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Metastatic Malignant Struma Ovarii Presenting As Paraparesisfrom a Spinal Metastasis

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Metastatic Malignant Struma Ovarii Presenting As Paraparesisfrom a Spinal Metastasis Case Reports Metastatic Malignant Struma Ovarii Presenting as Paraparesisfrom a Spinal Metastasis I. RossMcDougall,DavidKrasne,John W. Hanbery,and John A.Collins Division ofNuclear Medicine, Department ofDiagnostic Radiology & Nuclear Medicine, Department ofPathology, Division ofNeurosurgery, Department ofSurgery, Stanford University School ofMedicine, Stanford, California A 42-yr-oldwomanhada solItarymetastasesto herspine(T2)froma malignantstrumaovaril. Thethyroidwasexcludedas the siteof the primarycancer.Thelesioncausedparaparesis. Thespinalmetastasiswas treatedby surgeryandtwo dosesof 1311(200mCIeachtime).The patientrespondedverywellandis entirelyfreeof symptomsandsigns.Repeatwhole-body @ 1!, shows flQ @bflQrm8IIt@ J Nucl M@d3Oi4O7=@11,1OMQ truma ovarii is a very rare ovarian tumor which can For several years she had upper backache which had been present in various ways. It can be discovered on path attributed to stress; however, a radiograph from a chiroprac obogic examination of an asymptomatic ovarian mass, tor's office on 2/22/84 showed complete loss of the body of it can present as a cause of ascites and or hydrothorax, the second thoracic vertebra. it can cause hyperthyroidism (1-3) and very infre When she had an episode of acute abdominal pain in February, 1985 a left irregular, firm, tender ovarian mass (10 quently malignant transformation of the tumor can x 8 cm) was found on gynecologicexamination. A mature occur and be a source of metastases. This tumor is cystic teratoma containing benign thyroid tissue was diag extremely uncommon (4,5). It is defined as, “ateratoma nosed pathologically. In the months after the surgery she noted in which thyroid tissue is present exclusively or forms altered sensation from the breasts downwards. The sensory a grossly recognizable component of a more complex changes progressed until her transfer and first admission to teratoma―(6). The thyroid tissue is usually benign (7) our institute. She had a burning dysesthesia over the anterior but malignant changes have been described (8-13). thighs and numbness and tingling in lower legs and feet. She Distant metastases are extraordinarily rare (14) and had been incontinent of urine on several occasions in the Pardo-Mindan and Vazquez (15) in 1983 could only preceding2 mo. find 18 cases in the world literature including a patient On examinationshewaseuthyroid.Therewaslossoftouch oftheirs. Bony metastases from malignant struma ovarii and pain sensation from T2caudad. Power in lower limbs was have been described in only five patients (1,16-19). normal, reflexes were hyperactive, and plantar responses were equivocal. Computed tomographic (CT) and magnetic reso The purpose ofthis case report is to describe a patient nance imaging (MRI) scans showed a large tumor mass at the who has a single metastatic thyroid carcinoma (of ovar level of T2 involving the body of T2, with a bilateral and ian origin) in the spine. The primary ovarian lesion anterior paraspinal components, plus a mass projecting pos contained both benign thyroid and papillary thyroid teriorly which impinged on the spinal cord. A myelogram carcinoma. She has been treated successfully by surgery showed obstruction to flow ofcontrast. The patient underwent and radioiodine 131. an emergency operation to relieve pressure from the cord, to debulk as much tumor as possible, and to obtain tissue for CASE REPORT diagnosis. The tumor was extremely vascular and on frozen section the pathology was interpreted as metastatic follicular The patient, a 42-yr-old woman, was hospitalized in June, carcinoma probably from a thyroidal primary. Since the sur 1986. She had a spinal cord compression with sensory findings gical approach was anterior the thyroid was inspected and ofdecreased sensation to touch and pinprick from T2 caudad. palpated and judged to be normal. Thyroidectomy was not performed because of some uncertainty about the tissue of ReceivedMay 20, 1988;revisionaccepted Nov. 8, 1988. origin on frozen section. The spine was supported by a fibular For reprints contact: I. Ross McDougall, Div. of Nuclear graft and an external halo put in position. Medicine, Dept. of Diagnostic Radiology & Nuclear Medicine, The final pathologic diagnosis was follicular cancer of the Stanford University School of Medicine, Stanford, CA 94305. thyroid metastatic to bone. A thyroid scan (200 @@Ci'23I)was Volume30 •Number3 •March1989 407 technically difficult because of the external halo but showed the soft tissue mass. The patient was rescanned on 3/27/87, uptake in the thyroid and in the spinal lesion. A whole body 72 hr after 2 mCi'3'! and 4 wk after stopping exogenous ‘@‘iscan (2 mCi'3'I) confirmedthesefindingsand showedno thyroxine. At the time of scan TSH was 70 @tU/ml,and ‘@‘i other areas of uptake. By this time the ovarian pathology had uptake in the lesion 1%. No other lesions were noted on scan. been reviewed and the diagnosis of struma ovarii containing A second therapeuticdose of ‘@‘I(200 mCi) was prescribedat both typical thyroid and papillary carcinoma was made. that time and tolerated well. She continues to be normal in Thyroidectomy was advised for two reasons. Firstly, from all respects. Repeat whole body scan showed no abnormal the scans it was clear that normal thyroid was concentrating uptake of ‘@‘I,at the time of scanning TSH was >60 @iU/ml considerably more ‘@‘Ithan the spinal metastasis, and if the and Tg <5 ng/ml. metastasis was to be successfully ablated with ‘@‘I,removal of the bulk of normal thyroid would be important. Secondly, careful sectioning of the thyroid would ensure that a primary thyroid carcinoma was excluded. A near total thyroidectomy PAThOLOGY was undertakenon 7/4/86 with no complications. The oper ation was conducted after removal ofthe halo frame with the Ovarian Lesion patient's head and neck stabilized with sandbags. No cancer The left ovarian mass revealed abundant mature was found. appearing follicular thyroid tissue as well as areas of A second whole-body ‘@‘iscan one month after surgery, typical papillary carcinoma. The former was character without thyroid replacement, showed substantially more up ized by variably sized colloid-fihled follicles lined by take in the spinal lesion with only a small remnant of thyroid bland, low cuboidal thyroid type epithelium (Fig. 1A visible. Uptake of ‘@‘Iin the spinal lesion was 5%. The volume and B). This tissue had irregular, bluntly infiltrative of the cancer was estimated to be 50 G. The patient was borders into adjacent ovary, but no vascular invasion. treated with 200 mCi'3'! on 7/31/86. The neurological signs resolved entirely, the halo was removed in September, 1986, Foci of papillary carcinoma contained only occasional and she was able to commence studies in art school and papillary structures, which were characterized by greater recommence physical activities including tennis. A repeat MR cellularity with nuclear crowding and barge pleomorphic scan on 12/19/86 showed no impingement of the cancer on nuclei. “Orphan-Annie―chromatin clearing and nu the spinal canal and no cord compression with a decrease in clear grooves were also evident (Fig. 2A and B). A FIGURE1 A: Low powerof ovaryshowingirregularborderbetweenstrumaovarliandadjacentovarianstromaare noted(40x). B: Higherpowershowsfairlynormalfolliculararchitecturewith considerablevariationin folliclesizewhichsuggestsa predominantlybenignlesion(50x). 408 McDougall,Krasne,Hanberyetal The Journalof Nudear Medicine Spinal Lesion Identification of carcinoma in struma ovarii should Sections of bone from first and second thoracic ver be straightforward in instances where papillary carci tebrae showed metastatic follicular thyroid carcinoma noma is present because the diagnosis is primarily a which was histologically quite bland, and which closely cytologicone. However,becauseovarianstrumasare resembled the foblicubar thyroid tissue in the ovary (Fig. typically not encapsulated, consequently capsular 3A and B). Immuno-peroxide studies showed positive invasive criterion cannot be used. Diagnosis of follicular reaction to thyrogbobulin. No papillary carcinoma was carcinoma can be extremely difficult in the absence of found. vascular invasion. In our view, follicular carcinoma should not be diagnosed in a struma ovarn unless Thyroid metastases are documented or unequivocal vascular The entire thyroid specimen was sectioned at 2.0- invasion is present. We interpreted the vertebral metas mm intervals. Macroscopically and microscopically tases in this case as probable metastatic foblicular variant there was no evidence of a primary thyroid cancer or of papillary carcinoma. any evidence of metastases. Struma ovarii can be asymptomatic, can cause ascites and hydrothorax (Meig's Syndrome) (1), or hyperthy roidism (1—3)or rarely symptoms due to metastases DISCUSSION (8-18). Pardo-Mindan and Vazquez (15) could only find 17cases with metastases in the literature and added Struma ovarii is a rare ovarian tumor. To fulfill this a further case. Only five have been diagnosed with diagnosis the ovarian lesion should be composed en skeletal involvement which makes this case worthy of tirely of thyroid tissue or contain grossly recognizable reporting. Wynne et al. (16) described a 26 yr old who thyroid elements (6). In two series of 1000 and 2000 developed pain in the right ischium 1 yr after removal ovarian tumors, struma ovarii accounted for 0.3% (4) of a “benign―ovarian teratoma which contained only and 0.65% (5) of cases. Malignant
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