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Fetal dystocia due to neuroblastoma of the adrenals with metastases to the liver
440
AMERICAN JOURNAL OF OBSTETRICS AND GYNECOLOGY
References l. Gunning, R. B., and Ross, C. A.: 8tug., Gynee. & Obst. 70: 230, l!JW. 2. Barnes, A. 0.: AM. J. OBST. & GYNEr;. 41: 135, January, HJ41. 3. Pfannenstiehl, J.: Virehow 's Arch. 127: 305, 189~. 4. N.ehrkorn, A.: Virchow 's Arch. 151: :i2, 1898. 5. Wilms, M:.: Mischgeschwulste, Leipzig, 1899, 1L Georgi. 6. Menckelberg, J. G.: Virchow's Arch. 1'. path. Anat. 187: Hl, Hl07. 7. Cohnheim, J.: Virchow 's Areh. 65: Ii-i, 18/ii. 8. Weber, 0.: Virchow's Arch. f. path. Anat. 39: 216, 1867. 9. Glass, M., and Goldsmith, J. W.: A~L J. Om:lT. & GY:'\IEC. 41: 809, Peb., 1941. 10. Ewing, J.: Neoplastic DiseaseR, -1 Nl., Philadelphia, 1P40, W. B. Saunders Co. 11. Lochrane, C. D.: Proc. Roy. Soc. Meil. 26: 1429, Hl:l::>. 12. Snow, W.: Personal Communication to the Author. 13. Snow, W.: Clinical Roentgenology of Pregnancy, i'lpringtidd, Ill., 1942, Thoma~. Ehrlich, H. E.: AM. J. 0Bs~·. & GYNEC. 43: .1 021, 1942. Hoffmann, B. J.: AM. J. 0BST. & GYNEr'. 40: 28!J, ] n-10. Lebowich, R. J., et al.: Surgery 10: 411, 1\l-U. Shapior, R.: AM. J. 0BST. & ClYNE<·. 21: s:!, l ~:; 1.
FETAL DYSTOCIA DUE TO NEU:&OBLAITOlttA OF TBE ADRENALS WITH METASTASES TO THE LIVER TOBIAS WEINBERG,
M.D.,
A~D
H.
l\b~LVIN HADMA~,
B.S., M.D., F.A.C.S.,
BALTLMORE, .1\iD.
(!!'rom the Laboratories and Ol;stctric Srn:i1w of 8·inai Hospital)
LTHOUGH a number of eases of neuroblastoma of the adrenal with metastases to the liver in newborn infants or those several days old have been reported, only two reports can be found in the literature in which dystocia was caused by the presence of the tumor mass. The case report~d by Hagstrom1 described a breech delivery in which ob~ structing parts were encountered at the level of the trunk of the child. Digital examination revealed the prcsenee of a huge liver which later proved to be due to the presenee of m<•tastatic tumor masses. Portions of the liver had to he remowd lwfor<> deliYery eould be eompleted. Askin and Geschickter 's casc 2 wa:s that of a full-term fetus in which the abdomen had to be incised and fragments of the enlarged liver removed before the fetus could lw delivered. The apparent rarity of the eomplication of dystocia of this origin prompts the following report.
A
Case Report 'l'he mother of the infant was a twenty-five year-old primigravida, who was admitted to the Sinai Hospital with a history of spontaneous rupture of the membranes four honrs before admission. Prenatal examination had revealed a normal hony pelvis, whose configuration was such as not to interfere with the normal delivery of an average-sized infant. The antepartum course had been eompletely uneventful. After admission to the hospital, eontractions of the uterus beg~n only after stimulation with castor oil, quinine, and divided doses of pituitrin intramuscularly. Cervical dilatation progressed slowly and the head of the infant remained high in the pelvis until full dilatation, which occurred twenty-four hours after n~ptnre of the membranes. Soon after full dilatation the head descended to the midpelv.ic region, but further progress ceased. Approximately one and one-half hours after full dilatation, a midforceps operation was perfm·med and the patient was delivered of a live male child weighing 2,920 grams. No difficulty
WElNBERG ANb RAbMAN :
F'ETAL bYSTOClA
441
had been encountered in delivery of the head, but hard pressure from above had been required to deliver the torso, which appeared to encounter an obstruction. The cause of the difficulty was quite evident after the delivery of the infant, since marked distention of the abdomen, apparently with fluid, was obvious. The child died one hour after delivery. Gross Necropsy Findings.-The body was that of an apparently normally developed, full-term male child measuring 47 em. in length. The striking feature was the protuberance of the abdomen and the bulging in both flanks. The peritoneal cavity was found to contain approximately 200 c.c. of bloody fluid. The liver was greatly enlarged and extended to both lateral borders of the abdominal wall. The right lobe extended three fingerbreadths below the costal margin in the right midclavicular line; the left lobe was found to extend two fingerbreadths below the costal margin in the left midclavicular line. The parietal peritoneum in the region of the right kidney bulged forward rather .
Fig. 1.-Surface of liver showing bosselations produced ·b y metastases.
prominently, forming an oval-shaped mass measuring approximately 10 by 8 by 5 em. The peritoneum overlying this area had a bluish sheen. Further investigation in this area revealed a deeply hemorrhagic mass measuring 8 by 6 by 3 em., which occupied the site of the right adrenal and extended downward over the anterior surface of the kidney. It compressed the latter but was distinctly separate from it. It was soft and palpably cystic. Upon sectioning the .mass, considerable bloody fluid escaped, revealing the presence of numerous blood clots. In addition, areas of firm grayish-brown and red tissue were also visible. At the upper pole of the mass there was a thin rim of golden-yellow tissue resembling adrenal cortex. The left adrenal was found in its normal position and was relatively normal in shape but proportionately enlarged in all diameters. It measured 4 by 2 by 0.8 em., weighed 5.8 grams, and felt nodular. The cut surface showed a well developed .golden-yellow cortex, which surrounded oval-shaped nodules of grayishwhite tissue averaging approximately 1.0 em. each in diameter.
442
AMERICAN JOURNAL OF OBS'l'ETRICS AND GYNECOLOGY
Fig. 2.-Cut surface of liver showing extensive replacem ent of liver parenchyma by neoplastic tissue.
Fig. 3.-A. Section through rim of right adrenal tumor m·ass showing an area of Intact adrenal cortex above an area of Invasion bY the neoplasm. B. Section through the tumor mass In the right adrenal showing a typical area.
WEI~BERG
AND
RADMA~:
FETAL DYSTOCIA
443
The liver was huge, weighed 260 grams, and measured 15 by 8 by 4.5 em. The usual smooth, reddish-brown surface was interrupted practically everywhere by reddish and reddish-gray nodules of varying size, which produced a bosselated appearance (Fig. 1). The nodules varied in size from several millimeters to almost 2 em. Some of the nodules showed central umbilication. The cut surfaces revealed replacement of most of the liver parenchyma by nodules of grayish-white and brown tumor tissue (Fig. 2). The heart was normal in size and shape, and, except for the presence of a patent foramen ovale and patent ductus Botallo, was not remarkable. The other organs showed no gross abnormalities. :Microscopic Examination.-Sections of the right adrenal tumor mass showed large areas of hemorrhage, within which, there were strands and cords of tumor cells, which were round, had a scanty eosinophilic cytoplasm and large round and oval nuclei. Scattered mitoses were seen. In some areas formations suggestive of ''rosettes'' were seen. The golden-yellow rim of tissue at the upper pole was found to consist of intact adrenal cortex into which scattered strands of tumor tissue were seen to infiltrate (Fig. 3). The sections of the left adrenal showed similar tumor cell masses, which <'ccupied the medullary region, but extended into the cortex, being seen particularly in the interfascicular blood vessels of the latter. Small nests of tumor cells were found in the interstitial and capsular tissue of the pancreas, in a lymphatic in the wall of the gall bladder, and in blood vessels in the myocardium and cerebrum.
Comment The pathogenesis and histogenesis o:f the neoplastic involvement o:f the adrenals and liver will not be discussed, inasmuch as numerous erudite presentations are already present in the literature. 2 " 7 The probable mechanism effective in producing dystocia is, on the other hand, of pertinent interest. Just as in the cases previously cited/" 2 the liver in this case was greatly enlarged. But in addition, a bloody effusion was present in the peritoneal cavity in this infant. This certainly contributed to the difficulty of passage of the fetus through the birth canaL The presence of the fluid can be accounted for on the basis of peritoneal irritation by the tumor masses present in the liver. Although hemorrhagic degeneration in a neuroblastoma has been encountered quite frequently, it is enticing to use the presence of hemorrhage in this case to explain the rapid demise of the infant following delivery. It is possible, and even probable, that very little hemorrhagic infiltration was present in the tumor mass of the right adrenal prior to the onset of labor. This appears to be confirmed by the evident recent nature of the blood clot found in the tumor mass. Therefore, the following evolution of events is offered as being quite likely. With the onset of labor the head passed through the lower uterine segment with ease. However, when the abdomen approached this region, the muscular wall of the uterus failed to dilate sufficientlv to accommodate the unusual width, thus forming a constricting band. Therefore, increased pressure wa2 exerted upon the abdomen of the fetus. Inasmuch as the liver was huge, fluid was present. in the abdomen and the ribs at this age are quite supple, the constricting force would be transmitted by the above tissues and fluid to the soft tumor mass in the right
444
AA>lERICAN JOURNAL OF OBSTETRICS AND GYNl':COI,OGY
adrenal region. Ultimately a crushing pressure would be exerted and then hemorrhage would occur into the disrupted tissue of the tumor mass. Accordingly, as the left adrenal cortex was partly invaded by tumor tissue and only little of the eortex of the right adrenal remained intact, one might blame death upon the shoek concomitant with sudden interruption of a sufficient suppl~' of adrenal (•ortical hormone to maintain the vis a tergo. References Hagstrom, H. 'l'.: AM. ,J. OBST ..\NJJ (hxEo. 19: fi'i:\ Hno. Askin, J. A., anrl Geschiekter, C. P.; .T. Perliat. 7: 1;31, l!Jil;i. Scott, E., Oliver, .M.G., and Oliver, .M. IL: Am . •J. Cancer 17: il\Hi, J!f:l:L Thompson, R. M.: Ann. Int. Med. 16: 1ll0ll, June, 1942. Lewis, D., and Gesehickter, C. F.: Arc· h. Surg. 28: Hi, l!l:J-4. Redman, J. L., Agerty, H. A., Barthmaier, 0. F., anr1 Fisher, II. R.: .Am.•T. Dis. Child. 56: 10£t7, HJ:lR. 7. Blacklock, .T. W. S.: .T. Path. and B:u·t. 34: 27, 1!ia+.
J. 2. :J. 4. 5. 6.
RECURRENT PLACENTA PREVIA PAUL
H.
P~IED,
M.D.,
PHIT"ADJ!;LPHIA, PA., "\ND RICHARD TORPIN, AUG"LS'PA,
M.D.,
(iA.
(From the Department of Obstetrics and Gynecology, Un.i11en;ity of Georgia School of Medicine rmd Hospital)
T
HE incidence of placenta previa varies from 1:150, as reported by the Committee on Prenatal Maternal Care of the White House Conference, to 1:200 to 1:1,500 as noted by various authors. 1 However, a review of the literature reveals that only nine cases of recurrent placenta previa in successive pregnancies, 3-7 and one in nonsuccessive pregnancies8 have been reported. l'lore cases of this type have un.doubterlly been observed but not reported, for De Lee1 and Rivett/ in discussing rt1current placenta previa, speak of a greater than reported :frequency. A much larger number of cases in successive pregnancies should be expected in considering the incidence and the predisposing factors of placenta previa. However, this low incidence is apparently du1,3 to a failure to report cases and to a few principles of prophylaxis, which may also be of avail in preventing the abnormality. I desire to present another case of recurrent placenta previa in successive pregnancies.
Case Report Mrs. A. C., aged 19, in the thirty-second week of her first pregnancy, entered the University Hospital at 9 :45 P.r.r. on lVIay 7, 1941, because of sudden, painless vaginal bleeding. The estimated loss of blood was 300 c.c. The prenatal period, as observed in our clinic, was within normal limits. Examination revealed a white female, who had moderate vaginal bleeding and who was not in labor. Her blood pressure was 130/80 and the pulse rate was 100. The uterus had a one-plus tone, was slightly tender in the lower segment area, and measured 28 crri. above the symphysis pubis. A single fetus lay in the L.O.P. position \\
AMERICAN JOURNAL OF OBSTETRICS AND GYNECOLOGY
References l. Gunning, R. B., and Ross, C. A.: 8tug., Gynee. & Obst. 70: 230, l!JW. 2. Barnes, A. 0.: AM. J. OBST. & GYNEr;. 41: 135, January, HJ41. 3. Pfannenstiehl, J.: Virehow 's Arch. 127: 305, 189~. 4. N.ehrkorn, A.: Virchow 's Arch. 151: :i2, 1898. 5. Wilms, M:.: Mischgeschwulste, Leipzig, 1899, 1L Georgi. 6. Menckelberg, J. G.: Virchow's Arch. 1'. path. Anat. 187: Hl, Hl07. 7. Cohnheim, J.: Virchow 's Areh. 65: Ii-i, 18/ii. 8. Weber, 0.: Virchow's Arch. f. path. Anat. 39: 216, 1867. 9. Glass, M., and Goldsmith, J. W.: A~L J. Om:lT. & GY:'\IEC. 41: 809, Peb., 1941. 10. Ewing, J.: Neoplastic DiseaseR, -1 Nl., Philadelphia, 1P40, W. B. Saunders Co. 11. Lochrane, C. D.: Proc. Roy. Soc. Meil. 26: 1429, Hl:l::>. 12. Snow, W.: Personal Communication to the Author. 13. Snow, W.: Clinical Roentgenology of Pregnancy, i'lpringtidd, Ill., 1942, Thoma~. Ehrlich, H. E.: AM. J. 0Bs~·. & GYNEC. 43: .1 021, 1942. Hoffmann, B. J.: AM. J. 0BST. & GYNEr'. 40: 28!J, ] n-10. Lebowich, R. J., et al.: Surgery 10: 411, 1\l-U. Shapior, R.: AM. J. 0BST. & ClYNE<·. 21: s:!, l ~:; 1.
FETAL DYSTOCIA DUE TO NEU:&OBLAITOlttA OF TBE ADRENALS WITH METASTASES TO THE LIVER TOBIAS WEINBERG,
M.D.,
A~D
H.
l\b~LVIN HADMA~,
B.S., M.D., F.A.C.S.,
BALTLMORE, .1\iD.
(!!'rom the Laboratories and Ol;stctric Srn:i1w of 8·inai Hospital)
LTHOUGH a number of eases of neuroblastoma of the adrenal with metastases to the liver in newborn infants or those several days old have been reported, only two reports can be found in the literature in which dystocia was caused by the presence of the tumor mass. The case report~d by Hagstrom1 described a breech delivery in which ob~ structing parts were encountered at the level of the trunk of the child. Digital examination revealed the prcsenee of a huge liver which later proved to be due to the presenee of m<•tastatic tumor masses. Portions of the liver had to he remowd lwfor<> deliYery eould be eompleted. Askin and Geschickter 's casc 2 wa:s that of a full-term fetus in which the abdomen had to be incised and fragments of the enlarged liver removed before the fetus could lw delivered. The apparent rarity of the eomplication of dystocia of this origin prompts the following report.
A
Case Report 'l'he mother of the infant was a twenty-five year-old primigravida, who was admitted to the Sinai Hospital with a history of spontaneous rupture of the membranes four honrs before admission. Prenatal examination had revealed a normal hony pelvis, whose configuration was such as not to interfere with the normal delivery of an average-sized infant. The antepartum course had been eompletely uneventful. After admission to the hospital, eontractions of the uterus beg~n only after stimulation with castor oil, quinine, and divided doses of pituitrin intramuscularly. Cervical dilatation progressed slowly and the head of the infant remained high in the pelvis until full dilatation, which occurred twenty-four hours after n~ptnre of the membranes. Soon after full dilatation the head descended to the midpelv.ic region, but further progress ceased. Approximately one and one-half hours after full dilatation, a midforceps operation was perfm·med and the patient was delivered of a live male child weighing 2,920 grams. No difficulty
WElNBERG ANb RAbMAN :
F'ETAL bYSTOClA
441
had been encountered in delivery of the head, but hard pressure from above had been required to deliver the torso, which appeared to encounter an obstruction. The cause of the difficulty was quite evident after the delivery of the infant, since marked distention of the abdomen, apparently with fluid, was obvious. The child died one hour after delivery. Gross Necropsy Findings.-The body was that of an apparently normally developed, full-term male child measuring 47 em. in length. The striking feature was the protuberance of the abdomen and the bulging in both flanks. The peritoneal cavity was found to contain approximately 200 c.c. of bloody fluid. The liver was greatly enlarged and extended to both lateral borders of the abdominal wall. The right lobe extended three fingerbreadths below the costal margin in the right midclavicular line; the left lobe was found to extend two fingerbreadths below the costal margin in the left midclavicular line. The parietal peritoneum in the region of the right kidney bulged forward rather .
Fig. 1.-Surface of liver showing bosselations produced ·b y metastases.
prominently, forming an oval-shaped mass measuring approximately 10 by 8 by 5 em. The peritoneum overlying this area had a bluish sheen. Further investigation in this area revealed a deeply hemorrhagic mass measuring 8 by 6 by 3 em., which occupied the site of the right adrenal and extended downward over the anterior surface of the kidney. It compressed the latter but was distinctly separate from it. It was soft and palpably cystic. Upon sectioning the .mass, considerable bloody fluid escaped, revealing the presence of numerous blood clots. In addition, areas of firm grayish-brown and red tissue were also visible. At the upper pole of the mass there was a thin rim of golden-yellow tissue resembling adrenal cortex. The left adrenal was found in its normal position and was relatively normal in shape but proportionately enlarged in all diameters. It measured 4 by 2 by 0.8 em., weighed 5.8 grams, and felt nodular. The cut surface showed a well developed .golden-yellow cortex, which surrounded oval-shaped nodules of grayishwhite tissue averaging approximately 1.0 em. each in diameter.
442
AMERICAN JOURNAL OF OBS'l'ETRICS AND GYNECOLOGY
Fig. 2.-Cut surface of liver showing extensive replacem ent of liver parenchyma by neoplastic tissue.
Fig. 3.-A. Section through rim of right adrenal tumor m·ass showing an area of Intact adrenal cortex above an area of Invasion bY the neoplasm. B. Section through the tumor mass In the right adrenal showing a typical area.
WEI~BERG
AND
RADMA~:
FETAL DYSTOCIA
443
The liver was huge, weighed 260 grams, and measured 15 by 8 by 4.5 em. The usual smooth, reddish-brown surface was interrupted practically everywhere by reddish and reddish-gray nodules of varying size, which produced a bosselated appearance (Fig. 1). The nodules varied in size from several millimeters to almost 2 em. Some of the nodules showed central umbilication. The cut surfaces revealed replacement of most of the liver parenchyma by nodules of grayish-white and brown tumor tissue (Fig. 2). The heart was normal in size and shape, and, except for the presence of a patent foramen ovale and patent ductus Botallo, was not remarkable. The other organs showed no gross abnormalities. :Microscopic Examination.-Sections of the right adrenal tumor mass showed large areas of hemorrhage, within which, there were strands and cords of tumor cells, which were round, had a scanty eosinophilic cytoplasm and large round and oval nuclei. Scattered mitoses were seen. In some areas formations suggestive of ''rosettes'' were seen. The golden-yellow rim of tissue at the upper pole was found to consist of intact adrenal cortex into which scattered strands of tumor tissue were seen to infiltrate (Fig. 3). The sections of the left adrenal showed similar tumor cell masses, which <'ccupied the medullary region, but extended into the cortex, being seen particularly in the interfascicular blood vessels of the latter. Small nests of tumor cells were found in the interstitial and capsular tissue of the pancreas, in a lymphatic in the wall of the gall bladder, and in blood vessels in the myocardium and cerebrum.
Comment The pathogenesis and histogenesis o:f the neoplastic involvement o:f the adrenals and liver will not be discussed, inasmuch as numerous erudite presentations are already present in the literature. 2 " 7 The probable mechanism effective in producing dystocia is, on the other hand, of pertinent interest. Just as in the cases previously cited/" 2 the liver in this case was greatly enlarged. But in addition, a bloody effusion was present in the peritoneal cavity in this infant. This certainly contributed to the difficulty of passage of the fetus through the birth canaL The presence of the fluid can be accounted for on the basis of peritoneal irritation by the tumor masses present in the liver. Although hemorrhagic degeneration in a neuroblastoma has been encountered quite frequently, it is enticing to use the presence of hemorrhage in this case to explain the rapid demise of the infant following delivery. It is possible, and even probable, that very little hemorrhagic infiltration was present in the tumor mass of the right adrenal prior to the onset of labor. This appears to be confirmed by the evident recent nature of the blood clot found in the tumor mass. Therefore, the following evolution of events is offered as being quite likely. With the onset of labor the head passed through the lower uterine segment with ease. However, when the abdomen approached this region, the muscular wall of the uterus failed to dilate sufficientlv to accommodate the unusual width, thus forming a constricting band. Therefore, increased pressure wa2 exerted upon the abdomen of the fetus. Inasmuch as the liver was huge, fluid was present. in the abdomen and the ribs at this age are quite supple, the constricting force would be transmitted by the above tissues and fluid to the soft tumor mass in the right
444
AA>lERICAN JOURNAL OF OBSTETRICS AND GYNl':COI,OGY
adrenal region. Ultimately a crushing pressure would be exerted and then hemorrhage would occur into the disrupted tissue of the tumor mass. Accordingly, as the left adrenal cortex was partly invaded by tumor tissue and only little of the eortex of the right adrenal remained intact, one might blame death upon the shoek concomitant with sudden interruption of a sufficient suppl~' of adrenal (•ortical hormone to maintain the vis a tergo. References Hagstrom, H. 'l'.: AM. ,J. OBST ..\NJJ (hxEo. 19: fi'i:\ Hno. Askin, J. A., anrl Geschiekter, C. P.; .T. Perliat. 7: 1;31, l!Jil;i. Scott, E., Oliver, .M.G., and Oliver, .M. IL: Am . •J. Cancer 17: il\Hi, J!f:l:L Thompson, R. M.: Ann. Int. Med. 16: 1ll0ll, June, 1942. Lewis, D., and Gesehickter, C. F.: Arc· h. Surg. 28: Hi, l!l:J-4. Redman, J. L., Agerty, H. A., Barthmaier, 0. F., anr1 Fisher, II. R.: .Am.•T. Dis. Child. 56: 10£t7, HJ:lR. 7. Blacklock, .T. W. S.: .T. Path. and B:u·t. 34: 27, 1!ia+.
J. 2. :J. 4. 5. 6.
RECURRENT PLACENTA PREVIA PAUL
H.
P~IED,
M.D.,
PHIT"ADJ!;LPHIA, PA., "\ND RICHARD TORPIN, AUG"LS'PA,
M.D.,
(iA.
(From the Department of Obstetrics and Gynecology, Un.i11en;ity of Georgia School of Medicine rmd Hospital)
T
HE incidence of placenta previa varies from 1:150, as reported by the Committee on Prenatal Maternal Care of the White House Conference, to 1:200 to 1:1,500 as noted by various authors. 1 However, a review of the literature reveals that only nine cases of recurrent placenta previa in successive pregnancies, 3-7 and one in nonsuccessive pregnancies8 have been reported. l'lore cases of this type have un.doubterlly been observed but not reported, for De Lee1 and Rivett/ in discussing rt1current placenta previa, speak of a greater than reported :frequency. A much larger number of cases in successive pregnancies should be expected in considering the incidence and the predisposing factors of placenta previa. However, this low incidence is apparently du1,3 to a failure to report cases and to a few principles of prophylaxis, which may also be of avail in preventing the abnormality. I desire to present another case of recurrent placenta previa in successive pregnancies.
Case Report Mrs. A. C., aged 19, in the thirty-second week of her first pregnancy, entered the University Hospital at 9 :45 P.r.r. on lVIay 7, 1941, because of sudden, painless vaginal bleeding. The estimated loss of blood was 300 c.c. The prenatal period, as observed in our clinic, was within normal limits. Examination revealed a white female, who had moderate vaginal bleeding and who was not in labor. Her blood pressure was 130/80 and the pulse rate was 100. The uterus had a one-plus tone, was slightly tender in the lower segment area, and measured 28 crri. above the symphysis pubis. A single fetus lay in the L.O.P. position \\