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Pathology (2015), 47(7), December

Sir, Mature cystic teratoma (dermoid cyst; MCT) is the most common benign tumour of the ovary.1 Occasionally, fat necrosis is the only evidence that an MCT has been present.2 We found a different appearing lesion which we also ascribe to a previous MCT. Our lesion appeared as close-packed laminated saccules associated with a foreign body giant cell reaction. The pathogenesis is discussed. The patient was a 71-year-old female who had bilateral salpingo-oophorectomy for a small left ovarian fossa lesion that had not changed significantly in size or characteristics over the previous almost 10 years surveillance. The patient requested removal of the lesion in view of her sister who carried a diagnosis of ovarian cancer. There was no previous surgical history. At surgery, the previously imaged lesion

was identified as a pedunculated uterine fibroid. The tubes and ovaries, which appeared normal, were removed. Pathological examination confirmed that the pedunculated uterine lesion was a leiomyoma. The tubes and ovaries were normal macroscopically, but microscopically one of the ovaries contained an incidental finding of an unencapsulated 6 mm nodule in the medulla (Fig. 1). The nodule was composed of many close-packed fairly uniform saccules up to 1 mm in diameter (Fig. 2). The saccules contained pale eosinophilic concentric laminations and were bordered by a histiocytic giant cell reaction. Some saccules contained a central amorphous core or were calcified. Between the saccules, there were small collections of foreign body giant cells, some of which contained brown granules. When the block was cut to extinction, a single 0.7  0.05 mm fibrillary structure identified as a hair shaft was observed in a few levels in the centre of the block (Fig. 3A). Under polarised light it showed weak birefringence supporting the interpretation that it was a hair shaft (Fig. 3B). The material within the saccules was weakly alcian blue positive at pH 2.5. Some of the brown granules in the giant cells were Perls’ positive indicating haemosiderin. Immunoperoxidase showed the contents of the saccules were positive for CD68, probably due to lysosomes, but negative for involucrin, carcinoembryonic antigen (CEA), epithelial membrane antigen (EMA), thyroglobulin and glial fibrillary acid protein (GFAP). We made a diagnosis of MCT based on the unequivocal evidence of the hair shaft. MCT was the only plausible explanation of a hair shaft within an ovary which has not undergone previous surgery. Supportive evidence is that MCT is the most common ovarian tumour and unusual degenerate changes such as myospherulosis, fat necrosis and keratin granulomas may be seen in MCT.2–3 To explain the unusual degenerative appearance, we considered myospherulosis, fat necrosis, keratin granulomas and other substances which might be produced by a teratoma. Myospherulosis is rare condition with a distinctive multisaccular appearance. The saccules are bordered by multinucleated foreign-body giant cells or fibrous walls and contain numerous spherules.4 The spherules are degenerating red blood cells. The pathogenesis is a long-standing chemical reaction between extravasated erythrocytes and exogenous or endogenous lipids. Our case showed the low power

Fig. 1 The medulla of the ovary shows a small lesion composed of closepacked saccules (whole slide mount, H&E).

Fig. 2 Saccules lined by histiocytic giant cells and containing laminated material (H&E).

4. Battaglia A. The importance of multidisciplinary approach in early detection of BAP1 tumor predisposition syndrome: clinical management and risk assessment. Clin Med Insights Oncol 2014; 8: 37–47. 5. Murali R, Wiesner T, Scolyer RA. Tumours associated with BAP1 mutations. Pathology 2013; 45: 116–26. 6. Harbour JW, Onken MD, Roberson ED, et al. Frequent mutation of BAP1 in metastasizing uveal melanomas. Science 2010; 330: 1410–3. 7. Wiesner T, Obenauf AC, Murali R, et al. Germline mutations in BAP1 predispose to melanocytic tumors. Nat Genet 2011; 43: 1018–21. 8. Carbone M, Ferris LK, Baumann F, et al. BAP1 cancer syndrome: malignant mesothelioma, uveal and cutaneous melanoma, and MBAITs. J Translat Med 2012; 10: 179. 9. Busam KJ, Wanna M, Wiesner T. Multiple epithelioid Spitz nevi or tumors with loss of BAP1 expression: a clue to a hereditary tumor syndrome. JAMA Dermatol 2013; 149: 335–9. 10. Wiesner T, Fried I, Ulz P, et al. Toward an improved definition of the tumor spectrum associated with BAP1 germline mutations. J Clin Oncol 2012; 30: e337–40. 11. Wadt K, Choi J, Chung JY, et al. A cryptic BAP1 splice mutation in a family with uveal and cutaneous melanoma, and paraganglioma. Pigment Cell Melanoma Res 2012; 25: 815–8. 12. Murali R, Wilmott JS, Jakrot V, et al. BAP1 expression in cutaneous melanoma: a pilot study. Pathology 2013; 45: 606–9.

DOI: 10.1097/PAT.0000000000000337

Laminated saccules: a residuum of an ovarian mature cystic teratoma

Copyright © Royal College of pathologists of Australasia. Unauthorized reproduction of this article is prohibited.

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Fig. 3 (A) Fragment of hair shaft overlying saccule (H&E). (B) Hair shaft showing weak birefringence under polarised light. Same field as (A).

multisaccular appearance of myospherulosis, but the contents of the saccules were laminations rather than spherules. However, the prerequisites of myospherulosis were present. The haemosiderin in the histiocytic giant cells indicates previous haemorrhage and the hair shaft is presumptive evidence that lipid was present, as hair formation in MCT is associated with a subcutis. We suggest our lesion was an ancient form of myospherulosis where degenerate red blood cells have been engulfed and destroyed by macrophages which themselves have died leaving only their cytoplasmic contents of lysosomes. In the differential diagnosis of the multisaccular structure, we considered fat necrosis and degenerate other substances that might be produced by an MCT. Fat necrosis may be seen in MCT where it appears as variably sized clear vacuoles engulfed by foreign body giant cells.2 Against fat necrosis, the contents of the saccules were laminated and not optically clear. MCT may have epidermal, neural and thyroid components. There was no evidence of these components on routine histology or immunoperoxidase. It remains possible that diagnostic substances may have been removed by phagocytosis over an extended period of time. Since there was no previous surgery and the lesion was deep in the ovarian parenchyma, exogenous structures such as partially degraded suture material were excluded. In summary, we describe an unusual incidental finding in the ovary of a 71-year-old woman of a small, multisaccular structure containing laminated eosinophilic material, which we postulate to be an ancient form of myospherulosis from an MCT. Conflicts of interest and sources of funding: The authors state that there are no conflicts of interest to disclose. Aadil Rahim1 Ran May Saw1 John Bailey2 James Scurry3 1

Faculty of Health Sciences, University of Newcastle, Newcastle, 2Private Practice, Toronto, and Department of Obstetrics and Gynaecology, John Hunter Hospital, Newcastle, and 3Anatomical Pathology, HAPS, Newcastle, and Faculty of Health Sciences, University of Newcastle, Newcastle, NSW, Australia Contact A/Prof James Scurry. E-mail: [email protected]

1. Templeman CL, Fallat ME, Lam AM, et al. Managing mature cystic teratomas of the ovary. Obstet Gynecol Surv 2000; 55: 738–45. 2. Ramdial PK, Bagratee JS. Membranous fat necrosis in mature cystic teratomas of the ovary. Int J Gynecol Pathol 1998; 17: 120–2. 3. Vuong PN. Spontaneous myospherulosis developing at contact with a mature cystic dysembryoma of the ovary. Pathogenetic discussion apropos of a case diagnosed by laparoscopic biopsy. J Gynecol Obstet Biol 1988; 17: 15–8. 4. Phillip V, Becker K, Bajbouj M, et al. Myospherulosis. Ann Diagn Pathol 2013; 17: 383.

DOI: 10.1097/PAT.0000000000000338

Combined serous carcinoma and neuroendocrine carcinoma of the fallopian tube Sir, Primary fallopian tube carcinoma is rare, accounting for approximately 0.14–1.8% of female reproductive malignancies.1 Combined serous carcinoma (SC) and neuroendocrine carcinoma (NEC) have been reported in the ovary and endometrium.2,3 However, combined SC and NEC of the fallopian tube has not been reported in the literature, based on a PubMed/ Medline search. Herein, we present the first case of a combined high-grade SC and NEC of the fallopian tube. A 57-year-old post-menopausal woman was admitted to our hospital for pelvic pain and urinary frequency. She had a history of hysterectomy for uterine leiomyoma about 20 years prior. Computed tomography (CT) showed a 7.6  6.2 cm irregular mass in the left adnexal site and seeding nodules in the peritoneum. Exploratory laparotomy was performed. During the surgery, a solid mass was found at the left fallopian tube, with a normal appearing left ovary and right adnexa (Fig. 1A). The outer wall of the small bowel was also infiltrated by the tubal mass. Microscopically, the tumour was composed of two morphologically distinct components. The central area of the tumour contained a definitive high-grade SC. The SC areas were composed of tumour cells showing papillary, glandular, and solid growth patterns with highgrade nuclear atypia (Fig. 1B). The SC areas were surrounded by an undifferentiated tumour component. The undifferentiated tumour component was characterised by solid sheets of medium to large sized cells with abundant mitotic figures, apoptotic bodies, and necrotic foci. The tumour cells were densely packed and showed amphophilic to eosinophilic

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Laminated saccules: a residuum of an ovarian mature cystic teratoma.

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