110 Communications in bfief
Mar l , 1~ 78 Am. J. Obstet. 'Gynecol.
Fig. 1. Paracervical neuron containing herpesvirus-like intranuclear inclusion. (Hematoxylin and eosin. x400.)
associated with ~ he demonstrable presence of virus in affected epithelium. Between acute attacks the virus is no longer demonstrable in the epithelium. The herpesviruses, as part of their general epidermotropism, are neurotropic viruses, and a neural habitat has iong been suspected during periods of latency. In the case ofHSV-1 it has been demonstrated that the virus is present in the trigeminal ganglia in latent periods, 1 and a neural location has also been demonstrated in experimental infection with herpesvirus.2 In the case of herpetic cervicitis a similar association with ganglion cells has been postulated, with the sacral sensory ganglia being the most likely site . The case reported here illustrates not only the rare morphologicall y apparent involvement of ganglion cells but also the appropriately close location of the affected cells, those being in, or at least on, the cervix itself. The patient, a 25-year-old woman, presented with a fungating and foul-smelling cervical mass, histologically diagnosed as a mixed o r adenosquamous carcinoma. Wertheim hysterectomy was carried out, and it was confirmed that the tumor was grossly confined to the cervix. Microscopically , however, both lymphatics and veins showed tumor permeation. Forty-three lymph node s showed no metastases. In the operative specimen were a number of paracervical nerves and their associated ganglia . In a number of these, sharply outlined eosinophilic intranuclear inclusions were present (Fig. 1), usually rendered more conspicuous by the peripheral margination of chromatin and the formation of a clear halo. Positive virologic identification of the inclusions was not made, but their appearance was consistent with that of herpesvirus hominis, probably type 2. The patient did poorly and, despite subsequent radiotherapy and chemotherapy, she died approximately six months following initial diagnosis.
The present case is reported not as definitive evidence of an etiologic role for herpesvirus in the genesis of cervical cancer but as a morphologically demonstrable association between herpesvirus-like inclusions and rapidly disseminating and rapidly fatal carcinoma . The report is intended to stimulate directed search by virologic methods for more conclusive evidence of herpesvirus persistence in surgical and autopsy material from cases of carcinoma of the cervix . REFERENCES l. Bastian , F. 0., Rabson, A. S., Yee, C. L., el al.: Herpesvirus
hominis: isolation from human trigeminal ganglion, Science 178: 306, 1972. 2. Stevens, J. G., and Cook, M. L. : Latent infections induced by herpes simplex viruses, Cancer Res. 33: 1399, I 973.
A unique case of aorticoduodenal fistula following carcinoma of cervix RAJ K. GUPTA, M . D. , F .C .A . P ., F.I.A. C . K. E . ROGERS, M . D . , F.R.C.S .(C. ) Departments of Pathology arul Surgery, The St. Catharin e> General Hospital, St. Catharines, Ontario, Canada , and Department of Pathology, State University of New York at Buffalo, School of Medicine, Buffalo, New York AN uNusuAL AND dramatic cause of massive upper gastrointestinal bleeding is the rupture of an aneurysm
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©
1978 The C. V. Mosby Co .
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ot the abdominai aorta into the iumen of the third portion of duodenum. This is attributed to the fixed position and close proximity of the abdominal aorta to the third part of the duodenum in which these aorticoduodena! fistulas are kno,vn to occur....~\!though in the majority of cases these fistulas are caused by primary aortic disease. occasional instances of their occurrence as a late complication following aortic graft replacement and as a result of anastomotic breakdown following a surgical corrective procedure of occlusive disease of the aorta are also described in the literature. t-o Aorticoduodenal fistulas following penetrating abdominal injuries, tuberculous mesenteric lymphadenitis, and peptic ulcers are rare but do occur, resulting in sudden and massive gastrointestinal bleeding. 7• R In 1966, one of us (R. K. G.) reported the first case of an aorticoduodenal fistula following an invasive carcinoma of pancreas. 9 The present communication deals with an example of an aorticoduodenal fistula caused by invasive carcinoma of cervix. In this case cytohistologic studies were done with the use of autopsy material. To our knowledge this is the first case to be described in the literature. In 1962, advanced carcinoma of the cervix was diagnosed in a 69-year-old Caucasian woman. Her history was considered unreliable. In 1962, gynecologic examination revealed a deep ulcer crater in the cervix and extensive involvement of the cervix by the tumor, which extended bilaterally into the parametrium. A biopsy confirmed the diagnosis of invasive epidermoid carcinoma. Since the tumor was considered inoperable, two applications of radiation therapy (7,200 mg. hours) by a modified Fletcher technique were given to the uterine and vaginal cavity. This was followed by an additional 4,000 rads over four weeks to the pelvic wall. She disappeared until 1965, and nothing was known in the interim period regarding tlle state of her health.. In 1965, she ,,;as admitted to another hospital after sustaining injury to the back. Roentgenograms showed degenerative changes in the second and third lumbar discs. During this admission she complained of occasional symptoms of seizures and of pain in the shoulders and both lower limbs. No evidence of any recurrent or metastatic tumor was found. A skeletal roentgenographic survey was negative. Throughout the short hospital course she remained depressed and incoherent. She was cared for by a psychiatrist because of a continuous state of depression and was treated svmptomatically between 1965 and 1973, without any change in personality or symptoms. She refused to be examined in detail for a checkup or other medical consultation. From time to time she was lost to follow-up. She was finally admitted to the hospital in the latter part of 1974, with bilateral ankle edema, generalized arthritis, nervousness, anxiety, and depression. She complained of intermittent vague pain in the abdomen and epigastrium. Extensive laboratory investigations and roentgenograms were negative. She would not cooperate for a gynecologic examination. An intravenous pyelogram was negative. One morning, she suddenly complained of severe pain in the abdomen and chest, radiating to the !umbar spine. This was associated with vomiting of massive, bloody gastric contents. This resulted in a precipitate drop of blood pressure. The hemoglobin fell from 11.5 to 8.16 Gm. per 100 mi. Packed red cell
Communicatio'ls in brief 111
transfusions were given. She showed some clinical improvement. Subsequently she developed a temporary loss of consciousness from which she made an uneventful recovery. An electrocardiogram showed flattened T waves and an inversion in a VL leads, suggesting anterior myocardial ischemia. An electroencephalogram and brain scans were negative. Examination after oral barium administration was suggestive of a prepyloric ulcer. In the next few days her fiuid intake became poor. She remained dehydrated. Intravenous fluid therapy and Levin tube feedings were given. Two days before death she became hypotensive with shortness ot breath. Films of the chest taken with a portable x-ray machine showed evidence of pneumonitis. Sputum culture grew ampicillinresistant Klebsiella organisms. Tracheobronchial suction was done with some relief in respiratory distress. The low hemoglobin persisted. Black tarry stools were noted. On the morning prior to death, she again developed sudden, massive hematemesis, rectal bleeding, a precipitate drop in blood pressure, and low hemoglobin. Rapid transfusions were given. Emergency esophagogastwscopy-d uodenoscopy was done. The esophagus, stomach, and duodenum were fuU of bright blood. An ulcer could not be visualized. The source of bleeding was suspected to be a duodenal uket. Later in the afternoon another episode of severe bleeding occurred. She passed massive biood and dots through the re•.!Urn. Despite all measures she suddenly died in shock. At autopsy the entire gastrointestinal tract wa~ fuH of recent blood and clots. In the prepyloric region a (l ..'i em. chronic ulcer was found. However, there was no bleeding vessel in the base of the ulcer. The liver was replaced (20 per cent) by metastatic tumor. The meseteric and aortic lymph nodes were matted, enlarged, and replaced by metastatic rumor. In the third part of the duodenum, a 2 .."> by 2 em., ragged, firm, ulcerated lesion with necrotic debris was noted. The entire tissue in the surrounding area was hrm, necrotic, and adherent up to the abdominal aorta. This area of duodenum communicated with the abdominal aorta bv a fistulous tract. The abdominal aorta was opened and showed a throughand-through involvement of the entire wall with .m ulcerated 1.5 em. opening on the intimal surface with firm margins. A probe was passed from this ulcerated area in a reverse direction toward the third portion of duodenum, which was entered without difficulty. The communicating hstulous tract behveen the third part of the duodenun1 and 1he abdomina! aorta was opened and was found to be full of recent blood and clots throughout its entire length. The surrounding tissue lining the fistulous tract was friable, firm, and necrotic with gross features suggestive of metastatic tumor. The re't of the abdominal cavity showed tumor tissue binding the bowel loops and mesentery. which extended down to the pelvic cavity. The uterine fundus and both tubes and ovaries appeared to be merged and replaced by tumor. The cervix could not be identified and was totally replaced by a firm. hhrotic tumor. Several histologic sections were taken from these organs and from the tissue at the third part of the duodenum, the abdominal aorta, and the area of aorticoduodenal fi~tula. In addition, several cytologic imprints obtained at postmortem examination were immediately made from all the tissues, fixed, and stained with Papanicolaou stain. Examination of organs in the chest and brain was negative for metastatic disease. Papanico!aou=stained cytologic imprints fro1n :be t..~ird part of the duodenum, the abdominal aorta, and the aorticoduodenal fistula, including other sites of metastatic tumor in the abdominal and pelvic cavitv, showed cells ha1·ing fea-
112 Communications in brief
Mav 1, 1978
Am. J Obstet .. Gynecol.
Fig. I. Section from abdominal aorta, showing features of metastatic epidermoid carcinoma from cervix invading wall of aorta. In some fields central tumor necrosis in individual group of tumor cells gives an impression of a pseudogianduiar appearance, a finding which is not uncommon in invasive epidermoid carcinomas. (Hematoxylin and eosin. x 820.)
tures consistent with invasive epidermoid carcinoma. Hematoxylin and eosin-stained sections from all of these tissues showed features of invasive epidermoid carcinoma (Fig. !).
In the case under discussion it was our impression that the aorticoduodenal fistula was apparently produced by direct invasion of the third portion of the duodenum by metastatic carcinoma from cervix. A detailed gynecologic follow-up from 1962 until the final admission was not available and resulted in considerable handicap regarding exact diagnosis, clinical management, and any previous evidence of recurrence or metastasis by the tumor. However, it is conceivable to assume that radiation therapy in 1962 may have afforded some control of the disease since the patient lived for about 12 years following the initial diagnosis of advanced primary carcinoma of cervix. The cause of massive gastrointestinal bleeding remained an enigma at the time of final admission and was ascertained only at postmorten examination, which revealed an aorticoduodenal fistula resulting from invasion of the abdominal aorta by carcinoma of the cervix. Also no significant atherosclerosis of the abdominal aorta was found despite the age of the patient; thus, atherosclerosis did not cause vveakening of the \vall
and, consequently, the aorticoduodenal fistula. Other mechanisms, such as thrombosis of the vasa vasorum caused by a tumor, which have been described as being important factors in the causation of aortic perforation, were not seen in our case. 10 It is of interest to note that to some extent the clinical events of massive gastrointestinal bleeding in the patient followed a pattern somewhat similar to that in cases of aorticoduodenal fistula of other causes." However, one difference was the fact that, instead of several separate periodic episodes of bleeding over a longer period of time, this patient developed a sudden unexpected episode of bieeding that occurred oniy a few days prior to death. Also the interval between the first episode of massive bleeding and the terminal episode of life-threatening bleeding was very short. Therefore, there was neither time for a clinical diagnosis and investigation nor time to even consider any treatment. The exact cause of intermittent bleeding in most of these patients following the occurrence of aorticoduodenal fistula is not known. In our case it appeared that the first episode of massive gastrointestinal bleeding followed by a brief stoppage before the death of patient might have been due to a temporary closure of
Volume 131 Number I
the fistulous tract by the tamponade as a result of tense clot in the duodenum. However, this mechanism seemed to last for only a short period since the terminal episode of gastrointestinal bleeding, just prior to death, was very massive and uncontrollable. In similar fistulas of other causes, periodic episodes of gastrointestinal bleeding are almost always known to afford ample time for a diagnosis and effective surgical correction. REFERENCES I. Reckless,]. P. D., McColl, 1.. and Taylor, G. W.: Aorta-
2.
~-
4.
5.
6. 7.
8.
9. 10. 11.
enteric fistulae. Uncommon complication of abdominal aortic aneurysms, Br.]. Surg. 59: 458, 1972. \Vyatt, G. ~v1., Rauchway, ~f. I., ru1d Spitz, H. B.: Roentgen findings in aorto-enteric fistula, Am. J. Radio!. 126: 714, 1976. Bagnuolo, W. G., and Bennett, H. D.: Non-traumatic aortic perforations into gastrointestinal tract, Am. Heart J. 40: 7114. 1950. Garrett, H. E .. Beall, A. C., Jr., Jordan, G. L., Jr., and DeBakey, M. E.: Surgical considerations of massive gastrointestinal tract hemorrhage caused by aortoduodenal fistula, Am. J. Surg. 105: 6, 1963. Levy, M. ]., Todd, D. D., Iillihei, C. W., and Varco, R. L.: Aorticointestinal fistulas following surgery of the aorta, Surg. Gynecol. Obstet. 120: 992, 1965. Phillips, R. B., and Jarrell, S. E.: Arteriosclerotic aortic aneurysms with rupture into the duodenum, Ann. Surg. 159: 375, 1964. Frosch, H. L., and Horowitz, ,V.: Rupture of abdomina! aorta into duodenum through sinus tract created by tuberculous mesenteric lymphadenitis, Ann. Intern. Med. 21: 481, 1944. Dash. P. M.: Perforation of duodenal ulcer into aorta, Br. Med. [. 1: 570, 1940. Ghaphery, A. D., Gupta, R. K., and Currie, R. D.: Carcinoma of head of pancreas with aortoduodenal fistula, Am.]. Surg. lll: 580, 1966. Postoloff, A. V., and Cannon, W. M.: Genesis of aortic perforation secondary to carcinoma of esophagus, Arch. Pathol. 41: 533, 1946. Law, S. W., Garrett, H. E., and DeBakey, M. E.: Gastrointestinal hemorrhage due to rupture of abdominal aortic aneurysm, Gastroenterology 43: 680, 1962.
Ultrasound diagnosis of occipitothoracic meningocele at 22 weeks' gestation RUDY E. SABBAGHA. M.D. RICHARD DEPP. M.D. DENISE GRASSE, R.D.M.S. INGRID KIPPER, R.T. Drpartmrnt of Obstetrics and Gynecology, Northwestern Uni<•n,itv-Prentire Women·., Hospital, Chicago, Illinois ARE developing an increasing OBSTETRICIANS awareness of the benefits of ultrasound examination early in pregnancy. particularly in the presence of
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Communications in brief
113
high-risk criteria such as: (1) a history of poor obstetric outcome, (2) vaginal bleeding, (:\) questionable date~. and (4) a fundal height smailer or larger t al heart tones were audible. The ultrasound examination revealed a large sonolucent (fluid-filled) mass extending from the occipital to the upper thoracic area (Fig. 1). At the base of the mass, a solid protuberance was noted (Fig. 1). The mass originated below the biparietal plane; hence, a good measurement of the biparietal diameter (BPD) could be obtained. The BPD was equivalent to a mean gestational age of 22 weeks (20 weeks bv patient's menstrual dates). Real-time ultrasound examination clearly
showed the mass to move simultaneously with
fet;~l
cephalic
motion (Fig. 2). A sonar diagnosis of occipitothoracic meningocele or encephalocele was made. No other gross fetal anomaly was noted by ultrasound imaging. The placenta was located along the right anterolateral wall of the uterus. The echogram outlining the fetal congenital anomaly was shown to the couple, and fetal outcome was discussed; they elected to have the pregnancy terminated. Amniocentesis was performed in the left lower abdominal area and a small amount of amniotic fluid was removed. Then 200 ml. of hypertonic saline was infused imo the amniotic cavity over a period of 20 minutes. The patient went into labor 44 hours after hypertonic saline amnioinfusion, and an abnormal male tetus weighing 520 grams (Fig. 3) was delivered (54.5 hours after injection). The postdelivery cour•w wa~ uneventful. The pathology report confirmed the presence of an occipitothoracic meningocele, the base of which was lined \vith fibrous tissue. There was no neural tissue in the 'ac.
Ultrasonic antenatal screening is not presenrly performed on a routine basis in the United States or in any other part of the world. Obviously. one of the benefits of mass sonar screening is the diagnosis of m~or congenital anomalies, particularly neural tube defects. The early detection of these anomalies will give the pregnant woman and her partner the option of pregnancy termination prior to fetal viability. Nevertheless, mass screening by ultrasound is not yet feasible because of the cost-benefit ratio, on the one hand, and iack of a sufficient number of trained personnel, on the other. It *Flint Laboratories, Deerfield. Illinois.