Giant benign mesenteric cysts (mesothelioma and lymphangioma): A report of two cases

Abstract

Introduction and importance

Mesenteric cysts are a rare group of intra-abdominal tumors located in the mesentery or omentum, most of this lesion are asymptomatically but occasionally presents with non-specific symptoms, which makes diagnosis difficult.

Case presentation

We present two cases of giant mesenteric cyst in our Institution, the first case is a women of 23 years with previous gynecologic surgery with chronic abdominal pain and large abdominal mass CT showed a giant intraabdominal 30 × 25 × 15 cm, the patient went to laparotomy and a large mesenteric tumor found. The Histopathology reported a Benign cyst mesothelioma (BCM). The second case is a 47 year male with intermittent abdominal pain and abdominal CT shows a 20x21x15 cm tumor. Laparotomy was performed. Histopathology reported a large lymphangioma.

Clinical discussion

BCM tumors are rare intrabdominal lesions often diagnosed in premenopausal women. Its etiopathogenesis is still unclear, associated risk factors are endometriosis, pelvic inflammatory disease and previous pelvic surgery. Lymphangiomas are an infrequent cyst tumors. It results from a failure in the evolutionary development of the lymphatic system, often localized in the head and neck and its localization in the abdomen is even rare. The definitive diagnosis is made by histopathology.

Conclusion

Mesenteric cysts diagnosis if often challenge due to nonspecific symptoms and significant overlap in imaging appearance between the different entities, its diagnosis should be in mind when encounter a cystic abdominal tumor, surgical complete removal of this tumors is the optimal treatment to prevent recurrence and complications.

Highlights

• •Mesenteric cysts are unusual tumors with variably pathogenesis and have multiple differential diagnosis.
• •Usually presents with nonspecific clinic and the diagnosis can be challenging.
• •Images can be helpful describing these tumors, but histopathology is the gold standard for diagnosis.
• •Complete surgical resection is recommended to prevent recurrence and eliminate the potential risk of malignancy.

1. Introduction

Mesenteric cysts are rare intra-abdominal tumors and include a diverse group of histological entities. They occur in adults and children with an incidence of 1/100,000 admissions in adults and 1/20,000 in children [1].

Cystic peritoneal tumors can be localized anywhere through the gastrointestinal tract from the duodenum to the rectum and from the base of the mesentery to the retroperitoneal. This tumors can occur in any part of the mesentery but most frequently originate from the mesentery of the small bowel (ileum: 60 %) and mesocolon (ascending colon: 24 %), retroperitoneum 14.5 %. The etiology of mesenteric cysts is unclear, but a failure of the lymph nodes to communicate with the lymphatic or venous systems or the blockage of the lymphatic system as a result of previous pelvic surgery, trauma, pelvic inflammatory disease, infection, endometriosis, or neoplasia have been suggested as contributing factors [2,3].

Variable, unspecific and indolent symptoms are more frequent in adults while acute abdomen is a typical clinical presentation in children due to malformation such as volvulus, necrosis, and/or perforated bowel [4].

Imaging studies aid in the differential diagnosis, but histopathology remains the diagnostic gold standard. Management is dictated by the terrain, particularly the nature of the tumor, its relation to adjacent anatomical structures, and associated symptoms. Complete surgical resection prevails compared to a conservative approach due to the risk of complications. The choice between open or laparoscopic surgery should be determined based on the perioperative risk [1,5,6].

2. Methodology

This case series include two cases of giant benign mesenteric cyst reported at a second level hospital in the department of surgery at our institution.

This case series has been reported in line with PROCESS criteria [18].

3. Case Report

The First case is a 23-year-old woman with BMI 32.42 kg/m² with background of hypothyroidism in treatment with levothyroxine, surgical record uterine curettage 3 years ago for missed abortion. The patient came to urgent department referring for al least four months ago with abdominal distention, dyspepsia, diffuse abdominal pain mostly referred in right hypochondrium. During her admission in the Hospital her laboratory had Leucocyte 11.57 μ/L, hemoglobin of 14.38 g/dL hematocrit 44.38 % platelets 454 μ/L, TP 11.10 seg, TTP 37.90 seg, INR 1 S/U, fibrinogen 447 mg/dL, CA 125 at 5.50 U/mL, CA 19.9 at 2.30 U/mL.

At physical examination, an abdominal mass was encountered in the right upper quadrant, without clear limit borders, rest of the exploration was normal.

Abdominal-Pelvic CT was obtained an reveled a large cyst displacing right kidney caudally and medially at the level of L5, dimensions are 28 × 25 × 15 cm in its cephalocaudal, transverse and AP axes respectively and a volume of 5775 cc. Uterus and annexes were normal (Fig. 1).

Fig. 1.

Abdominal CT Large benign cyst mesothelioma.

For the CT images a preoperative diagnosis was made for large right hemiabdomen cyst with a suspected diagnosis for large right complex renal cyst.

The patient went to mid-line Laparotomy where was encountered with a giant cyst that displace the intestinal, right kidney and liver approximately 30 × 25 cm dependent of ileum mesentery with no involvement of intestine (Fig. 2, Fig. 3). Complete resection of the cyst was made and punctured, it was obtained 5.5 L of citrine yellow liquid. The cyst was sent to pathology. It reported large benign cyst mesothelioma. Fig. 5 A,B. No further analysis of the cyst was made.

Fig. 2.

A) Large benign cyst Mesothelioma (BCM). B) After extraction of Bening cyst Mesothelioma.

Fig. 3.

A and B Abdominal CT that show an intraabdominal Lymphangioma cyst.

Fig. 5.

Histopathology. First case BMC Images A and B Hematoxylin and eosin staining showing lined by a single layer of cuboidal or flattened mesothelial and some Histiocytic cells of the cyst wall. Second Case Images C and D Hematoxylin and eosin staining showing multiple lymphocyte cells compatible with mesenteric lymphangioma.

The postoperative course was uneventful. At the 6-month follow-up, the patient was completely relieved of pain, there were no signs of recurrence.

3.1. Case 2

A 47 years old male patient with BMI 30.10 kg/m² with no history of comorbidities, presents to the urgent department with abdominal intermittent pain and abdominal tumor that has been increase since the last 6 months. At his admission at the hospital his laboratory had Leucocyte 4.9 μ/L, hemoglobin of 15.68 g/dL hematocrit 47.37 % platelets 329 μ/L, the rest of his laboratory was normal. At physical examination, it was discovered a painful abdominal tumor in the right hemiabdomen that involve costal border and the umbilicus.

A CT scan was obtained and reported a large cystic tumor 20.73 cm × 21 cm × 15 cm, and a volume approximately 3 L that displace the stomach and the intestine (Fig. 3).

For the CT images a preoperative diagnosis was made for a large cyst with suspected omentum origin.

The patient went to mid-line Laparotomy where was encountered a Giant cyst tumor dependent of ileum mesentery that displace the stomach, and the small bowel to the left. The report pathology shows a Lymphangioma cyst (Fig. 5 C, D). No further analysis of the cyst was made (Fig. 4).

Fig. 4.

A and B Intraoperative Giant Lymphangioma cyst.

The patient follow up at 6 months show no recurrence of the tumor.

4. Discussion

Mesenteric cysts are a heterogeneous group of intra-abdominal cystic lesions located in the mesentery or omentum that may occur in any portion of the GI tract, from the duodenum to the rectum. Most frequently cysts are localized in small bowel mesentery (ileum in 60 %) and mesocolon (ascending colon in 24 %), retroperitoneum 14.5 % [3,7,8].

The most recent classification by Ros et al. [9] and developed by De Perrot et al. [1] distinguishes according to tissue of origin including (a) lymphatic (simple and lymphangioma); (b) mesothelial (simple, benign and malignant cystic mesothelioma); (c) enteric (duplication and enteric cyst); (d) urogenital; (e) mature cystic teratoma; and (f) non-pancreatic pseudocysts (traumatic and infective) [10].

The only genuine malignant tumor in this classification is malignant cystic mesothelioma which may, simulate the gross appearance of benign cystic mesothelioma and therefore lead to misdiagnosis [9].

4.1. Benign cyst mesothelioma (BCM)

BMC is known by various names in the literature, including peritoneal inclusion cyst, multilocular inclusion cyst, multicystic mesothelioma, and benign multicystic mesothelioma, reflecting the uncertainties around lesion pathogenesis [15].

This tumor occurs mainly in young to middle-aged women at an average age of 37 years. The lesion is usually described as a benign tumor with a low risk of malignant transformation. The prevailing theory suggests that Bening cyst mesothelioma may be a peritoneal reaction secondary to a chronic irritant with mesothelial cell entrapment, reactive proliferation, and cystic formation. Chronic inflammation, previous surgery procedures, endometriosis recurrent peritonitis episodes associated with peritoneal dialysis, are described in the literature as predisposing factors of this tumor [16].

Histologically, bening cyst mesothelioma demonstrates numerous small cystic spaces lined by a single layer of cuboidal or flattened mesothelial cells. The cysts are variably sized and often contain pale eosinophilic proteinaceous fluid [17].

Lymphangiomas are benign tumors, probably congenital, are more common in the cervical (75 % and axillary regions (25 %). They are unusual in abdominal and pancreas location less than 1 %. Its incidence is estimated at around 1:100,000 and 1:20,000 admissions in adults and in children. These lymphatic tumors are divided in: 1) simple, with capillary lymphatic channels; 2) cavernous, with dilated lymphatics and the presence of capsule; and 3) macrocystic malformations, clinically termed “cystic hygroma”. This is the most common type, and the most affected sites are head and neck [11,12].

The disorder is characterized by a thin and irregular cyst wall with multiple internal septa, composed of endothelial cells, smooth muscle cells, foam cells, and lymphoid tissue [14].

The clinical presentation of mesenteric cysts is dependent of the size of the cyst, its location, and the presence or absence of complications. Patients can present in one of three ways: (a) asymptomatic: these patients are picked up incidentally on routine physical examination, during abdominal surgery, or routine imaging as seen in one of our patients. Approximately 40–45 % of patients present this way; (b) nonspecific abdominal complaints: which include pain and distension, occasionally associated with nausea and vomiting, diarrhea, constipation, and loss of weight; and (c) acute abdomen: due to complications of the cyst and can occur in approximately one-third of patients. Some of these complications include intestinal obstruction, volvulus, hemorrhage (secondary to trauma and erosion), infection, or cyst rupture [13].

Radiologic methods for diagnosis and investigation include ultrasound, which can define the cystic nature of the lesion and identify solid components, debris and septations within the lesion, Computed tomography is the most used form of investigation providing good anatomical detail and, with the aid of contrast, may provide information on relations of the mass to vascular and other structures [10].

The BMC lesions MRI is considered the best imaging technique for demonstrating the anatomic relation of the mass with the pelvic structures. At MRI, the cysts have high T2-weighted and low T1-weighted signal intensity, unless hemorrhage is present, which could result in higher signal intensity on pre contrast T1-weighted images. In the case of lymphangiomas the MRI show the multiseptated cystic appearance, which typically has high T2-weighted and low T1-weighted signal intensity [15].

The laboratory investigation does not usually yield any significant information. In addition, it is rarely necessary to perform additional diagnostic procedures that may further characterize the cyst, such as fine needle aspiration with cytological analysis or explorative laparoscopy [4,8].

The first-choice treatment is complete excision via surgery. Laparotomy or laparoscopy might be used to remove the cyst. Simple aspiration or marsupialization may be considered when short bowel syndrome is predicted but is typically not advised due to infection and frequent recurrence. If enucleation cannot be performed safely due to the cyst wall adhering to the mesenteric tissue and other tissues nearby, a resection of the neighboring organ may be necessary [10,13].

5. Conclusion

Mesenteryc cyst are a rare benign neoplasm that is associated with no well-defined symptoms, typical clinical, or imaging data. Consequently, the diagnosis remains unclear and the definitive diagnosis requires histology. The treatment is complete surgical removal of the tumor to prevent further recurrence.

Author contribution

Jesús Eduardo Prior-Rosas: Investigation, Methodology, Supervision, Writing paper.

Brenda Mejía-Ruíz: Writing-original Draft, Methodology.

Brenda Areli Magdaleno-Becerra: Investigation, Conceptualization.

Chantal Guadalupe Nava-Tenorio: Visualization, Investigation.

Sheila Marilyn Alonso-Domínguez: Validation, Supervision.

Gabriela Eugenia Botello-Ortiz: Supervision, Investigation.

Consent

Written informed consent was obtained from the patients for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Ethical approval

This report was prepared following the ethical standards of the institutional ethics committee and with the 1964 Helsinski Declaration. This case of series is not containing any personal information. Our institution does not require ethical approval for reporting individual cases or case series. Mexican Social Security Institute.

Guarantor

Dr. Jesús Eduardo Prior Rosas.

Research registration number

Not applied.

Funding

This study did not receive any funding.

Conflict of interest statement

The authors declare that they have no conflict of interests.