A Benign Cystic Teratoma with Gastrointestinal Tract Development

Transcription

1 236 YAMASHINA AND FLINNER A.J.C.P. February 1985 tern. 10 We think our tumor also represents an example 3. Brown NK, Smith MP: Neoplastic diathesis of patients with carcinoid: Report of a case and four other neoplasms. Cancer of a collision variety of two independent tumors in a 1973; 32: unique one-upon-another pattern. 4. Cheijfec G, Gould VE: Malignant gastric neuroendocrinoma: Gastric composite tumors appear to have a better ultrastructural and biochemical characterization of their secretory prognosis than ordinary gastric carcinoma. 912 activity. Hum Pathol 1977; 8: The prognosis for collision tumors of double primaries is not Clin Pathol 1963; 16: Gibbs NM: The histogenesis of carcinoid tumors of the rectum. J known, but from the few available cases in the GI tract 6. Klein HZ: Mucous carcinoid tumor of the vermiform appendix. Cancer 1974; 33: it appears that the adenocarcinoma impacts more heavily 7. Kubo T, Watanabe H: Neoplastic argentaffin cells in gastric and on prognosis 3,5,8,10 This is most likely, due to the intestinal carcinomas. Cancer 1970; 27: biologic behavior of GI carcinoids, which, in spite of 8. Lattes R, Grossi C: Carcinoid tumors of the stomach. Cancer 1956;9: their ability to produce early lymph node metastasis and 9. Murayama T, Imai T, Kikuchi M: Solid carcinomas of the infiltrative growth pattern, often seem to exhibit only stomach: A combined histochemical, light and electron microscopic study. Cancer 1983; 51: very low clinical malignancy Parks TG: Malignant carcinoid and adenocarcinoma of the stomach. Acknowledgment. The authors thank Drs. Thomas V. Colby and Ernst Eichwald for their review of this manuscript. 11 References 1. Azzopardi JC, Pollack DJ: Argentaffin and argylophil cells in gastric carcinomas. J Pathol Bac 1963; 86: Bates HR, Belter LF: Composite carcinoid tumor (argentaffinomaadenocarcinoma) of the colon. Dis Colon Rectum 1967; 10: Br J Surg 1970;57: Peison B, Benisch B: Simultaneous occurrence of malignant carcinoid and adenocarcinoma of stomach. Arch Pathol 1983; 107: Roger LW, Murphy RC: Gastric carcinoids and carcinomas: Morphologic correlates of survival. Am J Surg Pathol 1979; 3: Soga J, Tazawa K, Aizawa O, Wada K, Tuto T: Argentaffin cell adenocarcinoma: An atypical carcinoid? Cancer 1971; 28: A Benign Cystic Teratoma with Gastrointestinal Tract Development BRENT WOODFIELD, DAVID ALLAN KATZ, M.D., CATHY JO CANTRELL, M.D., AND PATRICK J. BOGARD, M.D. The clinical and histologic features of a benign cystic teratoma with histologic evidence of almost full gastrointestinal tract development are discussed. The literature has previously described only bowel epithelium, segments of bowel, appendix, and esophagus separately. This is the first report of almost complete development of the gastrointestinal tract in a benign cystic teratoma. In this teratoma, the entire gastrointestinal tract from esophagus to colon is represented histologically. (Key words: Ovarian teratoma; Gastrointestinal tract; Homunculus) Am J Clin Pathol 1985; 83: BENIGN CYSTIC TERATOMAS have generated much interest due to their unusual structure and variety of "organoid" development. The fetus-like homunculus with its rudimentary development of skeleton, limbs, external genitalia, and head-iike structures is an example. Received January 19, 1984; received revised manuscript and accepted for publication March 26, Address reprint requests to Dr. Katz: Creighton University/Saint Joseph Hospital, 601 North 30th Street, Omaha, Nebraska Departments of Pathology and Obstetrics and Gynecology, Creighton University/St. Joseph Hospital, Omaha, Nebraska Other examples include rudimentary lung, maxillary bone with teeth, brain, thyroid, mammary, adrenal, and salivary gland tissue. The literature also makes reference to gastrointestinal development in the form of bowel segments and occasional reference to development of structures such as esophagus and appendix. No report has been made of extensive development of the gastrointestinal tract in one teratoma. The following is a case report of a benign cystic teratoma with development of esophagus, stomach, duodenum, small intestine, and colon. Report of a Case Patient 1, a 32-year-old para 1001 female, whose last menstrual period was November 23, 1982, presented to our hospital emergency

2 Vol. 83 No. 2 CASE REPORTS 237 \ ' V Y FIG. 1 (upper). Section of esophagus with overlying mature squamous epithelium. (Hematoxylin and eosin, X200). FIG. 2 (lower). Section of stomach with gastric glands. (Hematoxylin and eosin, X200).

3 «<: '-.,^v FIG. 3 (upper). Section of duodenum with Brunner's glands in the submucosa (Hematoxylin and eosin, X40). FIG. 4 (lower). Section of small bowel with prominent villi. (Hematoxylin and eosin, X100). l \, M

4 Vol. 83 No. 2 CASE REPORTS 239 FlG. 5. Cross-section of colon with glands lined by mucus secreting columnar epithelium. (Hematoxylin and eosin, X20). room on November 30 with dull right lower quadrant pain, bloating, and vague abdominal complaints. A large right adnexal mass was found on pelvic examination. On admission to our hospital on December 2, pelvic ultrasonography demonstrated a midline cystic 8.1 X 8.3 cm mass. Abdominal flat plate and barium enema demonstrated calcification over the left sacral area and extrinsic pressure on the sigmoid colon, respectively. Chest x-ray and complete blood count on admission were normal. The admission physical examination was otherwise unremarkable. Exploratory laparotomy was performed on December 12, 1982 with removal of a cystic right ovary and fallopian tube. The ovarian cyst measured 9 X 6 X 5 cm and was opened in the operating room. It was found to be consistent with a cystic teratoma. The uterus and left adnexa were found to be normal. The postoperative period was uneventful, with discharge on the fifth postoperative day. Pathology The specimen consisted of an ovary with attached fallopian tube. The ovary measured 9X6X5 cm and upon cutting into the ovary there was a large cystic structure that measured 8 X 5 X 3.5 cm. Within the cyst the usual elements of a dermoid cyst were noted, including portions of cartilaginous tissue, a tooth, hair, and yellowish necrotic cheese-like material. The wall of the cyst was thin and measured up to 0.2 cm. in thickness. Within the cyst there was a grayish-tan tubular structure present, resembling a developing gastrointestinal Table 1. Organized Gastrointestinal Development in Benign Cystic Teratomas Perls (1876) 5 Pommer(1890) 5 Repin(1892) 5 Arnsperger(1899) 5 Askanazy (1904) 5 Shattock(1904) 2 * Andrews (1912)' Schoenholz(1923) 2 * Schottenfeld(1938) 10 Willis (1948)" Willis (1951) 12 Novak (1952) 8 Herbut(1955) 6 Peterson (1955)' Bernstine(1959) 2 * As quoted from article by Bernstine. 1 Colon with meconium Cecum and appendix Colon with meconium Portion of esophagus Intestine opening directly into respiratory tract Homunculus-like structure with intestinal loops in a celomic cavity Bowel loops Loops of colon Appendix and ileum Extra ovarian teratomas with bowel segments Loops of intestine Loops of intestine Bowel segments Bowel segments Extra cystic colon with mesentery and meconium

5 240 WOODFIELD ET AL. A.J.C.P. February 1985 tract grossly, measuring 4.5 cm in length and with the diameter varying from 0.5 cm proximally to 1.0 cm distally. On cut section, a central lumen was noted throughout, lined by a mucosal surface. No suggestion of liver, pancreas, or gallbladder was noted grossly. The microscopic sections of the cystic teratoma again included the common elements seen in a benign cystic teratoma, including skin, hair follicles, adipose tissue, and cartilaginous tissue. The sections, however, from the tubular structure demonstrated all elements of the gastrointestinal system, including portions of the esophagus (Fig. 1), stomach (Fig. 2), duodenum (Fig. 3), small bowel (Fig. 4), and colon (Fig. 5). Discussion Benign cystic teratomas in a series of 225 cases from Blackwell were found to have ectodermal derivatives in 100%, mesodermal derivatives in 93%, and endodermal derivatives in 71% of cases. 3 Endodermal structures found included salivary, lung, bronchial, thyroid, and gastrointestinal epithelium. In three large series, gastrointestinal epithelium was present only 7-13% of the time Table 1 is a review of the organized gastrointestinal development in the literature to date. 1 Small and large bowel are reported most often. Most organized development has occurred in ovarian cysts, but is reported in other areas such as the sacrococcygeal region. 12 Other unusual cases include evidence of extra cystic colon, 2 esophagus, 5 cecum with appendix, 5 and ileum with appendix. 10 In this unusual case, esophagus, stomach, duodenum, small intestine, and large intestine are represented. References 1. Andrews HR: A small ovarian teratoma containing brain and well-formed intestine. Proc Roy Soc Med 1912, 62: Bernstine JB, Jernstrom P: Benign cystic teratoma of the ovary with extracystic segment of colon: An unusual case. Am J Obstet Gynecol 1959; 77: Blackwell WJ, Dockery MD, Masson JC, et al: Dermoid cysts of the ovary: Their clinical pathologic significance. Am J Obstet Gynecol 1946; 51: Caruso PA, Marsh MR, Minkowitz S, et al: An intense clinicopathologic study of 305 teratomas of the ovary. Cancer 1971; 27: Ewing J: Neoplastic diseases, A treatise on tumors. Philadelphia, WB Saunders, 1940, p Herbut PA: Gynecologic and obstetric pathology. Philadelphia, Lea and Febiger, 1953, p Marcial-Rojas RA, Medina R: Cystic teratomas of the ovary: A clinical pathological analysis of two hundred sixty-eight tumors. Arch Pathol 1958; 66: Novak E: Gynecologic and obstetric pathology. Philadelphia, WB Saunders, 1952, p Peterson WF, Prevost EC, Edmunds FT, et al: Benign cystic teratomas of the ovary: A clinico-statistical study of 1,007 cases with a review of the literature. Am J Obstet Gynecol 1955; 70: Schottenfeld LE, Littauer EV: Bilateral ovarian dermoid cysts. Am J Obstet Gynecol 1938; 35: Willis RA: Pathology of tumors. Third edition. London, Butterworths and Co, 1966, p Willis RA: Teratomas in Armed Forces Institute of Pathology, Atlas of Tumor Pathology, Section HI, Fascicle 9, Washington D.C., 1951, pp 9-37 Muddy Lung MASAYUKI NOGUCHI, M.D., YUJI KIMULA, M.D., AND TAKESABURO OGATA, M.D. A 31-year-old man, a racing car driver, was submerged in muddy water as the result of an accident. He died from respiratory failure after a 17-day clinical course. Foreign body granulomatosis and massive fibrosis of the lung were revealed at autopsy. The crystalline foreign bodies mainly were composed of silicon and ranged in size from 20 nm to 500 pm in diameter (average, 90 /im). Their distribution in the lungs corresponded to the areas of lung carnification. In this study, the authors demonstrate that near drowning in muddy water causes pulmonary silicate granulomatosis associated with carnifieating Received February 1, 1984; received revised manuscript and accepted for publication March 19, Address reprint requests to Dr. Noguchi: Pathology Division, National Cancer Center Research Institute, Tsukiji 5-Chome, Chuo-ku, Tokyo, 104, Japan. Department of Pathology, University Hospital, University of Tsukuba, Ibaraki and Department of Pathology, Institute of Basic Medical Sciences, University of Tsukuba, Ibaraki, Japan fibrosis of the lung and term the pulmonary changes "Muddy Lung." (Key words: Near-drowning; Silicate granulomatosis; Muddy lung) Am J Clin Pathol 1985; 83: IN VARIOUS CLINICAL and physiologic studies, it has been shown that pulmonary edema or some similar condition develops in the victims of near-drowning or submersion but rapidly reverses without any pulmonary abnormality. 6 It is not surprising that the pathologic