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CASE REPORT Table of Contents  
Ahead of print publication
Concurrent occurrence of primary mesenteric hydatid cyst with multiple calcified splenic granulomas: A rare presentation


 Department of Radiology, St. John's Hospital, Bengaluru, Karnataka, India

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Date of Submission04-Jun-2021
Date of Decision26-Jun-2021
Date of Acceptance30-Jun-2021
Date of Web Publication05-Aug-2021
 

  Abstract 


Hydatid cysts most commonly present in the liver and the lungs; however, they can appear more rarely in other locations, such as the mesentery, with a rather unclear mechanism of manifestation. Herein, a case of the simultaneous presence of hydatid cysts in the mesentery and multiple splenic calcified granulomas in a 62-year-old elderly man has been presented that was managed surgically by pelvic mesenteric cyst deroofing and subtotal cystectomy.

Keywords: Multiple splenic granulomas, primary mesenteric hydatid cyst, ultrasonography


How to cite this URL:
Reddy R. Concurrent occurrence of primary mesenteric hydatid cyst with multiple calcified splenic granulomas: A rare presentation. Apollo Med [Epub ahead of print] [cited 2021 Nov 30]. Available from: https://www.apollomedicine.org/preprintarticle.asp?id=323265





  Introduction Top


Hydatid disease is most commonly caused by Echinococcus granulosus in which humans are accidentally an intermediate host. The disease occurs in the liver in 70% of cases, the lungs in 25% of cases, and at other sites such as the spleen, kidney, pancreas, brain, ovary, mesentery, vertebra, and soft tissue of the neck.[1] Anaphylactic shock, rupture of cyst into the biliary tree, and rupture into the peritoneum are the most severe complications. Intraperitoneal hydatid cyst is usually secondary to rupture of the primary hepatic cyst. Primary mesenteric hydatid cyst is very rare. Diagnosis of hydatid cyst is achieved by the combination of serological tests and imaging, while the history of exposure is also important. Imaging findings vary depending on the stage of the cyst. In 2001, the World Health Organization developed a cystic echinococcosis classification system, to decide the appropriate management worldwide.[2] This system is based on ultrasound imaging findings of cysts. Classical imaging may consist of ultrasound examination, computed tomography (CT), and magnetic resonance imaging. Calcification of hydatid cysts can be present in 30% of all cases at the time of diagnosis.[3] In this article, a case of primary mesenteric hydatid cyst in a 62-year-old elderly man has been presented.


  Case Report Top


A 62-year-old elderly man presented with a 3-month history of intermittent attacks of abdominal pain, abdominal distention, recurrent vomiting, and nausea. Abdominal examination revealed a palpable mass (7 cm × 5 cm) at the periumbilical region, firm with restricted mobility. Laboratory investigations were unremarkable. High-resolution ultrasonography of the abdomen revealed a well-defined cystic intraperitoneal mass measuring 7 cm × 5 cm with multiple internal septations and no evidence of solid component [Figure 1]a and [Figure 1]b. Furthermore, there was mild splenomegaly with multiple subcentimetric calcific nodules scattered within the splenic parenchyma [Figure 2]. Hydatid serology was positive. Chest radiography was normal. A diagnosis of primary mesenteric hydatid cyst with multiple calcified splenic granulomas was made and the patient was referred to the Department of general surgery. At laparotomy, a cyst of approx. size 7 cm × 5 cm was noted in the small bowel mesentery. When the cystic lesion was opened a hydatid-laminating membrane and daughter cysts were found confirming the diagnosis of hydatid cyst. The cyst was irrigated with a scolicidal solution (hydrogen peroxide), and a subtotal cystectomy was performed. Further, the spleen was left untouched due to the imaging finding of calcified granulomas which represent dead/healed lesions. Furthermore, there was no history of hydatid disease, thus confirming the diagnosis of primary mesenteric hydatid cyst. The postoperative period was uneventful and the patient was discharged after 1 week. No recurrence was noted after a 2 years.
Figure 1: (a) High-resolution transverse ultrasonography image demonstrating a well-defined cystic lesion in the small bowel mesentery with multiple internal septations and the absence of solid component. (b) Color Doppler ultrasonography image showing absent internal vascularity within the septations

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Figure 2: High-resolution longitudinal ultrasonography image demonstrating mild splenomegaly with multiple calcified granulomas scattered within the splenic parenchyma representing healed hydatidosis

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  Discussion Top


Mesenteric hydatid cyst is the most unreported site in literature. These are rare lesions with the prevalence of 1:100,000 in adults and 1:20,000 in children.[4] The most common locations of hydatid cyst are liver and lungs but unusual locations such as brain, heart, pericardium, kidney, peritoneum, retroperitoneum, bone, soft tissue, and breast have been reported in the literature. Primary peritoneal echinococcosis is very rare and has been reported to occur in 2% of all abdominal hydatid diseases.[5] Clinical manifestations vary with the site and size of the cyst which ranges up to 10 cm and symptoms are due to space-occupying effect within the organs or systemic reactions due to perforation. Fortunately, they behave mostly as benign tumors, while malignancy accounts for 2% of cases.[6] There are no specific symptoms of mesenteric hydatid cyst and the disease usually remains asymptomatic for years. Clinical manifestation is due to the mass effect of enlarging abdominal cyst. The diagnosis may at times be difficult due to the unusual localization site; all abdominal cystic lesions including mesenteric, pancreatic, gastrointestinal duplication, ovarian cysts, and lymphangioma, must be considered in the differential diagnosis.

Isolated mesenteric cyst is a unique manifestation of hydatid disease. Owing to its rarity in area other than endemic areas and variable imaging appearances preoperative diagnosis of peritoneal/mesenteric hydatid disease is a clinical challenge so a suspicion of this disease is justified in all patients presenting with cystic mass, especially in endemic areas. The combinations of radiologic and serologic tests, especially in patients living in the endemic areas contribute to the diagnosis. High-resolution ultrasonography is the first line of screening for mesenteric hydatid cyst. CT scan is of particular importance in the designation of surgery strategy. Complement fixation test is positive in approximately 65%, and indirect hemagglutination test and Enzyme-linked immunosorbent assay (ELISA) have approximately 85% sensitivity.[7] The common complications are hydatid peritonitis (due to rupture of the cyst responsible for anaphylactic reaction), infection of the cyst, and compression of adjacent organs responsible for an occlusive syndrome.

Humans are usually asymptomatic for a long period while the cysts are growing slowly. In most cases, there is only one cyst, whereas in some cases, multiple cysts may be present (20%–40%).[8] Symptoms depend on the size and the number of cysts and possible compression of surrounding structures. As in the present case, signs of mesenteric hydatid disease may include nonspecific abdominal mass, pain due to traction on the mesentery, and pressure effects on adjacent organs. The signs and symptoms of liver hydatid disease, when present, include hepatomegaly, right upper abdominal or epigastric pain, nausea, and vomiting. Cyst leakage or rupture may lead to systemic immunological responses. Rupture in the peritoneal cavity may also cause secondary disease. In case of involvement of portal vein or biliary tract, cysts may be responsible for segmental or lobar liver atrophy. Retrograde spread from the liver via the hepatic and portal veins into the peritoneal cavity has also been reported.[9]

The treatment of choice is principally a careful and complete surgical excision; the partial or subtotal cystectomy can be performed to avoid adjacent organs injury.[10] The use of hypertonic saline or hydrogen peroxide solutions before opening the cavities tends to kill the daughter cysts and therefore prevent further spread or anaphylactic reaction. Mebendazole or albendazole are used as adjuvant therapy to surgery to prevent recurrence. However, in the case, the disease recurrence or multiple locations, chemotherapy should be used routinely.


  Conclusion Top


Isolated mesenteric cyst is a unique manifestation of hydatid disease. Simultaneous presence of cysts in different locations presents a low incidence in hydatid disease, while their presence in the mesentery is rarely reported. Owing to its rarity in area other than endemic areas and variable imaging appearances preoperative diagnosis of peritoneal/mesenteric hydatid disease is a clinical challenge so a suspicion of this disease is justified in all patients presenting with a cystic mass within the peritoneal cavity, especially in endemic areas. High-resolution ultrasonography is the first-line imaging investigation of choice in the diagnosis of primary mesenteric hydatid cyst and surgical excision remains the mainstay of treatment.

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.
Kushwaha JK, Gupta R, Mohanti S, Kumar S. Primary mesenteric hydatid cyst. BMJ Case Rep. 2012;2012 doi: 10.1136/bcr.03.2012.5996.  Back to cited text no. 1
    
2.
Paramythiotis D, Karakatsanis A, Bangeas P, Kofina K, Papadopoulos V, Apostolidis S, et al. Simultaneous hepatic and mesenteric hydatid disease-A case report. Front Surg 2017;4:64.  Back to cited text no. 2
    
3.
Köktekir E, Erdem Y, Gökçek C, Karatay M, Yılmaz A, Bayar MA, et al. Calcified intracranial hydatid cyst: Case report. Turkiye Parazitol Derg 2011;35:220-3.  Back to cited text no. 3
    
4.
Najih M, Chabni A, Attoulou G, Yamoul R, Yakka M, Ehirchiou A, et al. Isolated primary hydatid cyst of small intestinal mesentery: An exceptional location of hydatid disease. Pan Afr Med J 2012;13:17.  Back to cited text no. 4
    
5.
Kumar KS. A case of primary peritoneal hydatidosis. Med J Armed Forces India 2009;65:278-9.  Back to cited text no. 5
    
6.
De U. Primary abdominal hydatid cyst presenting in emergency as appendicular mass: A case report. World J Emerg Surg 2009;4:13.  Back to cited text no. 6
    
7.
Ahmed Z, Chhabra S, Massey A, Vij V, Yadav R, Bugalia R, et al. Primary hydatid cyst of pancreas: Case report and review of literature. Int J Surg Case Rep 2016;27:74-7.  Back to cited text no. 7
    
8.
Abdelmaksoud MM, Jamjoom A, Hafez MT. Simultaneous huge splenic and mesenteric hydatid cyst. Case Rep Surg 2020;2020:7050174.  Back to cited text no. 8
    
9.
Rasheed K. Hydatid cyst of liver complicated with budd-Chiari syndrome and portal vein thrombosis. N Am J Med Sci 2013;5:242-4.  Back to cited text no. 9
    
10.
Rexiati M, Mutalifu A, Azhati B, Wang W, Yang H, Sheyhedin I, et al. Diagnosis and surgical treatment of renal hydatid disease: A retrospective analysis of 30 cases. PLoS One 2014;9:e96602.  Back to cited text no. 10
    

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Correspondence Address:
Ravikanth Reddy,
Department of Radiology, St. John's Hospital, Bengaluru - 560 034, Karnataka
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/am.am_50_21



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