Volume Number
Communications
124 6
Fig. 3. Pregnancy in left rudimentary uterine cornu elevated by sponge
horn stick.
at laparotomy
up by the left tube. If the suction procedure done elsewhere on December 5 did remove gestational products and not just decidua, two ova must have been fertilized simultaneously, one fertilized ovum subsequently implanting in each horn of the double uterus. The pregnancy in the blind horn had a considerable danger potential.’ Fortunately, the midtrimester abortion was induced with prostaglandin, permitting discontinuation of induction when no progress was observed. Perseverance with prostaglandin administration or intra-amniotic injection of hypertonic saline could presumably have led to rupture of the horn. The same could have happened if the pregnancy had been allowed to continue.2 REFERENCES
1. Rolen, Obstet. 2. Latto,
A. C., Choquette, A. J., and Semmens, J. P.: Gynecol. 27: 6, 807, 1966. D., and Norman, R.: Br. Med. J. 2: 926, 1950.
Fatal maternal disseminated coccidioidomycosis in a nonendemic area WILLIAM
S.
FREDERICK ESTHER Department University
L.
FLEURY,
CHEATLE,
M.D.
M.D. M.D.
of Obstetrics and Gynecology, School of Medicine, Springfield,
APPROXIMATELY
in pregnancy
VANBERGEN,
J.
have
65 patients with been reported.
Southern Illinois
Illinois
coccidioidomycosis Of these, 37
cases
(supported
by the operator’s
hands).
in brief
661
Right
were disseminated, and 29 patients died. All disseminated cases were from the endemic areas.‘, 4 The following report is presented to reinforce the need for an awareness of disease states outside one’s own locale, especially in view of our highly mobile population. The patient was a gravida 3, para 1,27-year-old, Caucasian woman. She had briefly visited Phoenix, Arizona, at 18 weeks’ gestation. The pregnancy was uncomplicated until 27 weeks’ gestation when she complained of “blurred vision” and a “headache.” Two weeks later, she described a “very throbbing headache,” a “halo effect” in the left eye, “hot flashes,” and symptoms of an upper respiratory tract infection with sinusitis and a dry cough. Treatment with penicillin G, Tuss-Ornade,* and Tylenol with codeine had no effect. One week later, she was admitted to the hospital with a temperature of 103.8” F and expressive aphasia. The working diagnosis was a brain abscess. Chest x-ray, skull films, and brain scan were negative except for “clouding” of the left frontal sinus. High doses of penicillin and chloramphenicol were used without apparent effect. Temperature fluctuated widely. When afebrile, the sensorium and speech were clear; when febrile, she was confused and semiconscious. On the fifth hospital day (32 weeks’ gestation), she developed labored respiration, and chest x-ray showed striking changes from five days earlier, consisting of disseminated, interstitial, bilateral infiltrates with a fine nodular pattern. The patient’s liver and spleen had enlarged since the admission examination. Spontaneous labor commenced on the seventh hospital day, and she was delivered of a 4 pound 2 ounce vigorous female infant. The placenta is shown in Figs. 1 and 2. A frozen section of a placental infarct showed the endosporulating spherules of coccidioidomycosis. The placenta weighted 359 grams. A number of tan, irregularly shaped infarcts were scattered throughout the soft spongy lacerated tissue. A section taken through the edge of
662
Communications
in brief
Fig.
1. Cut
section
Fig. 2. Photomicrograph characteristic spherule
of placenta
filled
showing
the
of placenta showing with endospores.
one of the infarcts showed necrosis and acute inflammation and the presence of spherules of coccidioidomycosis. The placental tissue between these areas was not involved. Complement-fixation titers drawn on the third postadmission day for coccidioidomycosis were 1: 128. Cultures of maternal blood, sputum, spinal fluid, and vaginal secretions were all positive for coccidioidomycosis, but the patient died before these results were available. Coccidioidomycosis is caused by cidioides imitk It is indigenous in only
the a few
fungus areas
Cotin the
New World, chiefly in the arid areas of the Southwestern United States, adjacent parts of Mexico, and the San Joaquin Valley of California. The arthrospores, which exist in the soil, are blown by the wind and infect the majority of those living in these areas. Symptoms appear in about 40 per cent of those infected; the others are identified only by skin testing. Disseminated coccidioidomycosis is usually localized to the lung and simulates any common upper respiratory infection which clears with symptomatic treatment.
numerous
poorly
coccidioidomycosis.
defined
The
areas
arrow
of necrosis.
indicates
the
Dissemination occurs in less than 0.2 percent of those infected. In tissue, the organism appears as thickwalled spherules, 30 to 60 p in diameter, containing numerous rounded endospores. The tissue shows necrosis with infiltration by polymorphonuclear leukocytes; peripheral to this, in older lesions, is a granulomatous-type reaction. The tissue form of the fungus is not infectious: therefore, patients need not be placed in isolation. In disseminated cases, the complement-fixations test, precipitation test, or other immunologic tests should be positive. Diagnosis depends on demonstration of the fungus in sputum, cerebrospinal fluid, or tissue or a demonstrated rise in titer of serologic tests. Skin tests are frequently negative in disseminated disease. Eosinophilia of 5 to 8 per cent may be seen in peripheral blood. The present case illustrates several facets of the disease in pregnancy: 1. Patients who develop primary coccidioidomycosis when pregnant are at extremely high risk. If un-
Volume Number
Communications
124 6
treated, approximately 9.0 per cent die.3 Up to 50 per cent may be saved by early adequate treatment with amphotericin B. 2. Maternal genital tract involvement is rare.6 Our patient apparently had coccidioidomycosis in the vaginal secretions. 3. Placental involvement has been reported but is rare.‘, ‘3 6 The diagnosis in our patient was from placental involvement. 4. There have been only two cases of possible fetal involvement with the disease.‘, 4 Our patient also had no evidence of fetal coccidioidomycosis. 5. The 50 per cent reported fetal loss is from prematurity.4 The infant in the reported case died from hyaline membrane disease secondary to prematurity. 6. Passive transfer of maternal complement-fixing antibodies were shown in the cord blood.5 REFERENCES
1. Cohen, R.: Placental coccidiodes, Arch. Pediatr. 68: 59, 1951. 2. Harris, R. E.: Coccidioidomycosis complicating pregnancy, Obstet. Gvnecol. 28: 401. 1966. 3. Monif, d. R. G.: Infectious Diseases in Obstetrics and Gynecology, New York, 1974, Harper ti Row Publishers, pp. 267-27 1. 4. Smale, L. E., and Waechter, K. G.: Dissemination of coccidioidomycosis in pregnancy, AM. J. OBSTET. GYNECOL. 107: 356, 1970. 5. Smith, C. E., Saito, M. T., and Simms, S. A.: Patterns of 39,500 serologic tests in coccidioidomyocsis, J. A. M. A. 160: 546, 1956. 6. Vaughan, J. E., and Ramirez, H.: Coccidioidomycosis as a complication of pregnancy, Calif. Med. 74: 12 1, 1951.
Primary adenocarcinoma of the vermiform appendix in a gynecologic patient ANTHONY
P.
WILLIAM
M.
FICHERA, PETTY,
ROBERT
C.
PARK,
ROBERT
W.
MUIR,
MAJOR MAJOR COLONEL
(MC)
COLONEL
Departments of Obstetrics and Gynecology Surgery, The Walter Reed Army Medical D. C.
(MC) (MC) (MC)
USA USA
USA USA
and General Center, Washington,
PRIMARY adenocarcinoma of the appendix is a rare clinical entity that is virtually never diagnosed preoperatively. Hesketh’ showed that in 13 per cent of the female patients with this neoplasm the diagnosis
The opinions or assertions contained herein are the private views of the authors and are not to be construed as official or reflecting the views of the Department of the Army or the Department of Defense. Reprint Accomac
requests: Anthony St., Springfield,
P. Fichera, Major Virginia 22150.
(MC)
USA,
5817
in brief
663
was made because an incidental appendectomy accompanied a primary gynecologic procedure. During the past decade at The Walter Reed Army Medical Center 67 per cent of all primary appendiceal neoplasms in females were discovered on the gynecology service. Since the gynecologist is likely to encounter most appendiceal neoplasms in females, this case report and review are submitted. J. P., a 26-year-old white woman, para l-O-0-1, was admitted for evaluation of abdominal pain and ascites. The onset of symptoms occurred 3 weeks prior to admission. The past medical history and the review of systems were essentially noncontributory. Physical examination revealed a 5 foot 8 inch, 150 pound woman with stable vital signs. The examination was unremarkable except for abdominal and pelvic findings. The abdomen was markedly distended with ascitic fluid. On pelvic examination bilateral large adnexal masses and multiple cul-de-sac nodularities were found. The hemogram, urinalysis, and serology were within normal limits. The blood chemistries were unremarkable except for lactic dehydrogenase of 515 mu. per milliliter, total protein of 4.7 grams, and albumin of 2.7 grams. The chest x-ray, excretory urogram, upper gastrointestinal series, and barium enema were negative. The paracentesis fluid yielded class V cytology. On the fourth hospital day an exploratory coeliotomy was performed and 4,500 C.C. or ascitic fluid were removed. Both ovaries were involved with tumor and each measured 12 cm. in diameter. Multiple excrescenses existed on the peritoneum, small and large intestines, and their mesenteries. Metastases to four para-aortic and two left common iliac nodes were found. A frozen section analysis of the ovary was suggestive of a Krukenberg tumor. Evaluation of the intestinal tract revealed a neoplasm that encompassed the distal half of the appendix. A total abdominal hysterectomy with bilateral salpingooophorectomy, partial omentectomy, and appendectomy were performed. The postoperative course was uneventful. The permanent tissue slides were diagnostic of a primary colloid adenocarcinoma of the appendix. Management was discussed at a combined oncology-radiotherapy conference. Chemotherapy with 5-fluorouracil was selected. Despite this therapy rapid tumor growth occurred. Death occurred 13 months after the diagnosis was made. Primary appendiceal neoplasms account for about percent of all gastrointestinal tumors.’ Carcinoid, in contradistinction to adenocarcinoma, is the most common and least malignant tumor. On cross-section, it has a golden-yellow appearance.’ A malignant mucocoele is usually characterized by a dilated appendix containing gelatinous material or by a ruptured appendix and pseudomyxoma peritonei.’ This tumor, unlike adenocarcinoma, rarely spreads by the blood stream or by the lymphatics. Adenocarcinema may be a slow growing in situ lesion2 or it may have a rapid transcoelomic spread with peritoneal and ovarian deposits.’ About 66 per cent of all cases present as some form of appendicitis or appendiceal abscess.t Carcinoid, mucocoele, and adenocarcinoma in situ 0.5
Communications
124 6
Fig. 3. Pregnancy in left rudimentary uterine cornu elevated by sponge
horn stick.
at laparotomy
up by the left tube. If the suction procedure done elsewhere on December 5 did remove gestational products and not just decidua, two ova must have been fertilized simultaneously, one fertilized ovum subsequently implanting in each horn of the double uterus. The pregnancy in the blind horn had a considerable danger potential.’ Fortunately, the midtrimester abortion was induced with prostaglandin, permitting discontinuation of induction when no progress was observed. Perseverance with prostaglandin administration or intra-amniotic injection of hypertonic saline could presumably have led to rupture of the horn. The same could have happened if the pregnancy had been allowed to continue.2 REFERENCES
1. Rolen, Obstet. 2. Latto,
A. C., Choquette, A. J., and Semmens, J. P.: Gynecol. 27: 6, 807, 1966. D., and Norman, R.: Br. Med. J. 2: 926, 1950.
Fatal maternal disseminated coccidioidomycosis in a nonendemic area WILLIAM
S.
FREDERICK ESTHER Department University
L.
FLEURY,
CHEATLE,
M.D.
M.D. M.D.
of Obstetrics and Gynecology, School of Medicine, Springfield,
APPROXIMATELY
in pregnancy
VANBERGEN,
J.
have
65 patients with been reported.
Southern Illinois
Illinois
coccidioidomycosis Of these, 37
cases
(supported
by the operator’s
hands).
in brief
661
Right
were disseminated, and 29 patients died. All disseminated cases were from the endemic areas.‘, 4 The following report is presented to reinforce the need for an awareness of disease states outside one’s own locale, especially in view of our highly mobile population. The patient was a gravida 3, para 1,27-year-old, Caucasian woman. She had briefly visited Phoenix, Arizona, at 18 weeks’ gestation. The pregnancy was uncomplicated until 27 weeks’ gestation when she complained of “blurred vision” and a “headache.” Two weeks later, she described a “very throbbing headache,” a “halo effect” in the left eye, “hot flashes,” and symptoms of an upper respiratory tract infection with sinusitis and a dry cough. Treatment with penicillin G, Tuss-Ornade,* and Tylenol with codeine had no effect. One week later, she was admitted to the hospital with a temperature of 103.8” F and expressive aphasia. The working diagnosis was a brain abscess. Chest x-ray, skull films, and brain scan were negative except for “clouding” of the left frontal sinus. High doses of penicillin and chloramphenicol were used without apparent effect. Temperature fluctuated widely. When afebrile, the sensorium and speech were clear; when febrile, she was confused and semiconscious. On the fifth hospital day (32 weeks’ gestation), she developed labored respiration, and chest x-ray showed striking changes from five days earlier, consisting of disseminated, interstitial, bilateral infiltrates with a fine nodular pattern. The patient’s liver and spleen had enlarged since the admission examination. Spontaneous labor commenced on the seventh hospital day, and she was delivered of a 4 pound 2 ounce vigorous female infant. The placenta is shown in Figs. 1 and 2. A frozen section of a placental infarct showed the endosporulating spherules of coccidioidomycosis. The placenta weighted 359 grams. A number of tan, irregularly shaped infarcts were scattered throughout the soft spongy lacerated tissue. A section taken through the edge of
662
Communications
in brief
Fig.
1. Cut
section
Fig. 2. Photomicrograph characteristic spherule
of placenta
filled
showing
the
of placenta showing with endospores.
one of the infarcts showed necrosis and acute inflammation and the presence of spherules of coccidioidomycosis. The placental tissue between these areas was not involved. Complement-fixation titers drawn on the third postadmission day for coccidioidomycosis were 1: 128. Cultures of maternal blood, sputum, spinal fluid, and vaginal secretions were all positive for coccidioidomycosis, but the patient died before these results were available. Coccidioidomycosis is caused by cidioides imitk It is indigenous in only
the a few
fungus areas
Cotin the
New World, chiefly in the arid areas of the Southwestern United States, adjacent parts of Mexico, and the San Joaquin Valley of California. The arthrospores, which exist in the soil, are blown by the wind and infect the majority of those living in these areas. Symptoms appear in about 40 per cent of those infected; the others are identified only by skin testing. Disseminated coccidioidomycosis is usually localized to the lung and simulates any common upper respiratory infection which clears with symptomatic treatment.
numerous
poorly
coccidioidomycosis.
defined
The
areas
arrow
of necrosis.
indicates
the
Dissemination occurs in less than 0.2 percent of those infected. In tissue, the organism appears as thickwalled spherules, 30 to 60 p in diameter, containing numerous rounded endospores. The tissue shows necrosis with infiltration by polymorphonuclear leukocytes; peripheral to this, in older lesions, is a granulomatous-type reaction. The tissue form of the fungus is not infectious: therefore, patients need not be placed in isolation. In disseminated cases, the complement-fixations test, precipitation test, or other immunologic tests should be positive. Diagnosis depends on demonstration of the fungus in sputum, cerebrospinal fluid, or tissue or a demonstrated rise in titer of serologic tests. Skin tests are frequently negative in disseminated disease. Eosinophilia of 5 to 8 per cent may be seen in peripheral blood. The present case illustrates several facets of the disease in pregnancy: 1. Patients who develop primary coccidioidomycosis when pregnant are at extremely high risk. If un-
Volume Number
Communications
124 6
treated, approximately 9.0 per cent die.3 Up to 50 per cent may be saved by early adequate treatment with amphotericin B. 2. Maternal genital tract involvement is rare.6 Our patient apparently had coccidioidomycosis in the vaginal secretions. 3. Placental involvement has been reported but is rare.‘, ‘3 6 The diagnosis in our patient was from placental involvement. 4. There have been only two cases of possible fetal involvement with the disease.‘, 4 Our patient also had no evidence of fetal coccidioidomycosis. 5. The 50 per cent reported fetal loss is from prematurity.4 The infant in the reported case died from hyaline membrane disease secondary to prematurity. 6. Passive transfer of maternal complement-fixing antibodies were shown in the cord blood.5 REFERENCES
1. Cohen, R.: Placental coccidiodes, Arch. Pediatr. 68: 59, 1951. 2. Harris, R. E.: Coccidioidomycosis complicating pregnancy, Obstet. Gvnecol. 28: 401. 1966. 3. Monif, d. R. G.: Infectious Diseases in Obstetrics and Gynecology, New York, 1974, Harper ti Row Publishers, pp. 267-27 1. 4. Smale, L. E., and Waechter, K. G.: Dissemination of coccidioidomycosis in pregnancy, AM. J. OBSTET. GYNECOL. 107: 356, 1970. 5. Smith, C. E., Saito, M. T., and Simms, S. A.: Patterns of 39,500 serologic tests in coccidioidomyocsis, J. A. M. A. 160: 546, 1956. 6. Vaughan, J. E., and Ramirez, H.: Coccidioidomycosis as a complication of pregnancy, Calif. Med. 74: 12 1, 1951.
Primary adenocarcinoma of the vermiform appendix in a gynecologic patient ANTHONY
P.
WILLIAM
M.
FICHERA, PETTY,
ROBERT
C.
PARK,
ROBERT
W.
MUIR,
MAJOR MAJOR COLONEL
(MC)
COLONEL
Departments of Obstetrics and Gynecology Surgery, The Walter Reed Army Medical D. C.
(MC) (MC) (MC)
USA USA
USA USA
and General Center, Washington,
PRIMARY adenocarcinoma of the appendix is a rare clinical entity that is virtually never diagnosed preoperatively. Hesketh’ showed that in 13 per cent of the female patients with this neoplasm the diagnosis
The opinions or assertions contained herein are the private views of the authors and are not to be construed as official or reflecting the views of the Department of the Army or the Department of Defense. Reprint Accomac
requests: Anthony St., Springfield,
P. Fichera, Major Virginia 22150.
(MC)
USA,
5817
in brief
663
was made because an incidental appendectomy accompanied a primary gynecologic procedure. During the past decade at The Walter Reed Army Medical Center 67 per cent of all primary appendiceal neoplasms in females were discovered on the gynecology service. Since the gynecologist is likely to encounter most appendiceal neoplasms in females, this case report and review are submitted. J. P., a 26-year-old white woman, para l-O-0-1, was admitted for evaluation of abdominal pain and ascites. The onset of symptoms occurred 3 weeks prior to admission. The past medical history and the review of systems were essentially noncontributory. Physical examination revealed a 5 foot 8 inch, 150 pound woman with stable vital signs. The examination was unremarkable except for abdominal and pelvic findings. The abdomen was markedly distended with ascitic fluid. On pelvic examination bilateral large adnexal masses and multiple cul-de-sac nodularities were found. The hemogram, urinalysis, and serology were within normal limits. The blood chemistries were unremarkable except for lactic dehydrogenase of 515 mu. per milliliter, total protein of 4.7 grams, and albumin of 2.7 grams. The chest x-ray, excretory urogram, upper gastrointestinal series, and barium enema were negative. The paracentesis fluid yielded class V cytology. On the fourth hospital day an exploratory coeliotomy was performed and 4,500 C.C. or ascitic fluid were removed. Both ovaries were involved with tumor and each measured 12 cm. in diameter. Multiple excrescenses existed on the peritoneum, small and large intestines, and their mesenteries. Metastases to four para-aortic and two left common iliac nodes were found. A frozen section analysis of the ovary was suggestive of a Krukenberg tumor. Evaluation of the intestinal tract revealed a neoplasm that encompassed the distal half of the appendix. A total abdominal hysterectomy with bilateral salpingooophorectomy, partial omentectomy, and appendectomy were performed. The postoperative course was uneventful. The permanent tissue slides were diagnostic of a primary colloid adenocarcinoma of the appendix. Management was discussed at a combined oncology-radiotherapy conference. Chemotherapy with 5-fluorouracil was selected. Despite this therapy rapid tumor growth occurred. Death occurred 13 months after the diagnosis was made. Primary appendiceal neoplasms account for about percent of all gastrointestinal tumors.’ Carcinoid, in contradistinction to adenocarcinoma, is the most common and least malignant tumor. On cross-section, it has a golden-yellow appearance.’ A malignant mucocoele is usually characterized by a dilated appendix containing gelatinous material or by a ruptured appendix and pseudomyxoma peritonei.’ This tumor, unlike adenocarcinoma, rarely spreads by the blood stream or by the lymphatics. Adenocarcinema may be a slow growing in situ lesion2 or it may have a rapid transcoelomic spread with peritoneal and ovarian deposits.’ About 66 per cent of all cases present as some form of appendicitis or appendiceal abscess.t Carcinoid, mucocoele, and adenocarcinoma in situ 0.5
