Intra-Abdominal Multi Cystic Lymphangiomas: A Case Series with Adult and Pediatric Literature Review

Background: Multi-cystic Lymphangioma (MCL) is a rare intra-abdominal entity in adults. The diagnosis of a core biopsy specimen is challenging due to its rarity in general surgical pathology service. Invasive surgical management can be avoided with accurate core biopsy diagnosis.

Methods: We present six cases of adult intra-abdominal MCL and highlight their clinical, radiological, and pathological characteristics. Demographic, clinical, radiological, and histopathological parameters were collated for analysis.

Results: Six patients (out of 958,574 cases) were identified (prevalence: 0.6/100,000 cases). Previous abdominal surgery (67%, n = 4) and abdominal pain with palpable abdominal mass (67%, n = 4) at presentation were notable in this cohort. Abdominal CT showed cystic mass with septations, punctate calcification, and fatty halos raising the differential diagnosis of mesenteric cyst (n = 2), duplication cyst (n = 2), or disseminated metastases (n = 2). MCL was distributed across the jejunal mesentery (n = 3) and was managed with exploratory laparotomy and resection (n = 4). Characteristic histopathological features include multiple variably sized cystic spaces lined by attenuated flattened epithelium, cysts filled with proteinaceous fluid, and interspersed by stroma with several dense lymphoid aggregates. The lymphatic endothelium showed positivity for D2-40 and CD31. Post-operative follow-up at 50.4 ± 49.2 months did not show any clinical or radiological recurrence.

Conclusion: Adult intra-abdominal MCL are rare and radiologically indistinguishable from other intra-abdominal lesions. Diagnostic uncertainty on core biopsy evaluation in our series required invasive surgical exploration. The recognition of the histological triad of lymphoendothelial cysts, smooth-muscle or fibrous stroma, and associated lymphoid aggregates are diagnostic for MCL. Cystectomy alone is curative (144 months without recurrent).

Introduction

Lymphangiomas are designated as lymphatic malformations due to their morphological appearance of haphazard dilatations of lymphatic channels [1]. These lymphangiomas can present in isolation as either single cysts (either with one large cavity or with multiple septae causing a multicystic appearance) or as multiple congruent cysts. Rarely, these lymphangiomas can present as lymphangiomatosis when there are multifocal cystic masses distributed across a broad anatomical region. Lymphangiomas are predominantly seen in the cervical or axillary regions (95%) and the remaining 5% are found in the abdomen (mesentery, retroperitoneum, abdominal organs) and thoracic (lungs and mediastinum) compartments [2,3].

Intra-abdominal multi-cystic lymphangiomas (MCL) are rare and represent less than 5% of all lymphangiomas [4,5]. These are seen in both [6] adults (incidence = 1:175,000) [7-9] and pediatric (incidence = 1:20,000) [7,10] cohorts. Congenital-type pediatric MCL cases are often diagnosed early in life and are linked to developmental errors resulting in lymphatic malformation. Contrastingly, adult MCL cases are extremely rare and suspected to be associated with trauma, previous intra-abdominal surgery, inflammation, or radiation exposure rather than solely congenital. Both the adult and pediatric MCL are considered benign entities, and malignant transformation has not been reported in the scientific literature to date. The rarity of the adult MCL in the general surgical pathology service makes them diagnostically challenging, especially on a core biopsy. Clinically, these often present with abdominal distension [11,12], abdominal pain [13,14], large sized lesions, and most patients do not have co-morbidities. The larger size at presentation can be attributed to the insidious growth pattern of MCL combined with the potential volumetric capacitance within the abdominal cavity.

Radiologically, adult MCLs are seen as anechoic cysts by ultrasonography and as cystic masses with multiple septae by Computer Tomography (CT) [15]. However, pre-operative diagnosis is made in only a minority of patients [16]. These radiological findings are nonspecific and hence it cannot definitively distinguish MCL from the vast differential diagnosis for intra-abdominal cyst (Table 1). Therefore, the definitive diagnosis hinges on the identification of histopathologic features such as dilated spaces lined by lymphoendothelium showing immunohistochemical positivity for D2-40, CD31, and CD34 [17-19]. Laparotomy and segmental enterectomy is the most commonly employed surgical strategy [20] compared to isolated cystectomy [21]. Either strategy results in adequate gross total resection without recurrence [6,16,22] but the invasive surgical option can result in complications such as short gut syndrome [23] Figure 1.

Figures 1: Flowchart summarizing the differential diagnosis of intra-abdominal multicystic lesions.

The study aims to report the clinical, radiological, and pathologic features of six cases of adult intra-abdominal MCL and describe their integrated diagnostic insights through a comprehensive literature review. An additional objective is to highlight this rare entity for clinicians and surgical pathologists to enhance and increase the index of suspicion for an accurate diagnosis on core biopsy evaluation.

Case series

An index case of MCL was identified in 2024 which led to a retrospective search for intra-abdominal mesenteric lymphangioma in our laboratory information system since 2000 [24 years]. A total of 958,574 cases were searched which yielded 6 adult patients with MCL. The prevalence of MCL at our institution was estimated at 0.6 per 100,000 cases. Table 1 summarizes the patient demographics, clinical presentation, radiological findings, operative approach, histopathologic features, and clinical follow-up in these 6 patients with MCL. In this cohort, the median age at diagnosis was 58.8 ± 14.6 years (F: M=1:1) and the predominant clinical presentation was abdominal pain with palpable abdominal mass (67%, n = 4). Figure 2 (A-F) illustrates the key radiological image in each of the six cases of MCL. Abdominal CT imaging showed a cystic lesion with thin septation (100%, n = 6) with punctate calcification (50%, n = 3) and fatty halos (33.3%, n = 2). Radiologic differential diagnosis included mesenteric cyst, duplication cyst, lymphoma, disseminated peritoneal carcinomatosis, and advanced mesenteric panniculitis. MCLs were predominantly located in the jejunal mesentery (50%, n = 3), and were managed with exploratory laparotomy (66.7%, n = 4) with cystectomy (66.7%, n = 4).

Figures 2: Illustrates the key radiological image in each of the six cases of MCL. A: Case 1- Axial view of contrast-enhanced CT abdomen and pelvis (CTAP) which shows a large hyperdense central mesenteric mass (red arrow) with associated calcification and enlarged nodes. B: Case 2- Sagittal view of contrast-enhanced CTAP which shows a cystic lesion with rim calcification (red arrow) and no cleavage plane with the adjacent duodenum. C: Case 3- Axial view of contrast-enhanced CTAP which shows a minimally complex cystic lesion (red arrow) with a well-circumscribed thin wall, septations, minimal punctate calcifications, and layering debris (proteinaceous or hemorrhagic material). D: Case 4- Axial view of contrast-enhanced CTAP which shows a large mixed-density cystic mass with partial calcification (red arrow). E: Case 5- Coronal view of contrast-enhanced CTAP which shows a peripherally enhancing retroperitoneal cystic mass with coarse calcifications (red arrow). F: Case 6- Coronal view of gadolinium-enhanced MR AP which shows cystic retroperitoneal collection with lobulated margins, and few internal septations.

All 6 cases showed similar histologic findings. Figure 3A shows the masses being composed of variable-sized dilated multicystic spaces [*] which are lined by flattened endothelium. The intervening stroma contains multiple lymphoid aggregates [#] (Figure 3B) and collagenous fibers intermixed with smooth muscle fibers [+] (Figure 3C). There are histologic changes of cyst rupture as seen in Figure 3D by the presence of calcification [^], histiocytes and cholesterol clefts [//], and chronic inflammation in Figure 3E. One of the 6 cases from our series showed an MCL involving the submucosa [|||] of the small intestine as seen in Figure 3F.

Figures 3: Characteristic microscopic findings of multicystic lymphangioma on hematoxylin and eosin. MCL consists of A: variably sized cyst lined by endothelium [*] and B: lymphoid aggregates [#] with C: intervening stroma made from smooth muscle fibers [+]. MCL can also have D: cyst rupture with calcifications [^] and histiocyte infiltrate. E: Cyst rupture with cholesterol clefts [//] and chronic inflammation. F: MCL [|||] extending into the small intestine submucosa.

The immunohistochemistry (IHC) stains for D2-40 and CD31 highlight the cyst lymphatic endothelium lining (Figure 4A and 4B). The intervening stroma shows smooth muscle fibers which stain with SMA and Desmin immunohistochemical antibodies. (Figures 4C,D). There was no expression of Calretinin and HMB45. The integrated diagnosis based on histology and IHC profile confirms multi-cystic lymphangioma. All 6 patients in the case series remained asymptomatic from MCL recurrence during their follow-up period.

Figures 4: IHC results for MCL. (A) cyst lined by D2-40 (red arrow) and (B) CD31 positive lymphatic endothelium (red arrow). (C) SMA and (D) Desmin highlight smooth muscle fibers (red arrow) within the MCL stroma.

Discussion

Adult intra-abdominal MCL is a rare diagnostic entity in the gastrointestinal clinical and pathology service. A literature review yielded 154 articles in the English language describing intra-abdominal MCL in 118 adult cases [5,8,9,13,16,18-21,24-109] and 133 pediatric cases [5,7,11,12,14,17,23,110-162] indexed in Medline database. It is also notable that the majority were from the United States (n = 61), while the remaining were reported from South Korea (n = 32), France (n = 23), the United Kingdom (n = 20), and Japan (n = 17). We present the second case series on adult intra-abdominal MCL from Canada. Table 2 summarizes the salient clinical, radiological, and pathologic features in adult and pediatric populations. Supplemental Tables S1.1 - S2.2 show the comprehensive clinical, radiological, and pathologic features dataset for adult and pediatric MCL cases respectively.

Our literature review showed that intra-abdominal MCL was identified in both adults (mean: 40.36 years [range: 18-82]) and children (mean: 5.57 years [range: 0-17]). Overall, there was a slight male preponderance; however, in adult MCL cases there was a slight female predilection (M: F ratio = 1:1.2). Contrastingly, the pediatric cohorts showed a male predilection (M: F ratio 1.7:1). Lymphangiomas are considered malformations or hamartomas, and not true neoplasms. This embryonal malformation theory is partially supported by the fact that most cases are diagnosed during early childhood and a large number are diagnosed prenatally [163,164]. Genetic factors are considered to play a role in the pathogenesis of this entity including somatic mutations [165], the role of PIK3CA mutations [166], and in hereditary lesions the role of VEGFR-3 [167] encoding genes is implicated. Other possible etiologies include a traumatic origin resulting in bleeding or inflammation in the lymphatic channels, both leading to obstruction and subsequent secondary lymphangiomatous formation [168-170].

All six of our patients were symptomatic, which is in keeping with the overall finding of symptomatic clinical presentation in adults (85.59%) and children (72.18%) MCL in the literature. Abdominal pain is the most common presenting feature, followed by abdominal distension. Abdominal pain in the pediatric cohort is often described as more severe [115] and may be associated with a more acute presentation [7,140,171,172], especially if associated with a rapidly growing mass [140]. Contrastingly, adult patients with MCL present with a chronic pain of more than a few days/weeks duration [171]. Other findings at presentation include nausea and vomiting [30], fever, and features of co-morbidities if present, such as those of anemia [48]. Often a palpable mass is identified, usually related to the location of the lesion [110].

In our case series majority of the adult MCL cases showed multiple septated cysts and calcifications. Abdominal ultrasound and CT are common initial radiologic investigations in these patients. Ultrasonography is very sensitive and relatively specific for the evaluation of abdominal cystic masses, as shown in one study where the sensitivity was 87.9% and specificity 81.8% [173]. The lesions appear as sharply defined cystic or multicystic formations, often with internal septations [2,174,175]. CT findings for MCL demonstrated a predominant homogenous cystic lesion but fluid, blood, or fat may lead to heterogenous imaging findings [176]. Our literature review indicated that the most common finding was intra-abdominal multiloculated cysts (n = 29, 24.58%). Interestingly, multiloculation as a radiological feature was more frequently identified in adults compared to pediatric patients (47.5% vs. 23.3%, p - value <0.0001). This higher incidence could be from a combination of developmental progression, later diagnosis, radiologic detection sensitivity in ultrasound versus CT, and secondary complications (cyst infection, trauma or hemorrhage). Magnetic resonance imaging (MRI) has been shown to be more helpful because of its high resolution and its ability to delineate cystic and septal structures [22]. However, the usefulness of radiologic investigations for accurate preoperative diagnosis is limited [16] due to the overlap of findings with other cystic lesions in the abdomen [122,177-179] leading to a range of differential radiological diagnoses including disseminated carcinomatosis in adults.

Lymphangiomas are subdivided into three main types: capillary, cavernous, and cystic. The first two are predominantly cutaneous lesions; cystic lymphangiomas are generally found in the abdomen and retroperitoneum. All six of our cases were intra-abdominal MCLs. The literature identifies that most intra-abdominal MCLs are seen in the small bowel mesentery proper (jejunum followed by ileum, or unspecified location of the small intestine) and then in the colonic region (Figure 5). Overall, adult and pediatric MCLs can occasionally present with complications such as mesenteric vessel dilatation (n = 13, 5%) or bowel obstruction (n = 30, 10.7%) due to volvulus, ischemia, malrotation, internal hernia, and/or intussusception.

Figures 5: Summary of the anatomic distribution of intra-abdominal multi-cystic lymphangiomas within our literature review (154 articles).

Macroscopically the size of the intra-abdominal MCLs in our case series was approximately 5.6 ± 3.4 cm and were predominantly hemorrhagic (n = 2) in cyst content. Contrastingly, the average cyst size reported in our literature review was 13.7 cm in adults and 12.8 cm in the pediatric cohort. Overall, most cysts have a chylous fluid (n = 32, 26.3%), followed by serous fluid (n = 18, 15%), and hemorrhagic fluid in others (n = 9, 7.5%). Chylous fluid is milky because of the fluid’s abundant fat content, while the serous fluid is clear, and straw-colored in appearance. This variability in cyst fluid composition may be multifactorial and related to variable lymph stasis, the number of lymphatic communicating channels, and their fluid protein content [175,180,181]. Interestingly, most intra-abdominal MCLs in the literature were reported to be intact at surgery (n = 174, 98%) with a minority being ruptured (n = 5, 2%). Moreover, the chylous fluid was more frequently identified at grossing in adult MCL compared to pediatric MCL (37.3% vs. 16.5%, p - value <0.0001). This higher incidence could be from a multitude of factors. One such possibility is that in adults these lymphangiomas may have more established connections to other lymphatic channels and the thoracic duct resulting in leakage of chyle into these cystic spaces.

Our cases had endothelium-lined variable-size channels, smooth muscle fibers, and lymphoid aggregates in the wall, and some cases especially post-biopsy resected specimens had microcalcifications with inflammatory infiltrates and cholesterol clefts. The endothelium showed immunohistochemical positivity for D2-40 and CD31 and was negative for mesothelial immunohistochemical markers such as calretinin and WT1. Smooth muscle fibers were positive for SMA and Desmin. The histopathological triad for MCLs that were consistently identified in our case series and the literature included dilated lymphatic channels (n = 242, 96.4%), fibro-collagenous stroma with smooth muscle fibers (n = 175, 69.7%), and lymphoid follicles (n = 119, 47.4%). In addition, scattered chronic inflammatory infiltrate (n = 62, 24.7%), microcalcifications (n = 11, 4.4%), and cholesterol clefts (n = 28, 11.1%) have also been reported and some were concerning previous fine needle or core biopsy. Microcalcifications were associated with chylous or hemorrhagic contents and were seen especially in lesions located mostly in the jejunum [34,49].

The immunohistochemical profile is essential in diagnosing MCL from its differential diagnosis; lymphoma, adenocarcinoma, and hemangioma if there is secondary bleeding into the abdominal cavity. In our case series, the core biopsy on Case 1 raised the possibility of angiomyolipoma due to the histologic findings of vessels, muscle, and fat. It is important to consider the location of angiomyolipoma and IHC profile to rule out this differential. MCLs usually express markers seen in the lymphatic lining (D2-40, CD31, and CD34) [17-19]. The absence of CD34 [55,70] and factor VIII-related antigen (FVIII-RAG) [64] in the cyst lining has been documented in rare cases but these could be attributed to underlying preanalytical methodological issues. Similarly for other differential diagnoses, specific markers will help with the diagnosis e.g., mesothelioma will have positivity for calretinin, lymphoma for hematolymphoid markers, and adenocarcinoma for epithelial markers. A summary of the IHC findings in the adult and pediatric intra-abdominal MCLs in the literature review is shown in Figure 6.

Figures 6: Summary of positive (A) and negative (B) IHC findings for the cyst lining in the overall (adult and pediatric), adult-only, and pediatric-only intra-abdominal MCL cohort within the literature review.

The extent of surgical resection of intra-abdominal MCLs is dependent on the observation that these tend to involve the surrounding areas and therefore may potentially recur if incompletely excised [174,182]. Common surgical modalities include laparotomy with segmental enterectomy or laparotomy with cystectomy [171,183]. Adjunct surgical procedures after laparotomy are rare, but these depend on the location and extent of the lesion e.g. Whipple’s [51], splenectomy [146], and cholecystectomy [100]. Laparoscopic removal is a feasible option in children [184]. Laparoscopic removal of intra-abdominal MCLs has certain advantages over classical laparotomy, including a more precise dissection around the mesenteric vessels, minimal trauma to the abdominal structures, less pain, and a shorter hospital stay. More experience is needed to demonstrate the superiority of this method over conventional open techniques [185]. Interval elective surgery is recommended in patients with the asymptomatic presentation with documented interval regression [28]. Notably, laparoscopic cystectomy was more frequently performed in adult MCL compared to pediatric MCL (51.9% vs. 30.1%, p -value < 0.0001). Adult MCL tends to be more localized, allowing for complete cyst removal without bowel resection. Furthermore, segmentectomy has higher risks and functional consequences in adults, making cystectomy preferable. Lastly, pediatric surgical practice tends to have more experience with segmental resections rather than cystectomy.

Follow-up was reported in 153 patients (65 adults and 88 pediatric) in the literature; of these, 147 patients were reported as stable (average: 14 months for adults, 23 months for pediatric patients). Most cases do not have recurrences and remain stable [6,16,22,171,186]. Macroscopically complete resection, compared to other procedures, has the lowest rate of recurrence [4]. Patients with limited surgical resection were also noted to be stable in the literature. In one pediatric case series, a high rate of complications, including infections (approximately 6.6%) required additional interventional surgical procedures [182].

Adverse outcomes on follow-up in the literature have been observed only in 6 cases in the literature review. In one adult case, there was a small region of recurrence or possible residual lymphangioma noted on MRI at 6 months post-resection [20]. One pediatric patient had initial progression on ultrasonography at 6 months following incomplete surgical resection due to the risk of short bowel syndrome and proximity of the superior mesenteric artery [17]. It is possible that a combination of the findings of rupture, large size, multicystic nature, fibrous adhesions, and microcalcifications might be related to recurrence and progression [17,20]. The remaining four cases involved short gut syndrome in one child [23] and deaths in three children [23,127,142]. Notably, one adult patient was diagnosed with a large unresectable mass and was treated with the mammalian target of rapamycin (mTOR) inhibitor everolimus (activation of the mTOR pathway was reported at the pathological and transcriptomic levels). This patient eventually had a successful resection of the residual tumor following a major partial response to everolimus [32].

In the literature, the clinical presentation, radiologic findings, cyst size, cyst contents, histologic findings, immunohistochemical features, and follow-up were similar in adults and children. The only notable difference was slight female preponderance in adults and the reverse in children. Given these similarities, the best approach to diagnosis in both age groups is a biopsy and the use of histology and immunohistochemistry to demonstrate lymphatic endothelial lined cysts (D2-40 and CD31) for diagnosis. A minimally invasive surgical procedure appears to be adequate in most cases as the patients are stable on follow-up for both extensive and minimal surgery. A limitation of this literature review is that not all relevant findings such as clinical presentation, radiologic findings, cyst size, detailed gross, and histologic and immunohistochemical descriptions were reported in the published individual case reports and case series.

Conclusion

Adult intra-abdominal MCL is a rare entity that is clinically and radiologically indistinguishable from other intra-abdominal lesions. Due to diagnostic uncertainty on core biopsy evaluation, all cases in our series required invasive surgical exploration. The recognition of the histological triad of lymphoendothelial cysts, smooth-muscle or fibrous stroma, and associated lymphoid aggregates is essential to making a diagnosis of MCL. Our cohort shows that limited surgical intervention such as cystectomy without resection of adjacent organs is curative (144 months without recurrent).

Ethical approval

Standard ethical approval [E-Bio-021] has been collected and preserved by the author(s).

Author contribution

All authors contributed significantly towards this manuscript and have read and approved the final version of the manuscript.

Supplementary

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