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Vidhale SA, Chavarkar SP, Sudhamani S, Rao R. Cholesterolosis of ovary associated with benign serous cystadenoma. Indian J Pathol Microbiol 2022;65:496-8
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Vidhale SA, Chavarkar SP, Sudhamani S, Rao R. Cholesterolosis of ovary associated with benign serous cystadenoma. Indian J Pathol Microbiol [serial online] 2022 [cited 2022 May 3];65:496-8. Available from: https://www.ijpmonline.org/text.asp?2022/65/2/496/343200
We present a rare case of cholesterolosis of ovary with benign serous cystadenoma which to the best of our knowledge is the first description of this entity. Cholesterolosis refers to the accumulation of lipid, particularly cholesterol esters and triglycerides, within the macrophages of lamina propria and to a lesser extent, in the epithelium itself. It is commonly seen in the gallbladder but very rarely reported in ovary and poorly understood entity.
They are not true tumors, but reactive histiocytic proliferations that occur in both normolipidemic and hyperlipidemic conditions. Visceral aggregates of foamy macrophages which are unassociated with inflammation and/or hemorrhage are decidedly rare and almost exclusively encountered in the gastrointestinal tract, especially in the stomach. Xanthogranulomatous inflammation is a special form of chronic inflammation that is destructive to normal tissue of affected organs. It most commonly affects kidney but has also been reported in the gallbladder, stomach, anorectal area, bone, urinary bladder, testis, epididymis, vagina, and endometrium. It is characterized by dense infiltration of tissues by lipid-laden histiocytes admixed with inflammatory infiltrate.
It is important to recognize this entity as cholesterolosis may mimic many ovarian tumors, both on gross and microscopy.
A 46-year-old multiparous female presented to gynecology outpatient department with abdominal pain of one-and-a-half-month duration. On per abdomen examination, she had vague tenderness in her left iliac fossa. Her pelvic examination did not reveal any abnormal vaginal discharge. Ultrasound showed a 19.5 × 11.5 × 10 cms bilocular cystic lesion in the left adnexa from which left ovary was not seen separately. Serum CEA was 1.12 ng/ml (normal: ≤3 ng/ml) and CA125 was 117.9 U/ml (normal: 0–35 U/ml). Her routine blood reports including lipid profile were within normal limits. She had undergone tubal ligation 6 years back. Her left ovarian cyst was excised. We received a single biloculated cystic mass measuring 20 × 13 × 11 cms which was filled with thick brown hemorrhagic fluid. The mucosal surface showed yellowish streaks and white polypoid nodules ranging from 1 cm to 0.2 cm in diameter [Figure 1]. The attached fallopian tube was dilated. On frozen sections, cyst wall showed serous lining without evidence of malignancy; thus, a diagnosis of benign serous neoplasm of ovary was offered. Paraffin-embedded histopathological sections showed that the cyst wall and micro papillae were lined by columnar to cuboidal epithelium, with nuclear crowding and stratification focally. Subepithelium showed diffuse infiltrate of large foamy cells with vacuolated cytoplasm and centrally placed bland nuclei. There was no associated inflammatory infiltrate [Figure 2]. Focally individual tumor cells showed minimal cytologic atypia which was less than 10% of the cyst wall and less than 5 mm in confluent growth, thus ruling out borderline serous cystadenoma. The attached fallopian tube showed features of hydrosalpinx. Based on these features, a diagnosis of cholesterolosis of ovary with benign serous cystadenoma was made.
|Figure 1: Cut section shows numerous yellow streaks along with variably sized white gelatinous polypoid nodules (original image)|
|Figure 2: Photomicrograph showing cyst wall and micropapillae lined by cuboidal cells with diffuse collection of foamy histiocytes in the subepithelium(H and E, (x10; original image))|
Pusiol et al. have reported a case of xanthomatous serous cystadenoma of the broad ligament. This lesion was separate from the ipsilateral ovary and fallopian tube. Likewise Chetty et al. have reported a case of fallopian tube xanthoma.
We presume the xanthoma in our case was mostly post inflammatory from salpingitis and/or salpingo-oophoritis. It is possible that the only stigma remaining from preceding infection is foamy histiocytes. This is the simplest explanation, but the absence of accompanying inflammation and scar tissue goes against this hypothesis.
In cholesterolosis, it is postulated that the accumulation of lipid-laden histiocytes is due to increased mucous membrane synthesis of lipid or to the failure of macrophages to metabolize or excrete cholesterol. Increased absorption will occur if serum is supersaturated with cholesterol.
In terms of differential diagnosis, the major consideration was xanthogranulomatous inflammation occurring in the ovary and fallopian tube. The presence of foamy histiocytes (xanthoma cells) and chronic inﬂammatory cells are consistently observed and are diagnostic of xanthogranulomatous oophoritis. Xanthoma cells are histiocytes with abundant lipid-laden cytoplasm having a vacuolated appearance. Aggregates of such foam cells are responsible for the yellow color observed on gross examination. Analyzed from the reported cases, the emergence of foam cells could be relevant to the following factors. The first one is inefficient or inappropriate antibiotics applied in the early phase of infection which was not able to control multiplication of bacteria. Macrophages phagocytize lipid and transform into foam cells. The second one is lipid metabolic disorder which induces hyperlipidemia, and foam cells are formed when the lipid deposited in the affected region is phagocytozed by phagocytes. The third one is the use of intrauterine contraceptive device or drugs, the later contains lipoid substance.
In our case, inflammatory infiltrate was absent which could be explained by the complete resolution of mild inflammation. Although rare, the possibility of cholesterolosis of ovarian cyst should be considered when foamy histiocytes are unassociated with inflammatory infiltrate as surgical excision is curative.
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