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J$-mediated hemolytic disease of the newborn infant LARRY STEVEN
R. ORRICK, H. GOLDE.
M.D. M.D.
Department of Obstetrics and Gynecology, Medical Center, Tripler ,4MC, Hawaii
Tripler
Arm?
THE JR” ERYTHROCYTE antigen occurs with a very high frequency in the general population. Stroup and MacIlroy’ first reported sensitization to the Jra antigen in 1970 and screened 11,000 donors in the United States without detecting a single Jra-negative individual. MacIlroy and Stroup subsequently evaluated more than 18 patients with anti-Jr” antibody, and almost half were Japanese. In the Japanese population studied by Nakajima and Ito,’ 19,298 individuals were screened for JP antigen. The incidence of JP-negative persons was 0.03%. These authors described the onl) case reported to date of hemolytic disease of the newborn infant associated with JP sensitization. The affected offspring had a strongly positive direct antiglobulin titer and required phototherapy for hyperbilirubinemia. Recently we encountered a sensitized JPnegative patient and herein describe the management and outcome of the pregnancy.
A 35-year-old patient of Japanese ancestry, gravida 5, para 4, was noted on initial prenatal antibody screening at 10 weeks’ gestation to have a weakly reactive IgG antibody subsequently identified as anti-Jra. Initial antibody titers at 27 weeks’ gestation were negative with respect to saline and alThe views expressed herein are those of the authors and do not necessarily reflect the views of the United States Army or the Department of Defense. Reprint requests: Steven H. Golde. M.D., Department of Obstetrics and Gynecology, Tripler Army Medical Center. Tripler AMC. Hawaii 96859.
bumin titers; Coombs and Ficin (enzyme) Coombs titers were positive at < 1 : 1 and 1 : 4 dilutions, respectively. An initial amniocentesis for genetic screening performed at 17 weeks’ gestation resulted in a normal male karyotype. Five amniocenteses were performed for determination of bilirubin between 27 and 37 weeks’ gestation. The bilirubin value was initially in the low area of zone II at 27 weeks (A optical density at 450 nm = 0.147) and fell to zone 1 at 37 weeks (A optical density at 450 nm = nondetectable). A male infant, with Apgar scores of 7 and 9. blaod type A+, weighing 3,400 grams, was delivered spontaneously at 40 weeks’ gestation. The infant appeared to be of term gestation by clinical parameters, and the initial physical examination was unremarkable; there was no hepatosplenomegaly, icterus, or jaundice. At 24 hours of life the infant was visibly jaundiced with a total serum bilirubin value of 10.5 mgi100 ml. Total protein and hematocrit values were 5.4 mg/lOO ml and 50%, respectively. Phototherapy was initiated, and bilirubin determinations remained at 10 mg/lOO ml through the third day with a hematocrit 54% to 55% and total protein at 5.4 gm/lOO ml. Initial cord blood antigen/antibody studies with Ficintreated cells revealed an anti-Jr* antibody with comparable specificitv ttr maternal serum antibody.
The present case represents the second documented report of anti-Jr”-mediated hemolytic disease of the newborn infant. TritchlerL’ described a Jr”-sensitized pregnant patient who was monitored with serial amniocenteses and had an elevation in bilirubin. The infant was delivered at 3.5 weeks for presumed distress on the basis of elevated amniotic fluid creatinine. Hyperbilirubinemia developed in the neonate; however, no could be demonstrated in the Jr a incompatibilities newborn infant. Bilirubin measurements peaked at 8.2 mgi 100 ml at 2 days of life, and the hyperbilirubinemia was attributed to prematurity. In the present instance. and in the case reported by Nakajima and Ito, the anti-Jr” antibody detected in cord blood studies was best demonstrated by the Ficin antiglobulin technique. The two cases reported have both resulted in minor hemolytic manifestations. However, antenatal amniotic fluid assessment of fetal well-being in utero is probably 135
136
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prudent at the present time due to the relative lack of reported clinical experience with this antigeniantihod! system. The antibody titers in maternal sera are similar to the findings of Kendall, 1 who reported on a case in which anti-Jr” antibody did not react in saline or albumin systerns at 37” C. Kendall demonstrated the potent) of the anti-Jr” antibody in viuo 1>) means of 3’(Zr labeled erythrocytes. The half life of Jr”-positive eryt hrocytes in a Jr”-negative, sensitized recipient was only 80 minutes, suggesting that only Jr”-negative blood be used for transfusion in these parients. Therefore, identification of’the Jr”-sensitized pregnant woman requires that blood for both fetus and mother be located well in advance of’ anticipated delivery or cesarean section, because of demonstrated in viva potenc)- of the antigen and the rarity of compatible donors.
of corium. A neoplastic proliferation intrgrarctl h! groups cjt cells in the basal srrata of the epithelium was ohsc~ veti (FIN. I ). The cellular elements consisred of regular- nucleii. whi( h ~\WY small and hyperchromatic. with slightI\ visible ~1 topl;~sn~. No positive diagnosis could be made with this biops\ but the poss+ilily of metastasis of a pool-ly differentiarrd (arcinoma ~-2s suggested. The rest of the g\necologiral esamination showed a leiomyomatous uterus and appar-entl) c ksrir masses in both ovaries. Because of rhe clinital and hir)l+ findings, hysrerectomy and bilateral adnexectom) WCI-c done,
including excision of the site of the previous hiopsv in the upper rhird of the vagina. The pathologic examination t)f the sample
showed
bilateral
ovarian
endometriosis
u.ith c\sts
and
multiple uterine leiomyomas. A semiserial study of thr ~~ppct third of the vagina showed some remnants of the c-arc-inomatous proliferation, similar to those described in the prr\ious biopsy. ‘There was a pattern of cellular clustering that was definitely epithelial. with pallisading and a general structure similar to that of- a basocellular skin epithelioma (Fig. 2). 2’11~ neoplasm infiltrated the stroma without distortion, and rhrr-e
REFERENCES
was a clear line of demarcation,
Stroup, M., and MacIlroy, M.: Jr-Five examples of an antibody defining an antigen of high frequency in the Caucasian population, Proceedings of the Twenty-third Annual Meeting of the American Association of Blood Banks, San Francisco, 1970, p. 86. Nakajima, H., and Ito. K.: An example of anti-Jr” causing hemolytic disease of the newborn and frequency of the Jr” antigen in the Japanese population, VOX Sang. (Base])
margins. In the lining of the neoplasm the superficial vaginal epithelium showecl a discrete maturalional disorder. wirh cells
35:265,
Tritchler, 17:177,
1978.
J. E.: An example
of anti-Jra, Transfusion
1977.
Kendall, A. G.: Clinical impor-tance of the rare erythrocyte antibody anti-.Jr”. ‘l‘ransfllsion 16:646. 1976.
Basal cell-like carcinoma in the upper third of the vagina ARIEL E. NAVES JORGE A. MONT1 EDUARDO CHICHONI
EPIDERMOID CARCINOMA is the malignant entity most frequently seen in the vagina, followed by various types ofadenocarcinoma, which has a relationship with mesonephric remnants.’ In this report an account is given of detection of proliferation of a vaginal neoplasm that morphologically resembles that described by Tchertkoff and Sedlisz in the uterine cervix and called “cylindroma” or “basal adenoid carcinoma.”
A 42-year-old, white, nulliparous woman was examined because of metrorrhagia. without an? antecedent hormonal treatment. The gynecologic examination detected a projecting nodule in the upper third of the posterior vaginal wall, which measured 0.8 cm in its largest diameter. It was partially removed by biopsy. The biopsy specimen was wholly lined by nonulcerated vaginal epithelium and included a scarce amount OOOZ-9378/80/090136+02$00.20/0
@) 1980 The C. V. Moshy Co.
having
larger
nucleii
than
develop a clear horizontal These
changes
could
with
usual
no tumor
and without
at the WI-gkal
the lendrncy
polarity in the superficial
he called
dysplasia
(Fig.
to strata.
2).
This vaginal neoplasm has not been described in the literature. It is a carcinoma f’ormed by cellular groups with a pattern similar IO that of a basal cell epithelioma of’ the skin. It has a pattern of stromal infiltration that does not disrupt the conjunctive elements ot‘ the vaginal corium. and intraepithelial nests are found a~ the level of’ the basal layer of the vaginal epithelium. This tumor also has intraepithelial changes resembling dysplasia in the superficial vaginal epithelium. The morphologic features of‘the lesion remind LIS of the group of uterine cervical carcinomas described in the literature as “cylindromas” or “basal adenoid carcinomas.” Rosen and Dolan” extensively reviewed the literature on this neoplasm and separated these tumors into two groups, one with morphologic characteristics observed by both the optical microscope and the elcctron microscope similar to those observed in cylindramas at other locations (mammary gland. salivar) gland, etc.). There leas a second group with an infiltrative pattern similar to that of the cylindroma but formed by basal cells, without c>lindroma-like internal distribution. and characterized b\ ;I t’reque~lt association \vith invasive carcinoma of the uterine ccl-vis. These authors suggested that the latter morphologic. variety has a histogenetic origin from superficial or glandillar epithelium whereas the real cervical c\ lindroma is probably of mesonephric origin. We feel this case should be included in the group morphologicall) similar to that described by Rosen and Dolan as the group of carcinomas with basal cells. The peculiarity of this tumor is its localization in the upperthird of the vagina and not in the uterine cervix. The presence of the intraepithelial nests of ncoplastic pro-