Toxoplasmosis in a nine-year-old girl

Case Reports TOXOPLASMOSIS IN A NINE-YEAR-0LD GIRL PAUL FREEMAN, ~V~.D.,AND HELEN B. PRYOR, ]V[.I). REDWOOD CITY, CALIF. A

CASE of toxoplasmosis in a 9-year-old girl is presented which demonstrates three of the clinical tetrad of signs and symptoms plus a congenital cyst of the left upper lobe of the lung. There is no proved relationship between the lung cyst and the cerebral toxoplasmosis, but a question is raised concerning their probable interrelationship. Following Frenkel's classification, the case presented was of a patient in the subacute stage, surviving with symptoms of chorioretinitis, cerebral calcifications, and other signs of cerebral damage. The child's mother can be classed as having chronic, latent, asymptomatic toxoplasmosis. CASE

HISTORY

N. H., a white female child, was delivered by a breech extraction on Jan. 23, 1940. The birth weight was 6~ pounds. The prenatal course had been uneventful until about three weeks before term, at which time the mother had had an acute febrile illness with low abdominal pain and a profuse, foul, purulent vaginal discharge. Smear of the vaginal discharge revealed no predominant pathogenic organisms, and the mother recovered before delivery. The placenta grossly contained a mass of cystlike nodules, but no histologic study was made of this material. The infant was apparently normal at birth, and the routine well-baby cheeks during the first year of life revealed no abnormalities save a hemoglobin count of 60 per cent at 6 months of age, which responded to iron therapy. I-Iowever, she was slow in development. She sat alone at one year of age, walked alone at 15 months, and began to talk at about age 2 years. On Nov. I, 1944, at the age of 4 years, 8 months, the child became acutely ill with an upper respiratory infection for which she was hospitalized. She was found to have an extensive pyothorax of the left chest (Fig. 1). Thoracen tesis was performed with removal of 400 e.e. purulent material. Culture and guinea pig inoculations done on the material revealed no tubercle bacilli, and iViantoux tests using first and second strength PPD were negative. A thoracic surgeon saw the child in consultation and fMt that she had either an interlobar pyopneumothorax or a large lung cyst filled with pus. On Dec. 6, 1944, a thoracotomy with rib resection was performed in order to drain the chest. Purulent material continued to drain for about six weeks, after which healing occurred. The child's course for the next three and one-half years was characterized by frequent febrile illnesses associated with cough and chest pain. Periodic roentgenographic studies of the lung fields consistently revealed an area of rarefaction with a fluid level in the upper lobe of the left lung (Fig. 2): Because of developing strabismus and progressive loss of vision in the right eye, the child was seen by an ophthalmologist in April, 1945. The mother was told that loss of the vision was the result of sear tissue forming in the eye and that nothing could be done about it. But the child continued to go to school. 365

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Fig. 1.--Pyothorax

Fig- 2.--Film

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left chest displacing heart.

A u g . 25, 1948, s h o w i n g fluid l e v e l in u p p e r l o b e l e f t k u ] g .

FREEze, AN AND P R Y O R :

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An appraisal of N. at this time by her teacher included the following' description: " S h e walked with an ambling, loose-jointed gait. H e r shoulders seemed to be held high and her a r m motion was jerky. She entered into ahnost as m u c h p l a y activity as the other children a l t h o u g h she w a l k e d more than. she ran, and d i d n ' t r u n as f a s t as the others. " A l t h o u g h she seemed to do all or most of her seeing with her left eye, she held her head straight. She did not seem to s t r a i n to see w o r k on the b o a r d or on her desk. " S h e w r o t e v e r y lightly and her letters were p o o r l y formed. One m i g h t think t h a t lack of s t r e n g t h and coordination in her arms were responsible for such light pressure.

Fig. 3 . - - S h o w i n g s e v e r a l s m a l l c a l c i f i c a t i o n s in t h e r i g h t e e r e b r a ! c o r t e x .

" R e a d i n g was a b l a n k until suddenly she k n e w all the words in her first preprimer. A f t e r that, she seemed to grasp w o r d s ; a l t h o u g h it w a s m e m o r y or words as a whole and not g r a s p e d alphabetically or phonetieMly. " N u m b e r w o r k was b e y o n d comprehension. She h a d no interest in it and her conception of n u m b e r s was quite retarded. N. was not a d a y d r e a m i n g pupil. She k e p t b u s y a n d did w h a t she could quietly b u t slowly. She looked quietly h a p p y and not at all f r u s t r a t e d . " On Aug. 25, 1948, d u r i n g the Course of a routine physical e x a m i n a t i o n it was n o t e d t h a t N . ' s r i g h t f u n d u s w a s the seat of an extensive chorioretinitis centering a b o u t the m a c u l a r region. The left fundus a p p e a r e d normal. Also it was f o u n d t h a t there was some a t r o p h y of the muscles of her left hand. Roentgen studies of the skull r e v e a l e d several small ealcifieations in the right cerebral cortex (Fig. 3), r e s e m b l i n g those seen in toxoplasmosis. Because of the ehoriorctinitis and intraeranial calcifications, a tentative diagnosis of toxo-

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plasmosis was m a d e and the child was r e f e r r e d to the Uveitis Clinic at the U n i v e r s i t y of California H o s p i t a l for confirmation of this diagnosis. There the following tests and results were r e p o r t e d as follows : Tuberculin, second strength Brucella skin test Brucella agglutination Brueella complement fixation Frei test

Coc~idio~ycosis, skin test Histoplasmin , skin test Staphylococcus antigens Q-fever neutralization Toxoplasmosin, skin

PPD 4-plus -------

-

plus over

neg. neg. neff. neg. neg. neg. neg.

neff. minus

I t was also r e p o r t e d t h a t clinical findings on the child were typical of toxoplasmosis, and the failure to obtain a positive neutralization was due to a technical difficulty e n c o u n t e r e d in her test. A neutralization test for toxoplasmosis p e r f o r m e d on the child's m o t h e r was s t r o n g l y positive.

Fig. 4 . - - S c a t t e r e d l i m e s a l t s d e p o s i t s in fixed b r a i n v a r y f r o m 0.1 to 2.5 era. in d i a m e t e r .

I n N o v e m b e r , 1948, the child was a g a i n r e f e r r e d to a thoracic surgeon for an a p p r a i s a l of the p u l m o n a r y pathology. A c o m p a r i s o n of chest films t a k e n at this time with those t a k e n one y e a r p r e v i o u s l y revealed no increase in the size of the cyst, which m e a s u r e d 9 • 5.3 cm. in the a n t e r o p o s t e r i o r projection. I t exhibited a b o u t the same a m o u n t of fluid, o c c u p y i n g r o u g h l y the lower fifth of the cyst. No compression atelectasis of the s u r r o u n d i n g lungs was noted, and the thickness of the cyst wall h a d not increased. The thoracic s u r g e o n felt t h a t this r e p r e s e n t e d a congenital p u h n o n a r y cyst which should be removed. On Dec. 10, 1948, a t h o r a e o t o m y was done and the entire cyst excised. The cyst was a t t a c h e d along the lateral wail of the thorax, a n d t h e principal p o r t i o n was encased b y p u l m o n a r y tissue of the left u p p e r lobe. E x t e n s i v e fibrous reaction a n d i n f l a m m a t o r y adhesions of long s t a n d i n g w e r e encountered at all sites. The child was in good condition i m m e d i a t e l y a f t e r surgery. However, she u n e x p e c t e d l y w e n t into shock on the second p o s t o p e r a t i v e d a y and expired within a f e w hours.

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The following is a portion of the pathologist's report: " T h e brain after fixation eontains scattered small lime salt deposits which are demonstrated by x-ray and to a less extent grossly. These vary from 0.1 to 2.5 era. in diameter. There is a narrow, slightly depressed area 7 era. long in the right eerebral cortex. Beneath this area is a diverticulumlike protrusion of the right lateral ventricle which is 5 era. in diameter. This is located on a line with the anterior end of the temporal lobe. The brain tissue over this area is only 0.3 era. thick as compared to 4.5 era. on the uninvolved side." Histologic examination: " B r a i n : There are areas of cortical degeneration with calcification, regional gliosis, and thiekened leptomeninges. No toxoplasms are identified, although the changes present are consistent with a diagnosis of healed cerebral toxoplasmosis." The eye findings in this ease will be reported by Dr. Hogan in his series of cases from the Uveitis Clinic at the University of California. In May, 1949, Dr. Frenkel stated that " a b o u t thirty-five eases of toxoplasmosis in human beings have bee~l reported to date diagnosed either by autopsy or animal inoculation." Cases of toxoplasmosis in children have been reported from North and South Amer'ica and Europe. There is a wide geographical distribution of the cases in the United States. Sulfadiazine and Sulfamerazine control experimental toxoplasmosis in animals, but the toxoptasma may persist in the form of pseudocysts in the neural tissues of the host. Other drugs reported by Frankel as successful in treatment of toxoplasmosis include aureomycin, chloromycetin, Polymyxin B, and bacitraein. REFERENCES Callahan, W. P., Jr.: Incidence of Toxoplasma I n f e c t i o n in the St. Louis Area, Prec. Soc. Exper. Biol. & Med. 59: 68, 1943. Callahan~ W. P., J r , Russe]l, W. 0., and Smith, M. G.: H u m a n Toxoplasmosis. 01inicopathologic S t u d y W i t h P r e s e n t a t i o n of F i v e Cases and Review of the Literature, Medicine 25: 343-397~ 1946. Chatham~ E., and Blanc, G.: ~ o t e s et reflexions sur le toxoplasme et la toxoplasmose du gondi, Arch. Inst. P a s t e u r Tunis 10: 1, 1917. Frenkel, J. IK.: P a t h o g e n e s i s Diagnosis and T r e a t m e n t of H u m a n Toxoplasmosis, J. A. IV[. A. 140: 369-377, 1949. J a n k u , J.: P a t h o g e n e s i s and Pathologic A n a t o m y of Coloboma of the 1Kacula L u t e a in an Eye of Normal Dimensions and in a Microphthalmie Eye W i t h P a r a s i t e s in the Retina, Casop. lek. cesk. 62: 1021, 1054, 1081, 1111, 1923. Nicolle, C., and /V[arceaux, L.: Sur une injectione ~ carpo de L e i s h m a n (organismes roisins), Arch. Inst. P a s t e u r Tunis 4: 97~ 1909. Olafson, P., and Moulux, W. S.: Toxoplasma I n f e c t i o n in Animals, Cornell Vet. 32: 176, 1942. P i n k e r t o n , H. J., and I-Ienderson, R. G.: A d u l t Toxoplasmosis, Previously Unrecognized Disease E n t i t y S i m u l a t i n g Typhus. Spotted F e v e r Group, J. A. M. A. 116: 807, 1941. P i n k e r t o n , H., a n d Weinman~ D.: Toxoplasma I n f e c t i o n in ]Vlan, Arch, Path. 30: 374392, 1940. Sabin, A. B.: Toxoplasma N e u t r a l i z i n g A n t i b o d y in H u m a n Beings and Morbid Conditions Associated W i t h It, Proc. Soc. Exper. Biol. & Med. 51: 6, 1942. Sabin, A. B., a n d Ruchman~ I.: Characteristics of the Toxoplasma N e u t r a l i z i n g Antibody, Proc. Soc. Exper. Biol. & Med. 51: 1, 1942. Splendore, A.: U n Nuovo protozoa p a r a s i t e de conigli, Rev. Soc. Scient. 3: 109, 1908. Torres, C. ~I.: Sur une Nouvelle ~[aladie de l'homme, caracterise par la presence d~un p a r a s i t e intrace]ludaire tus proche du Toxoplasma et de l ' E n c e p h a l l t o z o o n dans le tissue musculaire cardiaque, ]e muscels du dque]Iette, Ie tissue ce]hl]aire sonscutan6 et le tissue nerveux, Compt. rend. Soc. de biol. 97: 1778, 1787~ 1797, 1927. Wolf~ A., and Cowen~ D.: Granu]omatous Encephalomyelitis Due to an Encephalitozoon, Bull. Neuroh Inst. New York 6: 306, 1937.