330

Letters to the Editor

The Journal o f Pediatrics August 1976

in pleural fluid in the second patient, the elevated serum transaminase values, the increased serum and urinary bilirubin levels, and the clinical picture support the diagnosis of viral hepatitis. CONCLUSION Since the pleural effusion and symptoms of hepatitis appeared concurrently in each patient, a n d since no other etiologic agent was found, we believe that viral hepatitis B m u s t be considered as a generalized disease a which m a y be associated with pleural effusion.

Pietro Cocchi, M.D. Mario Silenzi, M.D. Clinica Malattie Infettive dell'Universit& via L. Giordano, 13 50132 Florence, Italy REFERENCES 1. 2.

3.

Gross PA, and Gerding DN: Pleural effusion associated with viral hepatitis, Gastroenterology 60:898, 1971. Katsilabros L, Triandagillon G, Kontoyiannis P, and Katsilabros N: Pleural effusion and hepatitis. Gastroenterology 63:718, 1972. Conrad, ME, Schwartz, FD, and Young, AA: Infectious hepatitis, a generalized disease, A m J Med 37:789, 1974.

Hemolytic disease of newborn infant due to anti-U To the Editor: The blood group antibody, anti-U, was first identified in 1953 by Wiener, Unger, and Gordon 1 in the serum of an American black female. Burbi and associates 2 subsequently suggested that the antibody was the cause of erythroblastosis in a stillbirth. W e are presenting an infant who represents the first report of hemolytic disease of the newborn due to anti-U in which the antenatal m a n a g e m e n t was carried out using principles well established for RH hemolytic disease.

gestation were in Liley zone II. The foam test at 35 weeks' gestation indicated maturity of the fetal lungs. The infant had Apgar scores of 7 at 1 minute and of 9 at 5 minutes, respectively. Physical examination revealed a heart rate o f 140/minute and a respiratory rate of 48/minute. T h e liver edge was palpable 3 cm below the right costal margin and the spleen was palpable 2 cm below the left costal margin. There was no edema, ascites, or scleral icterus. Laboratory evaluation of cord blood revealed a total serum concentration of bilirubin of 4.4 m g / d l , a hemoglobin concentration of 10.5 g m / d l , a hematocrit value of 0.33, and a 4 + direct antiglobulin test. Microscopic examination of the peripheral blood smear revealed 170 nucleated red blood cells per high-powered field, and moderate anisocytosis and poikilocytosis. There were no spherocytes. A two volume exchange transfusion was performed at three hours of age because of the anemia and the strongly positive antiglobutin test. Blood for the exchange transfusion had been obtained from the American Association of Blood Banks, Milwaukee Blood Center. The infant was treated with phototherapy following the exchange transfusion. Her subsequent course was uncomplicated, and she was discharged from hospital at six days of age. The hematocrit value at three weeks o f age was 0.44. DISCUSSION Wiener and associates 1 selected the letter U for the new blood factor to avoid confusion with existing factors and to indicate a universal distribution. Additional studies ~ revealed that anti-U agglutinated the red blood cells of all 1,100 Caucasians who were tested, but failed to agglutinate the red blood cells of 12 of 989 blacks. Studies performed on the blood of relatives of four of the 12 U-negative individuals suggested that the factor was inherited by a pair o f allelic genes, U and u. Gene U controls the presence of the U factor and gene u its absence. The estimated genotype frequencies for blacks are 79.2% for U U , 19.6% for Uu, and 1.2% for uu. The phenotype U has been reported in Congo pygmies, South African, West African, and American blacks, and in an Indian family living in Natal. F u r t h e r investigations have revealed that the U cell antigen represents a distinct c o m p o n e n t of the MNSs gene complex and that there m a y be several alleles of the U antigen, U ~ and Ub. ~

Trina K. L. Austin, M.D. Jerry Finklestein, M.D. Donald M. Okada, M.D. Byron Myhre, M.D. Phillip Sturgeon, M.D. Department of Pediatrics Harbor General Hospital 1000 West Carson St. Torrance, Calif. 90509

CASE REPORT Int'ant M was a 2,434-gm female infant delivered by elective cesarian section after 35 weeks' gestation to a 20-year-old gravida 4, para 2, abortus 1 black female. Previous history revealed that her first pregnancy resulted in a 2,268-gm infant, and the second pregnancy terminated in a spontaneous abortion at 14 weeks' gestation. With the third pregnancy she delivered a 2,900-gin stillborn infant at 35 weeks' gestation. By a routine prenatal automated antibody screening method, one of us (P.S.) had identified a maternal anti-U serum titer of 1:128 at 8 weeks' gestation of the present pregnancy. At 30 weeks' gestation the maternal anti-U serum titer was 1:1024. The m o t h e r denied having received a blood transfusion. Results of serial amniocenteses performed at two-week intervals beginning at 31 weeks'

REFERENCES 1. Wiener AS, Unger LJ, and Gordon EB: Fatal hemolytic transfusion reaction caused by sensitization to a new blood factor U, J A M A 153:1444, 1953. 2. Burbi U, Degnan TJ, and Rosenfield RE: Stillbirth due to anti-U, Vox Sang 9:209, 1964. 3. Wiener AS, U n g e r L J, and C o h e n LP: Distribution and heredity of blood factor U, Science 119:734, 1954. 4. Go[dstein E, and Hoxworth PI: Investigation of the inheri-

Volume 89 Number 2

Letters to the Editor

tance of the U" and U ~ specificities of the U blood group factor, Transfusion 9:280 1969 (abstr). Stern K: Multiple differences in red cell antigens and isoimmunization, Transfusion 15:179, 1975.

Clostridia septicum bacteremia in a patient with aplastic anemia To the Editor." There are numerous reports in the literature of bacteremia with Clostridia species in patients with malignancies, especially of the hematologic variety.' :' To our knowledge, however, Clostridia bacteremia in a pediatric patient with aplastic anemia has not been reported. CASE REPORT

A 10-year-old black male, known to have idiopathic aplastic anemia for five months, was admitted with a 24-hour history of hematemesis and melena. The temperature was 38.6~ pulse 135 beats/minute, respiration 30/minute, and blood pressure 98/60 mm Hg. Ecchymotic areas were present on all extremities and a cardiac m u r m u r was heard. The only other notable physical findings were diffuse abdominal tenderness and slight tenderness of the right calf. Eight hours after admission the patient complained of pain and swelling of the right calf. There was no history of trauma to the calf. Crepitus was noted and seen on roentgenograms. Despite intravenous therapy with penicillin G, gentamicin, and clindamycin the patient's condition continued to deteriorate and he died 14 hours after admission. Blood cultures drawn prior to therapy eventually grew Clostridium septieum. Autopsy revealed hemorrhage and focal necrosis of the gastrointestional tract, Extensive gas and grampositive bacilli were found in the mucosa and submucosa of the colon. The right gastrocnemius also revealed many gram-positive bacilli and myonecrosis.

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case presented, the Clostridia localized in the tissues of the right calf and progressed to involve the thigh and abdomen. This aspect of the case is unique because it was not until this metastatic focus in the leg appeared, that the diagnosis of sepsis became evident. In the previous cases cited, '-~ the appearance of metastatic foci were not presenting manifestations of Clostridia sepsis. Although intravascular hemolysis was originally thought to be characteristic of Clostridia infection," there was no evidence of this phenomenon in our patient, or in the five pediatric patients reported by Alpern and Dowell.-' The data presented strongly suggest that when symptoms of acute abdomen are noted in a compromised host, especially when there is loss of integrity of the gastrointestional tract, bacteremia with Clostridia species should be suspected and anaerobic as well as aerobic pathogens should be sought in blood cultures. It is hoped that by routinely culturing the blood in such patients, appropriate therapy can be instituted before lethal metastatic loci develop. Blaise L. Congeni, M.D. George A. Nankervis, Ph.D., M.D. Department of Pediatrics Cleveland Metropolitan General Hospital 3395 Scranton Road Cleveland, Ohio 44109 REFERENCES

1. Wynne JW, and Armstrong D: Clostridial septicemia, Cancer 29:215, 1972. 2. Alpern RJ, and DoweU VR Jr: Clostridium septicum infections and malignancy, J A M A 209:385, 1969. 3. Cabrera A, Tsukada Y, and Pickren JW: Clostridial gas gangrene and septicemia in malignant disease, Cancer 18:800, 1965. 4. Bornstein DL, Weinberg AN, Swartz M, and Kunz L: Anaerobic infections: Review of. current experience, Medicine 43:207, 1964. 5. MacLennan JD: Histotoxic clostridial infections of man, Bacteriol Rev 26:177, 1962.

DISCUSSION Clostridia organisms invade the bloodstream primarily in patients with hematologic malignancies, l-a or hepatic cirrhosis, ~ especially when there is loss of integrity of the gastrointestional tract, i.e., hematemesis or melena. Five of the total of seven pediatric patients with clostridial sepsis repor!ed by Alpern and DowelF and Wynne and Armstrong' had e+idence of bleeding from the gastrointestional tract. Two of the three pediatric patients in the case reports of Cabrera and associates * were noted to have ulceration of t h e gastrointestional tract. It has been postulated that such a break in the integrity of the gastrointestional tract permits systemic penetration of gut organisms, the vast majority of which are anaerobes? Once systemic penetration has occurred, Clostridia may localize in devitalized tissues. In the From the Department of Pediatrics, Case Western Reserve University School of Medicine at Cleveland Metropolitan General Hospital.

Diazoxide-diphenylhydantoin interaction To the Editor: We read with interest the recent report by Roe and associates,' regarding the effect of diazoxide on diphenylhydantoin (DPH) metabolism. In their report of two case~,%therapeutic DPH levels were not attained with DPH doses of 17~and 29 m g / k g / d a y in the presence of diazoxide. We should like ~0 describe a similar case, including the serum and urinary response to a single IV injection of DPH, and suggest that the anticonvulsant may also interfere with the action of diazoxide.

Letter: Hemolytic disease of newborn infant due to anti-U.

330 Letters to the Editor The Journal o f Pediatrics August 1976 in pleural fluid in the second patient, the elevated serum transaminase values, th...
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