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Home , Struma ovarii

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@

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truma ovarii is a very rare 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 (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 containing benign 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 , 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 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 struma ovarii are thyroid. Biopsy of the ischium showed well differen very rare and most malignant struma have not metas tiated thyroid tissue. The patient reported by Eerland tasized (20—22). (18) was a 60-yr-old who had a metastasis to the cranial

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FIGURE2 A: Fociof papillaryarchitecturein the ovarianlesionare suggestiveof papillarycarcinoma(125x). B: Higherpower showing nuclear crowding and pleomorphism and open chromatin distribution and characteristic nudear grooves (arrows)(500x).

Volume30 •Number3 •March1989 409 FIGURE3 Vertebral lesion. A: Low power shpwing sheets of thyroid tissue infiltrating vertebral bone and soft tissue (40x). B: High powershowsdeceptivelyblandcytologyin metastasesin contrastto ovarianlesion(Fig.2B)(400x). vault. Although the 67-yr-old patient of DeBakay and asymptomatic syndromes. Ann mt Med 1970; 72:883— DeLeboczhy (19) had a brain metastasis, at the end of 893. 2. Brown WW, Shetty KP, Rosenfeld PS. Hyperthyroid their brief article they state the patient had “struma ism due to struma ovarii:demonstration by radioio metastases arising in the skull and brain―,but give no dine scan. Acta Endocrinologica 1973; 73:266—272. other details. Kempers et al. (1) describe a patient who 3. Gusberg SB, Danforth DN. Clinical significance of developed liver and bone metastases 7½yr after pres struma ovani. Am J Obstet Gynecol 1944; 48:537— entation with malignant struma ovarii. Gonzalez-An 542. 4. Kawahara H. Struma ovarii with ascites and hydro gulo et al. (8) make reference to a patient who had thorax. Am J Obst Gynecol 1963; 85:85—89. metastases to bone (1 7) but provide no clinical details. 5. WoodruffJD, Rauh JT, Markley RL. Ovarian struma. In this patient the skeletal lesion was proven patho Obstet Gynecol 1966; 27:194—202. logically to be thyroid and this was substantiated by its 6. Janovski NA, Paramanandhan TL. Ovarian tumors. ability to trap and concentrate iodine. The thyroid was Vol 4. Major problems in obstetrics and gynecology. Philadelphia: W.B. Saunders Co., 1973:89—90. excluded as the primary source by careful pathological 7. Marcus CC, Marcus SL. Struma ovarii: a report of 7 evaluation of 2-mm sections, which were all normal. cases and a review ofthe subject. Am J Obstet Gynecol Thyroidectomy made possible therapy of the spinal 1961;81:752—762. metastasis with ‘@‘I.There are few reports of treatment 8. Gonzalez-Angubo A, Kaufman RH, Braungardt CD, of functioning metastasis by this approach (1,11). One Chapman FC, Hinshaw AJ. of the thyroid arising in struma ovarii (malignant struma with a pulmonary lesion did well (14). Although the ovarii), a report of two cases and review of the liters number of cases of localized and metastatic malignant ture.ObstetGynecol1963;21:567—576. struma ovarii is small, it is thought that the prognosis 9. Yannopoulos D, Yannopoubos K, Ossowski R. Malig is similar to that of similar stage and type of thyroid nant strums ovarii. PatholAnn 1976; P403—413. cancer arising in the thyroid. 10. Rotton WN, Tovell HMM. Malignant struma ovarii: a case report. Bull Sloane Hosp Wom 1956; 2:12 1— 127. REFERENCES 11. Peterson WF. Malignant degeneration ofbenign cystic of the ovary: a collective review of the 1. Kempers RD. Dockerty MB, Hoffman DL, Bartho literature. Obstet Gynecol Sun' 1957; 12:793—830. lomew LG. Struma ovarii-ascitic, hyperthyroid, and 12. Hasleton PS, Kelehan P, Whittaker JS, Burslem RW,

410 McDougall,Krasne,Hanberyetal TheJournalof NuclearMedicine Turner L. Benign and malignant struma ovarii. Arch tacao de um caso clinico rev. BrasilMed 1954; 11:11. Pathol Lab Med 102:180—184. 18. Eerland LD. Malignant struma ofovary with extensive 13. DeGraff J, Van Vliet MAT, Miseree J. Malignant metastases into cranial vault in Japanese women. Ned strums ovarii. EurJ Obstet GynecolReprodBiol 1983; erl Tydschr Verlosk Gynaec 1936; 3@‘377. 16:31—35. 19. DeBakay L, DeLehoczky F. Cerebrat ‘etastasisof @ 14. WoodruffJD, Markley RL. Strums ovarii. Obst Gyn ovarian struma. Obstet Gynecol Sun' 194 P266. ecol1957;9:707—719. 20. Nieminen I, Von Numers C, Windholm 0. Struma 15. Pardo-Mindan FJ, Vazquez JJ. Malignant strums ova ovarii. Acta Obstet Gynecol Scand 1964; 42:399—424. rii. Light and electron microscopic study. Cancer 21. Scully RE. Germ cell tumors of the ovary. In: Sturgis 1983;51:337—343. SH, Taymor ML, eds. Progess in gynecology.Vol. 1.5. 16. Wynn HMN, McCartney JS, McClendon JF. Struma New York: Grune and Stratton, 1970:329—348. ovarii.Am J ObstetGynecol1940;39:262—269. 22. Scully RE. Recent progress in . Human 17. Gurfinkel M. Struma ovarii-metastase ossea A presen Pathol1970;1:73—98.

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