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Case Report |
1
Department of Radiology, Saitama Medical School, 38 Morohongo Moroyama Iruma,
Saitama, 350-0495 Japan.
2
Department of Radiology, Otsu Municipal Hospital, 2-9-9 Motomiya Otsu, Shiga,
520-0804 Japan.
3
Department of Radiology, Kasukabe Municipal Hospital, 7-2-1 Chuo Kasukabe,
Saitama, 344-0067 Japan.
4
Department of Obstetrics and Gynecology, Kasukabe Municipal Hospital, Saitama,
344-0067 Japan.
5
Department of Pathology, Dokkyo University, School of Medicine, 880
Kitakobayashi Mibu Shimotsuga, Tochigi, 321-0293 Japan.
Received September 20, 2000;
accepted after revision December 5, 2000.
Address correspondence to S. Kawamoto.
Introduction
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We present a case of a 24-year-old woman with multiple spherical structures floating in a large cystic mass in the lower abdomen and pelvis seen on sonographic and MR images. A mature cystic teratoma of the ovary was the presumed preoperative diagnosis. The patient underwent excision of the lesion, and pathologic diagnosis was consistent with a mature cystic teratoma. The striking aspect of multiple spherical structures in the cyst was caused by multiple spherical desquamative keratin formations.
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MR imaging was performed for further characterization of the lesion. A cystic mass measuring 22 x 24 x 12.5 cm occupied the lower abdomen and pelvis, extending to the level of the upper pole of the kidneys. There were numerous small spherical structures approximately 1 cm in diameter and two larger ones (3-4 cm) floating in the nondependent portion of the cystic mass. On sagittal T1-weighted images, the outer portion of the spherical structures was slightly hyperintense to the surrounding fluid, but not as hyperintense as subcutaneous fat. The center of the spherical structures was relatively hypointense compared with the outer portion. There was a fluiddebris level in the dependent portion of the cystic mass (Fig. 1B). On sagittal and coronal T2-weighted images, the outer portion of the spherical structures was hypointense, and the center relatively hyperintense (Figs. 1C and 1D). The normal left ovary could not be identified. The uterus and the right ovary were of normal appearance, and no other abnormal findings were detected in the abdomen or pelvis. Mature cystic teratoma arising from the left ovary was considered as a preoperative diagnosis.
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Laparotomy revealed a large cystic tumor of the left ovary. The tumor contained numerous small and two larger black spherical structures (Fig. 1E). Hair was present in some of these spherical structures. The internal surface of the cystic tumor was smooth, and there was no solid component. The right ovary and the uterus were normal.
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The diagnosis at pathology was mature cystic teratoma of the ovary. Contained within the cyst wall were cartilage, skin and its appendices, muscle, ganglion, peripheral nerve, salivary gland, and fatty tissue. There was no evidence of immature features, and no evidence of malignancy. A striking finding was numerous dark black, small spherical structures of a claylike material within the cyst. Two larger spherical structures also had similar texture. Microscopic examination found desquamative keratin spherules containing fibrin, hemosiderin, and hair. Azan stain failed to show a fat component in these spherules.
The patient's postoperative course was uneventful, and she was discharged 12 days after surgery.
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Unusual associated findings of mature cystic teratoma may result in occasional diagnostic difficulty. To our knowledge, only a small number of cases of cystic teratoma with multiple mobile spherules or globules have been reported in the literature. These lesions were found in the ovary [5, 6], retroperitoneum [7], and mediastinum [8]. In the case of cystic teratoma of the ovary, the nodules consisted of sebaceous debris with skin squames and hair [6]. In mature cystic teratoma of the mediastinum, mobile globules consisted of pastelike material, fat, and hair [8]. In retroperitoneal mature cystic teratoma, fat deposition was seen around hair tissue [7]; these spherical structures have been called intracystic fat balls [5, 7]. In our patient, however, fat was not revealed in the spherules with azan stain, but it was possible that the fat component had dissolved during the fixation process. In the reported cases and our patient, the center of the spherical structures showed relatively lower signal intensity on T1-weighted MR images, corresponding to hair or a softer component [5, 6]. It has been speculated that each spherule was formed by the aggregation of sebaceous matter around a nidus (a tiny focus of debris, disquamative material, or fine hair shaft), and may be formed into a discrete mass because of the difference in physical and thermal properties of the material being deposited [7]. The specific gravity of spherules or globules is lower than that of the surrounding fluid, so that they float and are mobile in the cyst. Other than mature cystic teratoma, similar multiple spherules in the cystic mass have been reported in a patient with an epidermoid cyst in the floor of the mouth [9]. In this case, the spherules consisted of multiple spherical keratin formations. To the best of our knowledge, the appearance of multiple spherules floating within a pelvic cystic tumor has not been found in other tumors, and we think this appearance is pathognomonic for a cystic teratoma.
In summary, we present a case of mature cystic teratoma of the ovary containing numerous spherules floating in the cyst. Sonography and MR imaging revealed numerous spherical structures floating in the cystic mass, which is thought to be pathognomonic for a cystic teratoma. Microscopically, these spherical structures were composed of desquamative keratin containing fibrin, hemosiderin, and hair.
Acknowledgments
We thank Hisataka Kobayashi for valuable discussion and editorial
assistance.
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