woodlands School who offered a great deal of assistance in taking the photographs of.the patients. Also to Dr. Kerwood who recognized the importance of carrying out basic research and made the services of the school available. iv TABLE OE CONTENTS Chapter Page I INTRODUCTION 1 The problem . . . . 2 Statement of the problem. . . . . . . . . . . 2 Importance of the study 3 De f i n i t i o n of patterns 3 II REVIEW -Off THE LITERATURE ON CAPILLAROSCOPY. . . . 5 Morphology of the normal c a p i l l a r y pattern. . . 7 III EXPERIMENTAL METHOD . .10 Subjects ' 10 Method .10 Apparatus . . 11 Analysis of the results 14 Hypothesis 1 .14 Hypothesis 2 .14 Hypothesis 3 17 IY DISCUSSION 19 V SUMMARY AND CONCLUSIONS Summary 29 Conclusions 29 REFERENCES 30 APPENDIX .36 I Poem by Karl Shapiro 37 v; • LIST OP FIGURES FIGURE PAGE 1 The c a p i l l a r y microscope with attachments. .. . . .12 2 The microfilm reader with reostat used for viewing the c a p i l l a r y f i l m s t r i p s 13 3 Basic morphological pattern of mongolism compared to normal group of Gibson, Bosley and G r i f f i t h s (1956) 15 4 Basic morphological pattern of phenylketon-urias compared to normal group of Gibson, Bosley and G r i f f i t h s (1956) .16 5 Basic morphological pattern of mongolism and phenylketonuria 18 6 Photomicrograph of the c a p i l l a r y nailbed of a mongoloid displaying the t y p i c a l pattern of an underlying plexus with a tortuous morphology 22 7 Photomicrograph of the c a p i l l a r y nailbed of a mongoloid showing an immature plexus with corresponding undeveloped morphology . 23 8 Photomicrograph of the c a p i l l a r y nailbed of a phenylketonuria showing plexus and un-developed morphology . . . . . . . . .24 9 Photomicrograph of the c a p i l l a r y nailbed of a phenylketonuria displaying an immature morphological pattern 25 LIST OF FIGURES (continued) v i 10 Photomicrograph of the c a p i l l a r y nail"bed of a student at Dawson Summer School showing the t y p i c a l immature morphological pattern with plexus 27 11 Comparison of "basic morphological pattern of retarded school children with mongoloids and phenylketonurias 28 CHAPTER I INTRODUCTION Following the monumental work of Itard, the French physi-cian, who developed a system for the education of defective children, there sprang a new interest centred on mental deficiency. Another French p s y c h i a t r i s t , Philippe P i n e l , re-moved the chains from patients and thus i n i t i a t e d the program of s c i e n t i f i c , humane treatment towards individuals a f f l i c t e d with mental abberrations. He divided the f i e l d of mental i l l -ness into mania, melancholia, dementia, and idio c y . In the la s t area, research has moved slowly but the present interest and concern over these mentally defective children, provided a new impetus which has ignited vast f i e l d s of research. Karl Shapiro (56), a contemporary poet, voices the layman's attitude towards mental deficiency in his poem Mongolian Idiot. He paints a pathetic picture of mongolism, which the modern s c i e n t i s t i s endeavouring to correct. (See Appendix I)' Advances i n biochemistry, physiology and more refined methods i n genetics, has shown that mental deficiency may be due to a malfunctioning of parts of the bodyj the i n s u l t i n g agent being either endogenous or exogenous. The removal or correction of the disturbing influence may, and i n some cases, has allowed the c h i l d to progress at a normal rate of develop-ment. Selected c l i n i c a l disorders have provided encouraging results i n this new approach. A dramatic example may be seen i n phenylketonuria; a disorder where an enzyme system i s inadequate to convert phenylalanine to i t s normal metabolite, 2. tyrosine. The main group of mental defectives chosen for this study was the mongoloids. Although t h e i r etiology has "been discussed extensively elsewhere (1, 2, 3, 4, 10, 11, 15, 20, 21, 27, 32, 33, 34, 35, 36, 37, 39, 44, 45, 50, 52, 54, 57, 58, 60} s u f f i c e i t to say here that the disorder i s primarily one of prenatal dysfunction. That the maturational processes have been arrested at this stage i s indicated c l e a r l y through the physical malformation and mental development they display. As to the causation of the anomaly, considerable doubt exists whether i t i s of constitutional or environmental o r i g i n . Leader (1932) noted that when the general development of a person i s arrested a pathological development of the vessels r e s u l t s . Instead of the c a p i l l a r i e s developing i n a normal way they tend to r e t a i n t h e i r primitive shapes and to assume bizzare forms. This forms the basis for the selection of mongolism to study. I f Leader's statement i s correct, the mongoloid group should display primi-t i v e and bizzare morphology. I. THE PROBLEM Statement of the problem. The purpose of the study was to test the hypothesis (1) that the mental defectives as a group d i f f e r e d from the normals as a group i n th e i r c a p i l l a r y morph-ology; (2) that differences exist within the defectives in r e l a t i o n to the i r c a p i l l a r y structure and, (3) that the c a p i l -l a r y morphology may be used as a diagnostic tool for the border-3. l i n e cases. Importance of the study. The primary aim of the present study was to consider the group of mongoloids i n order to establish another physical correlate which may be instrumental i n a s s i s t i n g the c l i n i c i a n in his diagnosis. The a b i l i t y to diagnose the borderline cases is of importance. It is desired that the material co l l e c t e d w i l l increase the data that is ac-cumulating concerning this disorder. It is hoped that in the not too distant future a l l the relevant facts may be drawn to-gether and a solution found to the problem. A quotation from Harley Williams' Don Quixote of the Microscope (63), is apropos at this point, "Ramon y Cajal had found the brain an impenetrable forest, but had turned i t into a d e l i g h t f u l park". D e f i n i t i o n of patterns. The c l a s s i f i c a t i o n the present study i s compared with found that the "normal" d i s t r i b u t i o n of c a p i l l a r i e s was a series of discrete categories. These have been defined by the authors (Gibson, Bosley & G r i f f i t h s 1956) and are reproduced below. Pattern A - This i s represented by those c a p i l l a r i e s that have no warpings or indentations of either the afferent or the efferent limb, no twisting of either limb about i t s e l f or i t s opposite component, and no excessive distention of either the afferent or the efferent limb, or the t i p of the loop. pattern B - This is represented by those c a p i l l a r i e s that show a certain amount of warping or indentation of one or both limbs. No twisting of one limb around the other or i t s e l f i s evident. 4. Pattern C - This i s represented by those c a p i l l a r i e s that show a single crossing in their passage to and from the sub-papillary plexus of deeper vessels. They may also show some warping or indentation, but the single twist i s the prime consideration i n c l a s s i f i c a t ion. Pattern D - This is represented by those c a p i l l a r y loops whose limbs show more than one twist about each other. They may, in addition, show some warpings, indentations or excessive distention. Pattern E - This is represented by those vessels in which the main feature i s an obscure twisting of one limb upon i t s e l f . Thismay be seen i n either or both of the arms. Sometimes a certain amount of twisting s i m i l a r to the type seen i n Pattern C is evident.also. Pattern E - This i s represented by those c a p i l l a r i e s with an anomalous morphology at the t i p of the loop. The general picture resembles the head of a femur i n outline. This anomalous morphology usually ex-tends into the efferent limb. Pattern G - This i s represented by those c a p i l l a r i e s that do not c l e a r l y spring from the deeper sub-pa p i l l a r y plexus. They appear to represent an im-mature horizontal network of vessels. Pattern H - This i s represented by c a p i l l a r i e s that resemble flowery shapes with many diverse twistings. They are usually i n d i s t i n c t i n outline, presenting a t y p i c a l blurred appearance. Pattern I - This pattern i s represented by an extreme modification of Pattern B. The loops present a meandering course as they pass from the afferent to the efferent limb. A horizontal plexus is sometimes p l a i n l y v i s i b l e . Pattern J - This pattern i s represented by very short, thin vessels. 5. CHAPTER II REVIEW OP THE LITERATURE OH CAPILLAROSCOPY As- early as 1663, Johan Christophorus Kolbasis explored c a p i l l a r y microscopy as a new t o o l which could he used as a possible diagnostic procedure. His success i n this venture i s unknown, hut he l a i d the cornerstone which was to have great importance. In 1866 Strieker, following his studies on the excised membrane of the frog, described the structure and function of c a p i l l a r i e s . Rouget in 1873 reported an independent action of the c a p i l l a r i e s which was brought about by means of spindle shaped, smooth muscle c e l l s which had the power to contract and expand. In 1893, Spalteholz reported his careful anatomic studies of the vascular system of the human skin which he found to be supplied from the underlying tissues through a large number of small ar t e r i e s forming an anastomosing i r r e g u l a r plexus. Heimberger made an observation of considerable interest. He found that around each c a p i l l a r y loop in the p a p i l l a e there is a continuous lymphatic sac, traversed by a number of threads or septums, which hold the c a p i l l a r y loop i n place. It was not u n t i l 1912 that Lombard carried out the pioneer work in the f i e l d . He found that i f a drop of glycer-ine was placed on the back of the hand previously cleaned with alcohol and a bright l i g h t brought to bear on the area, a be a u t i f u l picture of the p a p i l l a e and the s u p e r f i c i a l vessels 6. was o b t a i n e d . By c h o i c e the a r e a examined became the f i n g e r n a i l bed, f o r here the c a p i l l a r i e s a r e a r r a n g e d i n a h o r i z o n t a l p l a n e and g e n e r a l l y o n l y one l a y e r t h i c k . Many e x p e r i m e n t e r s have worked i n t h i s a r e a t o show c a p i l l a r y morphology by p h o t o m i c r o g r a p h i c means ('6, 12, 14, 19, 24, 25, 26, 28, 29, 30, 62, 64),. S i n c e the use o f c a p i l l a r y m i c r o s c o p y began, a l l the e a r l y s t u d i e s by b o t h American and German a u t h o r s showed t h a t t h e r e i s a d e f i n i t e and d i r e c t r e l a t i o n s h i p between the development of the c a p i l l a r y s t r u c t u r e and mental development. Examina-t i o n s o f German a u t h o r s ( r e p o r t e d by Hauptman and Myerson, 1948) shovir t h a t the c a p i l l a r i e s o f s c h o o l c h i l d r e n have a normal p a t t e r n i n 67% o f the c a s e s , whereas c h i l d r e n o b s e r v e d from an ungraded s c h o o l had normal c a p i l l a r i e s i n o n l y 2 9 % o f the c a s e s . The a u t h o r s suggest t h a t c a p i l l a r o s c o p i c e x a m i n a t i o n may h e l p i n the d i f f e r e n t i a l d i a g n o s i s of m e n t a l a b n o r m a l i t i e s a r i s i n g e i t h e r t h r o u g h c o n s t i t u t i o n a l o r e n v i r o n m e n t a l e t i o l o g y . Powdermaker (1929) made one o f the e a r l i e s t a t t e m p t s to r e l a t e s t r u c t u r e to f u n c t i o n . She s t u d i e d the c a p i l l a r y form i n r e l a t i o n to c o n s t i t u t i o n a l and secondary h y p o p h r e n i a s . The importance of t h i s s t u d y l i e s i n t h e p o s s i b i l i t y o f a s s i s t i n g i n the e a r l y d e t e c t i o n of a c o n s t i t u t i o n a l d e f e c t and i n d i f f e r e n t i a t i n g i t from an e n v i r o n m e n t a l d e f e c t . Of p a r t i c u l a r note i n Powdermaker's s t u d y was h e r o b s e r v a t i o n t h a t i n c o n s t i -t u t i o n a l i d i o t s and low grade i m b e c i l e s , i n whom t h e r e was a h i g h p r o p o r t i o n o f p h y s i c a l a b n o r m a l i t i e s ( 8 8 % ) , t h e r e was a 7. marked tendency f o r a retarded or abnormal development of the c a p i l l a r i e s ; t h i s was much less marked i n the few cases which showed in approximately normal physical development. In the moron and high grade imbecile group there were r e l a t i v e l y few primitive forms. In the cases i n which these forms were found, there was a s i g n i f i c a n t l y greater proportion of emotional, en-docrine and neurologic disorders. No primitive forms were found i n cases of secondary o r i g i n and in stable children with normal, physical and mental development. N Jaensch, with Hoepfner and Wittneben, in the same year, published a book from a study of 3000 cases. They asserted that when the general development is markedly defective the c a p i l l a r i e s may never lose the main ch a r a c t e r i s t i c s of the primitive network form, and both limbs of the c a p i l l a r y loops w i l l have a wide c a l i b r e . Morphology of the normal c a p i l l a r y pattern. The problem of determining a normal form of c a p i l l a r i e s i s as old as the f i r s t observation. There i s considerable discrepancy through-out the l i t e r a t u r e concerning th i s matter, but most of the data can be assigned to one of two c l a s s i f i c a t i o n s . The f i r s t cat-egory designates the normal pattern, for the n a i l bed as con-s i s t i n g of straight forward hairpin loops. In this group, any deviations from the "normal" form constitutes r e l a t i v e "abnormalities" (8, 9, 38, 41 , 42, 46, 51, 53, 59) . The other category i s designated by more than one form as consisting of 8. the "normal" p a t t e r n . The authors i n t h i s group have concluded that i t i s d i f f i c u l t to e s t a b l i s h a "normal" pattern'due to the wide v a r i a t i o n s found i n the c o n t r o l groups (7, 12, 13, 16, 17, 23, 24, 25, 26, 28, 29, 30, 40, 41, 42, 43, 55, 64). Gibson, B o s l e y and G r i f f i t h s (1956), r e c o g n i z i n g the impor-tance of the problem, set out to determine a normal morpholog-i c a l p a t t e r n . They s e l e c t e d f o u r hundred "normal" i n d i v i d u a l s and c a p i l l a r o s c o p i c exminations were recorded. An a n a l y s i s of the r e s u l t s r e v e a l e d not one but s e v e r a l forms were i n d i c a t i v e of a normal p a t t e r n . The c h i e f l i m i t a t i o n of adopting t h i s pro-cedure l i e s i n s t a t i s t i c a l a n a l y s i s . The data c o l l e c t e d i n d i -cates c a p i l l a r y morphology on a t o t a l b a s i s only and does not p r o v i d e the morphology f o r a s i n g l e n a i l bed. In the present study, a comparison between c l i n i c a l groups was d e s i r e d , t h e r e f o r e , t h i s c l a s s i f i c a t i o n was used. Powdermaker (1929) has c l a s s i f i e d the v e s s e l s based on degrees of m a t u r i t y , w i t h the f i n a l stage ( s t r a i g h t h a i r p i n loop) r e p r e s e n t i n g the "normal" p a t t e r n . S i n c e her c l a s s i f i -c a t i o n i s based on the maturation of the v e s s e l s , t h i s i s an opportune time to d i s c u s s the development of the c a p i l l a r i e s . The developmental sequence begins w i t h an arabesque of v e s s e l s w i t h no p a r t i c u l a r order. It i s an anastomosing a r t e r -i o l a r plexus a r i s i n g i n the deepest l a y e r of the dermis p r e s e n t at b i r t h . There are no t r u e c a p i l l a r y loops and the upper end of the corium r e p r e s e n t s a f l a t l i n e . At one month the sub-p a p i l l a r y plexus begins to disappear and up to f o u r or f i v e 9. months saddle shaped forms sprout from the primitive plexus„ They grow gradually in the d i r e c t i o n of the corium; but not before the f i f t h month does one f i n d a few h a i r p i n - l i k e c a p i l l a r y loops with d i f f e r e n t i a t i o n in c a l i b r e . At the same time, the horizontal network of fine blood vessels increases in size and complexity. The corium becomes wavy and assumes the c h a r a c t e r i s t i c scalloped appearance with each c a p i l l a r y growing into one p a p i l l a e . From the f i f t h or s i x t h month to one year the c a p i l l a r i e s develop to the mature picture with a disappearance of the wide loops and subpapillary plexus. At this p a r t i c u l a r stage the c a p i l l a r y morphology i s con-sidered complete. The pattern i s persistent, changing only with old age, and any t o r t u o s i t y in children i s considered abnormal, (Leader, 1932). 10. CHAPTER III EXPERIMENTAL METHOD The problem as stated necessitates a c a p i l l a r y assessment on two groups of mental defectives. The c a p i l l a r y analysis was determined after the c l a s s i f i c a t i o n of Gibson, Bosley and G r i f f i t h s (1956). A further analysis was made on whether a subpapillary plexus was present, for Hauptman and Myerson (1948) stress that i n normal individuals, no subpapillary plexus i s seen. Sub.j ects. The experimental groups f o r th i s study consisted of one hundred and twenty-six mongoloids and sixteen phenyl-ketonurias taken from Woodlands School for Mentally Retarded Children, New Westminster, B.C. Mongolism was chosen because i t i s one of the largest groups of mental defectives. The group phenylketonuria was selected because of the interest i t has aroused since P o l l i n g f i r s t noted the disorder i n 1934 as one of the r e l a t i v e l y few "inborn error" of metabolism. Although the t o t a l population of the two groups were used, the thickness of the epithelium prevented obtaining photomicro-graphs in every case. This, however, is a technical, d i f f i c u l t y that was unavoidable. Method. The equipment used folded very conveniently into two carrying cases. Because of the r e l a t i v e ease to move the 11. equipment, the patients were examined on the wards at the school. The fourth finger, l e f t hand, was selected for obser-vation. It was prepared with zylene, dried, and cedar o i l then spread over the n a i l bed. Apparatus. A modified Zeiss Opton standard stereoscopic binocular dissection microscope was used. An extra arm was f i t t e d for the finger clamp assembly. A 35 mm. Canon camera with a Mecablitz electronic f l a s h was attached. The electronic f l a s h (l/500 second) proved an invaluable addition since i t removed the d i f f i c u l t y of aperture settings and moving f i n g e r s . An a u x i l i a r y L e i t z Wetzlar skin and c a p i l l a r y microscope lamp was used for focusing. (See Figure l ) . The prints were made on Kodak, Linagraph Ortho safety f i l m ; a very fast orthochromatic f i l m of moderate contrast. The f i l m was developed i n Kodak D 19 and enlargements made on Kodabromide paper. It was found that viewing the f i l m s t r i p s i n a microfilm reader was most s a t i s f a c t o r y . The pictures could be enlarged, focused and the i n t e n s i t y altered instantan-eously. A Tri-purpose projector, manufactured by the Society for Visual Education, Inc., with a Powerstat, manufactured by the Superior E l e c t r i c Do. were used i n projecting and il l u m i n -ating the f i l m . (See Figure 2). 12. Figure I t The c a p i l l a r y microscope with attachments. 13. Figure 2: The microfilm reader with reostat used for viewing the c a p i l l a r y f i l m s t r i p s . 14. ANALYSIS OF RESULTS' A t o t a l of one hundred and twenty-six mongoloids were used for the determination of c a p i l l a r y morphology. These results were contrasted against sixteen phenylketonurias, and both mentally defective groups were compared with the normal group of Gibson, Bosley and G r i f f i t h s (1956). The above mentioned investigators formulated a basic c a p i l l a r y pattern that appeared to be common to the majority of "normal" healthy individuals. This pattern consisted of the following forms designated A to J respectively. n A i fl f? ff (O ff J * * * A B G D E F G H I J 1) Hypothesis: The group of mongoloids display a mor-phological pattern no different from that found in the normal population. (See Figure 3). Computed chi square of 109.1 was s i g n i f i c a n t at the 0,01 l e v e l of confidence. Therefore the hypothesis i s rejected. 2) Hypothesis: The group of phenylketonurics display a morphology sim i l a r to the normal population (See Figure 4). Computed chi square of 296.20 was s i g n i f i c a n t at the 0,01 l e v e l of confidence. Therefore the hypothesis i s rejected. 15. B a s i c m o r p h o l o g i c a l p a t t e r n of mongoloids compared to normal group of Oioson, Boaley and G r i f f i t h s (1956). « •—. Phenylketonurias \ <ft Normals « s c o e F G M x x F i g u r e 4 Basic morphological pattern of phenylketonuria compared to normal group of Gibson, Bosley and G r i f f i t h s (1956). 17. 3) Hypothesis: There is no difference i n morphological pattern within the mentally defective groups of mongolism and phenylketonuria (See Figure 5). Computed chi square value of 23.41 was s i g n i f i c a n t at the 0.01 l e v e l of confidence. Therefore the hypothesis i s rejected. P B C D E F O H T J -Pigure 5 B a s i c m o r p h o l o g i c a l p a t t e r n of mongolism and p h e n y l k e t o n u r i a . 191 CHATTER TV DISCUSSION The primary aim of the present study was to determine the morphological pattern of the c a p i l l a r y structure of the mongolian i d i o t s (See Figure 6 ) . The re s u l t i n g pattern i s pre-sented i n Figure 3, along with a comparison to a "normal" group. It was observed that the mongoloids d i f f e r e d s i g n i f i -cantly from the "normal" population. The number of open loops (A and 33) were reduced i n the mongoloids. As has been previously stated, the type A struc-ture is considered by many to be representative of the normal pattern. In type G the difference i s enormous since this pattern i s seldom found. Pattern G has been described as those c a p i l l a r i e s which represent an immature horizontal network of vessels; thus with the increased appearance of Patterns G, H and I, the mongoloid group displays a rather immature and un-developed pattern (See Figure 7). As. observed previously, mongolism i s the result of f o e t a l arrest, thus there w i l l be an increased number of primitive forms since the delay i n c a p i l l a r y development should be correlated with the general structural or physical retardation. It has been stated by many authors (29, 30, 38, 53,) that the presence of a subpapillary plexus Is indicative of an im- . mature configuration. Gibson, Bosley and G r i f f i t h s (1956) did not report the presence or absence of a plexus in their normal 20. groups studied, therefore, reference to other l i t e r a t u r e on the subject has "been resorted to, i n order to support the discussion. It i s interesting to note that within the mongoloids the plexus occurred only in the early age l e v e l s . There were no cases past the age thirteen i n the females (which comprised 63.4% of the cases) and age eighteen (which comprised 68.4% of the cases) i n the males. This implies that in the remaining cases the configuration has progressed beyond the plexus to a more mature stage. If this implication is correct, then a very i n t e r e s t i n g inference can be made. Hauptman and Myerson (.1948) observed that i n mental retard-ation which has as i t ' s o r i g i n an endogenous agent, the c a p i l l a r y structure w i l l be immature and w i l l never undergo development at l a t e r periods. When this i s interpreted i n l i g h t of the above observation, i t becomes of paramount impor-tance to the age-long query whether the syndrome mongolism has i t s etiology i n c o n s t i t u t i o n a l or environmental factors. If the p r e c i p i t a t i n g cause of mongolism i s c o n s t i t u t i o n a l , then the c a p i l l a r y pattern should be primitive and never mature. However, i f the agent operating i s exogenous, that i s , operat-ing through the maternal environment, then the pattern may a l t e r with age, l o s i n g some of i t s undeveloped structure. Since t h i s appears to be the case, i t would seem that the etiology of mongolism exists i n environmental and not solely c o n s t i t u t i o n a l factors. Mongolism was compared with another group of mental 21. defectives the phenylketonurias. It was desirable to know whether mongolism d i f f e r e d from other mental d e f i c i e n c i e s , since this information i s necessary before the morphological pattern evolved could be used as an a i d d i a g n o s t i c a l l y . It was noted that the mongoloids d i f f e r e d s i g n i f i c a n t l y from the phenylketonurics. Both groups displayed a high frequency of plexus (See Figure 8 ) . In the mongoloid group there was 2Z.1% while i n the phenylketonuria group 56.2% occurred. Powdermaker (1929) noted that a correlation exists between the poorly developed c a p i l l a r i e s and a general defect i n the development of what has been c a l l e d the psychophysical person-a l i t y . Leader (1932) noted that when the general development of a person i s arrested a pathologic development of vessels r e s u l t s . Instead of the c a p i l l a r i e s developing they tend to reta i n t h e i r primitive >shapes and assume bizzare forms. Following the observation that the mongoloids and phenylketon-urias retained many primitive forms (See Figure 9), especially the plexus, i t was thought advisable to examine a group of children who were i n t e l l e c t u a l l y retarded so that a comparison with a less severely retarded group could be viewed i n respect to a cor r e l a t i o n existing between the resul t i n g behavior and the physical status. Normally, i t is considered by psychologists that i n t e l l i -gence i s primarily inherited and consequently of constitutional o r i g i n (Munn 1951). The c o n s t i t u t i o n a l l y retarded individuals whose malfunctioning was severe (mongoloids and phenylketonurics) Figure 6* Photomicrograph of the c a p i l l a r y nailbed of a mongoloid displaying the t y p i c a l pattern of an underlying plexus with a tortuous morphology. 23. Figure 7* Photomicrograph of the c a p i l l a r y nailToed of a mongoloid showing an immature plexus with corresponding undeveloped morphology. 2 4 . Figure 8: Photomicrograph of the c a p i l l a r y nailhed of a phenylketonuria showing plexus and undeveloped morphology. 25 Figure 9: Photomicrograph of the c a p i l l a r y nailbed of a phenylketonuria displaying an immature morphological pattern. 26. displayed a large proportion of undeveloped morphology (See Figure 10) which appeared to correspond with the less severely retarded individuals (Dawson Summer School students).(l).. Of the t h i r t y - s i x students a t o t a l of 22.2% possessed a plexus and retained much of the undeveloped structure as evidenced hy the increased occurrence of pattern G and I (See Figure 11). Thus i t appears that Powdermaker's and Leader's observations of a co r r e l a t i o n existing between structure and function i s tenable. Therefore, i t seems that the undeveloped pattern of the mental defectives bears a re l a t i o n s h i p to th e i r psycho-pathology and the presence of a plexus i s i n d i c a t i v e of re-tarded development. This conclusion i s upheld by the s i m i l a r i t y with the less retarded students. 1. Operated by the Y.M.C.A. for children i n grade school who have to repeat t h e i r year or have considerable d i f f i c u l t y with t h e i r school program. 27. Figure 10j Photomicrograph of the c a p i l l a r y nailhed of a student at Dawson Summer School showing the t y p i c a l immature morphological pattern with plexus. 4o F i g u r e I I Comparison of b a s i c m o r p h o l o g i c a l p a t t e r n of r e t a r d e d s c h o o l c h i l d r e n w i t h mongoloids and p h e n y l k e t o n u r i a . CHAPTER V SUMMARY A capillaroscopic examination of the c l i n i c a l groups, mongolism and phenylketonuria, was carried out at the Woodland' School f o r Retarded Children. The c a p i l l a r i e s were analysed a f t e r the ten basic morpho-l o g i c a l patterns set out by Gibson, Bosley and G r i f f i t h s (1956) A comparison was made within co n s t i t u t i o n a l retardation of severe (mongolism and phenylketonuria) and less severe (Dawson Summer School students) order. CONCLUSIONS 1. The mongoloids and phenylketonurics, as a group, di f f e r e d s i g n i f i c a n t l y from normals, as a group, i n t h e i r cap-i l l a r y morphology. 2. There was a s i g n i f i c a n t difference found between the morphological pattern of mongoloids and the phenylketonurics. 3. C a p i l l a r y morphology may be used on a group basis to assist i n diagnosis of borderline cases of mongolism. 4. There was a s i m i l a r i t y betv/een the morphological patterns of severe and less severe c o n s t i t u t i o n a l l y retarded ind i v i d u a l s . RE3TEREJIC1S 31. REFERENCES Armstrong, H., The etiology of mongolism, B.M. J . , 1:1106, 1928. Beldleman, B., Mongolism: a- selective review, Am. J . Ment  Def., 50:35, 1945. Benda, C , On the'tiology and the prevention of mongolism, Am. J. psychiat., 99:822, 1943. Bleyer, A., Indications that mongoloid imbecility i s a gamitic mutation of depressive type, Am. J. P i s . Child, 47:342, 1934. Brown, G.E. and G i f f i n , H.Z., Studies of c a p i l l a r i e s and blood volume in polycythemia vera, Am. J . Aust., 1:496, 1927. Brown, G.E. and Roth, G.M., Biomicroscopy of the surface c a p i l l a r i e s i n normal and pathological subjects, Med. J . Au s t r a l i a , A p r i l 2, 1927. Callandar, C*L., Photomicrographic studies of morphology of surface c a p i l l a r i e s in health and disease, J.A.M.A., 84:(5) 352, Jan.31, 1925. Carrier, E.B., Studies on the physiology of c a p i l l a r i e s : The reaction of the human skin c a p i l l a r i e s to drugs and other s t i m u l i , Am. J . Physiol. 61:528, 1922. Castel'lanos, M. and Gibson, W.C., Abnormalities of the c a p i l l a r y nailbed, Arch. Neurol. Psychiat., 63:140, 1950. Clark, R..M., The mongol: a new explanation, J . Ment. S c i . , 74:265, 1928.- " Clark, R.M., The mongol: a new exaplanation, J . Ment. S c i . , 79:328, 1933. Cobb, S., Cohen, M.E. and Badal, D.W., C a p i l l a r i e s of the n a i l f o l d in patients with neurocirculatory asthenia (ef f o r t syndrome, anxiety neurosis), Arch. Neurol.  Psychiat. 56:643, 1946. Crawford, J.M. and Rosenberger, M., Studies on human cap i l l a r i e s : : an apparatus for cinematographic observation of human c a p i l l a r i e s , J . C l i n . Invest., 2:343, 1925. 32. 14. Crooks, H.E., Photography of epidermal ridges and super-f i c i a l "blood c a p i l l a r i e s of the finger, Med. B i o l .  I l l u s t . , 3*4 198, 1953. 15. Crowley, R.H., McKeown, T. and Record, R.G., Influence of the Pre-Hatal Environment on Post-Hatal Growth, B i r t . J .  Prev. Soc. Med., 8, 66-69, 1954. 16. Davis, E., Ca p i l l a r y microscopy with special reference to c a p i l l a r y petechiae, Am. J . Med. S c i . , 212:192-196, 1946. 17. Deutsch, E., C a p i l l a r y studies in Raynaud's disease, J . Lab. C l i n . Med. -26:1729, 1941, 18. Dobler, T,, Comparative capillaroscopic studies in the treatment of peripheral c i r c u l a t o r y disorders, Ther.  Umschan Berne, 10:7 89, 1953. 19. Duryee, W.A. and Wright, I.S., Studies in human c a p i l l a r i e s j present day techniques for the study of human c a p i l l a r i e s , Am. J . Med. S c i . , 185:664, 1933. 20. Engler, M., The causation of mongolism and i t s prognosis, Proc. Roy. Soc. Med., 38:211, 1944. 21. Engler, M., The causation of mongolism and i t s prognosis, Amer. J . Ment. Defec., 50:27, 1945. 22. Eiehrer, A., Les maladies des c a p i l l a i r e s , Concours Medical 77(7), p.621-4, Feb.1955. 23. Gibson, W.C., Bosley, P.G.H.J., and G r i f f i t h s , R.S., Photomicrographic studies on the nailbed c a p i l l a r y networks i n human control subjects, J . Nerv. Ment.  P i s . 125(3). 219-231, March, 1956. 24. G i l j e , 0., C a p i l l a r y microscopy in "the d i f f e r e n t i a l diagnosis of skin diseases, Acta Ormato Venereologica, 33:303, 1953. 25. G i l j e , 0., Kierland, R. and Baldes, E., C a p i l l a r y micros-copy in diagnosis of derrnatological diseases, J. of Invest. Dermatology, 22:3, March, 1954. 26. G i l j e , 0., O'Leary, P.A. and Baldes, E.J., C a p i l l a r y microscopic examination in skin diseases, Am. Archives  of Dermat. and Eyphilology, 68:136, 1953. 27. Goldstein, H., A study of mongolism and non mongoloid mental retardation in children, Arch. P e d i a t r i c s . 71:11 1954. 33. 28. G r i f f i t h , J.Q,., The frequent occurrence of abnormal cutan-eous c a p i l l a r i e s i n c o n s t i t u t i o n a l neurasthenic states, Am. J . Med. S c i . , 183:180, 1932. 29. Hauptman, A., C a p i l l a r i e s in the n a i l f o l d i n patients with neurosis, epilepsy and migraine, Arch. Neurol. Psychiat. 56:631, 1946. 30. Hauptman, M. and Myerson, A., Studies on the finger c a p i l l a r i e s in schizophrenics and manic depressive psychoses, J . Nerv. Mental P i s . , 108:91, 1948. 31. Heimberger, H. Contractile Function u. anat. Bar der mehschl. Capillaren. Zeitsch f. Z e l l f o r s c h u. mikv. Anar. 4, 713, 1937. 32. H i l l , B., Mongolism and i t s pathology, Quart. J. Med., 2:49, 1908-1909. 33. Ingalls, T.H., The pathogenesis of mongolism, Am. J. P i s . Child, 73:279, 1947. 34. Ingalls, T.H., Mongolism, S c i e n t i f i c American, 186:60-66 1952. 35. Jenkins, R.L., Etiology of mongolism, Am. J. P i s . Child, 45:506, 1933. 36. J e r v i s , G.A., Symposium on mongolism, Quat. Review Pediat., May, Aug. Nov. 126, 1953. 37. Lande-Champain, The etiology of mongolism, J . Child Psychiat 3:53, 1954. 38. Leader, S., C a p i l l a r y microscopy in children, Am. J . P i s . Child, 44?403, 1932. 39. Lipnitzky, S. and Boshes, B., A possible paternal factor in the etiology of mongolism, J. Hered. 33:155, 1942. 40. Lombard, W.P., The blood pressure i n the a r t e r i o l e s , c a p i l l a r i e s and small veins of the human skin, Am. J .  Physiol., vol.29, 335-362, 1912. : 41. Lovett Poust, J.W., The c a p i l l a r y system in patients with psy c h i a t r i c disorders: diminished c a p i l l a r y resistance as shown by the Gothlin p o s i t i v e pressure test, J . C l i n . E x p t l . Psychopathology. 16, 4:272, Dec., 1955. 34. 42. Lovett Doust, J'.W., The c a p i l l a r y system i n patients with psychiatric disorders: the ontogenetic structural determination of the n a i l f o l d c a p i l l a r i e s as observed "by photomicroscopy, J . Nerv. Ment. P i s . , - Vol.121, 6, 516, June-, 1955. 43. Lovett Poust, J.W., C a p i l l a r y system i n patients with psychiatric disorders: increased c a p i l l a r y blood pressures, A.M.A. Archives of Neur. and Psychiat., 74:137, August, 1955. 44. Mackintosh, J . , The etiology of mongolism, B.M,J., 2:129, 1928. 45. Macklin, M., Mongolian Idiocy: the manner of i t s inheritance, Amer. J. Med. S c i . , 179:315, 1929. 46. Mayer, K.M., Observations on the c a p i l l a r i e s of the normal infant, Am. J. P i s . Child., 22, 381, 1921. 47. M i l l e r , L.A., Inspection of c a p i l l a r i e s i n health and disease, Ohio State Med. J . 20:759, 1924. 48. Munn, N.L., Psychology: the fundamentals of human adjust-ment , Second edition, Boston: Houghton M i f f l i n Company, -$.101, 1951. . 49. Myerson, A., Finger c a p i l l a r i e s in schizophrenia and manic depressive psychoses, J . Nerv. Mental P i s . 108, 91, August, 1948. 50. Oster, J . , The causes of mongolism, Panish Medical B u l l e t i n 3:158, 1956. : 51. Paskin, H.A. and Brown, M., Constitutional differences between deteriorated and non-deteriorated patients with epilepsy, Arch. Neurol. Psychiat., vol.49, pp.49-55, Jan., 1943. 52. Penrose, L., Observations on the etiology of mongolism Lancet, 267:505, 1954. 53. Powdermaker, F., C a p i l l a r y forms in r e l a t i o n to certain problems in development, Arch. Neur. and Psychiat, 22:1207-1216, 1929. 54. Rosanoff, A.J. and Handy, M.L., Etiology of mongolism, Am. J . P i s . C h i l d , 48:764, 1943. 55. Roth, G.M., N a i l f o l d c a p i l l a r i e s i n man, Med. J. A u s t r a l i a . A p r i l , 1927. ~~ 35. 56. Shapiro, K., "Mongolian I d i o t " / F i f t e e n Modern American Poets, George P. E l l i o t t , New York: Rinehart and Company Inc., 1956. 57. Shuttleworth, G.E., Mongolian idiocy, B.M.J., 2:661, 1909. 58. Smith, A., and Record, RVG., Maternal age and b i r t h rank i n the aetiology of mongolism, B r i t . J . Prev. Soc. Med. 90:51, 1955. 59. Sokoloff, B., and Eddy, W.H., C a p i l l a r y f r a g i l i t y and stress, Mono. #3;, E.S.C., 1952. 60. Southwick, W.E., Time and stage i n development at which factors operate to produce mongolism, Am. J . P i s .  Child, 57:68, 1939. 61. SpaltehoTz, W., Die Verteilung der Blutgefasse, Haut. Arch, f. Anat (u. Physiol.) 1 (2,4), 1893. 62. Wertheimer, N., and Wertheimer, M., C a p i l l a r y structure: i t s r e l a t i o n to ps y c h i a t r i c diagnosis and morphology, J. -Nerv. Ment. P i s , vol.122, 1, p.14, July 1955. 63. Williams, H., Pon Quixote of the Microscope, London: Jonathan Cape, p.154-5, 1954. 64. Wright, I.S., C l i n i c a l value of human c a p i l l a r y studies in fever, mental deficiency, nephritis vascular diseases, clubbed f i n g e r , a r t h r i t i s , tobacco smoking and argyria, J . C l i n . Invest. 11:835, July 1932. 65. Wright, I.S. and Duryee, A.W., Human C a p i l l a r i e s i n health and disease, Arch. Int. Med., 52:545-575, 1933. A P P E N D I X APPENDIX I Mongolian Idiot A dog that spoke, a monster horn of sheep We mercilessly k i l l , and k i l l the thought, Yet house the parrot and l e t the centaur go, These being to their nature and those not. We laugh at apes, that never quite succeed At eating soup or wearing hats. Adam had named so many but not t h i s , This that would name a curse when i t had come, Unfinished man, or witch, or myth, or s i n , Not ever father and never quite a son. Ape had outstripped him, dog and darling lamb And a l l the kindergarten beasts. Enter the bare room of his mind and court His store of words with l e t t e r s large See how he handles clumsily those blocks With swans and sums; his colored picture books. At t h i r t y - f i v e he squeals to see the b a l l Bounce i n the a i r and r o l l away. P i t y and fear we give t h i s innocent Who maimed'his mother's beautiful i n s t i n c t ; But she would say, "My body had a'dog; I bore" the ape and nursed the crying sheep. He i s my kindness and my splendid g i f t Come from a l l l i f e and for a l l l i f e . " 

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