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LETTERS TO EDITOR  
Year : 2018  |  Volume : 61  |  Issue : 1  |  Page : 164-166
Multilocular peritoneal inclusion cyst, rare occurrence in men: A case report


Department of Pathology, Seth GS Medical College, Mumbai, Maharashtra, India

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Date of Web Publication22-Mar-2018
 

How to cite this article:
Mishra T, Karegar MM, Rojekar AV, Joshi AS. Multilocular peritoneal inclusion cyst, rare occurrence in men: A case report. Indian J Pathol Microbiol 2018;61:164-6

How to cite this URL:
Mishra T, Karegar MM, Rojekar AV, Joshi AS. Multilocular peritoneal inclusion cyst, rare occurrence in men: A case report. Indian J Pathol Microbiol [serial online] 2018 [cited 2019 Dec 13];61:164-6. Available from: http://www.ijpmonline.org/text.asp?2018/61/1/164/228174




Editor,

Multilocular peritoneal inclusion cyst (MPIC) is an unusual lesion arising from the peritoneal mesothelium. Although benign, local recurrences are common, often requiring repeated surgeries.[1],[2],[3] It usually occurs in the pelvis of reproductive-age women with a history of previous abdominal surgery, endometriosis, or pelvic inflammatory disease.[1],[3] Some experts believe these lesions represent a true neoplasm, while others advocate a reactive process.[1],[2],[3]

Adult men are rarely affected; as per our best knowledge, very few cases of MPIC in men have been published in literature.[4] We herein report a rare case of MPIC in an adult male.

A 43-year-old man presented with a painless lump in the right hypochondrium for the last 20 days. There was no history of any past surgery.

Ultrasonography and computed tomography (CT) showed a large multiseptate lesion located anterior to cecum and terminal ileum and deep to anterior abdominal muscle. Liver and visualized bowel loops were normal. On imaging studies, differential diagnoses of venolymphatic malformation, chronic loculated ascites, and pseudomyxoma peritonei were given.

On laparoscopy, myxoid stroma was seen in the right colonic area which was highly suspicious for pseudomyxoma peritonei, and hence, an exploratory laparotomy was done. A right hemicolectomy with the complete resection of the tumor was performed.

Gross examination revealed a large multiloculated cystic mass measuring 21 cm × 13 cm × 8 cm involving the mesentery and comprising of numerous translucent cysts ranging in size from 0.2 cm to 2.5 cm in diameter, filled with gelatinous material [Figure 1]. The cyst lining was uniformly smooth, and the septae were thin. Papillary excrescences, hemorrhage, necrosis, or solid areas were not identified. The ileum, cecum, and appendix were unremarkable. Microscopic examination showed multiple cysts, lined by bland flattened to cuboidal epithelium and separated by septae showing fibrosis, multiple lymphoid aggregates, and dense lymphoplasmacytic infiltrate [Figure 2]a and [Figure 2]b. On immunohistochemistry, the cells lining the cysts were strongly and diffusely positive for calretinin while they were negative for CD34 [Figure 2]c and [Figure 2]d. Hence, a diagnosis of MPIC was rendered.
Figure 1: Gross photograph shows: (a) large cystic mass involving the mesentery. (b) multiloculated mass with cysts of varying sizes

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Figure 2: (a) Variably sized multiple cystic spaces lined by flattened to cuboidal cell (H and E, ×200). (b) Intervening stroma showing fibrosis and prominent lymphoplasmacytic infiltrate (H and E, ×40). (c and d) The cells lining the cysts are strongly positive for calretinin (×100) while negative for CD34 (×40)

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The patient had a good postoperative recovery and had no recurrence on follow-up CT scan after 1 year of surgery.

In 1979, Mennemeyer and Smith first demonstrated the mesothelial origin of MPICs by electron microscopy.[2] It is a very rare lesion with <200 cases reported in the literature, of which only about 20% of cases are reported in adult men.[1],[4],[5] Presenting symptoms are nonspecific. The available imaging techniques do not differentiate it from other cystic neoplastic or inflammatory lesions.[6]

The gross and histopathological findings in the present case were similar to those described in literature. The most common differential diagnosis of MPIC is cystic lymphangioma.[2],[4] In difficult cases, IHC can assist as lymphangioma positively expresses endothelial markers and is negative for mesothelial markers, while the opposite is true for MPIC. Differentiation between MPIC and lymphangioma is important as the latter rarely recurs following surgical excision. Conditions from which MPIC may (rarely) require differentiation include reactive mesothelial proliferation; malignant mesothelioma with cystic change; pseudomyxoma peritonei; endometriosis; and ovarian carcinoma.[1],[2],[3],[6]

Thus, to conclude, MPIC is a very rare benign cystic tumor. This lesion has a nonspecific appearance on imaging and always requires careful histological evaluation. Despite a high recurrence rate, the prognosis is good after complete surgical resection since malignant transformation has very rarely been reported. A systematic follow-up of these patients is essential to detect recurrence and further management.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Ross MJ, Welch WR, Scully RE. Multilocular peritoneal inclusion cysts (so-called cystic mesotheliomas). Cancer 1989;64:1336-46.  Back to cited text no. 1
    
2.
Mennemeyer R, Smith M. Multicystic, peritoneal mesothelioma: A report with electron microscopy of a case mimicking intra-abdominal cystic hygroma (lymphangioma). Cancer 1979;44:692-8.  Back to cited text no. 2
    
3.
Weiss SW, Tavassoli FA. Multicystic mesothelioma. An analysis of pathologic findings and biologic behavior in 37 cases. Am J Surg Pathol 1988;12:737-46.  Back to cited text no. 3
    
4.
Stojsic Z, Jankovic R, Jovanovic B, Vujovic D, Vucinic B, Bacetic D, et al. Benign cystic mesothelioma of the peritoneum in a male child. J Pediatr Surg 2012;47:e45-9.  Back to cited text no. 4
    
5.
Safioleas MC, Constantinos K, Michael S, Konstantinos G, Constantinos S, Alkiviadis K, et al. Benign multicystic peritoneal mesothelioma: A case report and review of the literature. World J Gastroenterol 2006;12:5739-42.  Back to cited text no. 5
    
6.
Romero JA, Kim EE, Kudelka AP, Edwards CL, Kavanagh JJ. MRI of recurrent cystic mesothelioma: Differential diagnosis of cystic pelvic masses. Gynecol Oncol 1994;54:377-80.  Back to cited text no. 6
    

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Correspondence Address:
Toshi Mishra
Department of Pathology, Seth GS Medical College, Mumbai, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_480_16

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