In the present study, we first systematically compared the clinical characteristics and prognosis of ML and OL with the largest sample sizes from two centers and found that OL patients were more prone to misdiagnosis and infection before the definite diagnosis. Correspondingly, compared with ML patients, the size of cysts in OL patients was significantly larger, and cysts with turbid fluid were more frequent in OL patients. In addition, our findings demonstrated that laparoscopic surgery could be the preferred surgical treatment for OL patients with a good prognosis.
A total of 148 ML patients and 53 OL patients were evaluated, both groups exhibiting a predominance of males, similar to previous studies [3–5]. Although with the development of prenatal ultrasound, it was possible to indicate excessive peritoneal effusion or abdominal cystic masses in the fetus during pregnancy, it seemed impossible to distinguish ML from OL [20, 21]. Before the cysts developed complications, most ML and OL patients were asymptomatic or had mild symptoms and only received conservative treatments without imaging examinations. Their conditions were often misdiagnosed, sometimes as gastroenteritis [4]. In addition, even after the occurrence of complications, the symptoms of both diseases are various, nonspecific and difficult to diagnose and distinguish [5]. As observed in our study, except for vomiting, the presenting symptoms did not differ significantly between ML and OL patients. The presence of vomiting often indicates bowel obstruction, which might be related to the compression of the intestine by the enlarging cyst or the occurrence of intestinal volvulus [22, 23]. Our previous studies have confirmed a high rate of intestinal volvulus in ML patients [6, 11]. Compared with OL patients, ML patients might have more urgent symptoms, so it is recommended that the imaging examinations be performed as soon as possible. Consequently, ML patients in our study had a lower misdiagnosis rate.
Interestingly, we noted that the laboratory results showed that OL patients had a higher percentage of infection. There are two possible reasons for this phenomenon. First, as observed in our study, more OL patients had hemorrhage of cyst at admission, and hemorrhage could activate the body’s immune system, leading to the mild elevation of inflammatory markers, such as C-reactive protein [24, 25]. Second, cysts with severe hemorrhage might be more susceptible to infection. In adult intracerebral hemorrhage, the size of the hematoma was a predictor of infection development [26]. In ML patients, the size of the cyst has been shown to identify the presence of preoperative hemorrhage or infection. Our results also showed that OL patients had significantly larger cysts than ML patients, and cysts with turbid fluid occurred more frequently in OL patients. However, it was difficult to accurately distinguish between hemorrhage and infection from the cysts’ appearance, so the infection process after hemorrhage could not be further analyzed. In addition, in our study, the presence of C-reactive protein at > 8 mg/L or white blood cells at > 10 × 109/L indicated an infection, possibly leading to some bias in the results.
Previous studies have shown that complete surgical resection is an effective and durable treatment for abdominal lymphatic malformations with a good prognosis [2, 27, 28]. However, for some complex abdominal lymphatic malformations that had multicentric and diffusely infiltrated lymphatic malformations involving important organs, such as type III and type IV ML reported by Kim SH et al., complete surgical resection is very difficult with a high recurrence rate, so medications and sclerotherapy could be the first choice of treatment. However, the long-term prognosis still needs further study [6, 28]. For OL patients, the recommended treatment is complete surgical resection. Since it is challenging to distinguish ML and OL through preoperative imaging examinations, surgery is necessary for patients suspected of the above two diseases [2]. Our study revealed that OL patients had a better surgical prognosis than ML patients, especially those with complete OL excision, none of whom experienced the recurrence of cysts. This may be related to the high proportion (94.3%) of macrocystic-type cyst in OL patients. During the surgical treatment of OL patients with the macrocystic-type cyst, the omentum involved could be boldly removed to reduce recurrence. In the surgical treatment of ML, however, the surgeon should avoid more bowel resection whenever possible, and some small cysts in ML with microcystic-type or mixed cystic-type cyst cannot be identified by the naked eye [9, 28]. Therefore, ML patients are more likely to have a recurrence after surgical treatment.
The last point we have observed is that, compared with ML patients, more OL patients underwent laparoscopic surgery without any definite influencing factor. This would imply that, consistent with previous studies, almost all OL patients were eligible for laparoscopic surgery [10, 15, 29]. Previously reported laparoscopic surgery for abdominal lymphatic malformations included two surgical approaches. One was complete laparoscopic surgery, which is widely used in adult patients with a large abdominal cavity, and the other was laparoscopic-assisted exploration to locate and decompress the cyst of lymphatic malformations first, followed by cyst excision through a slightly expanded umbilical incision, which is more commonly used in pediatric patients [16, 30–32]. The cysts with large size in pediatric patients could impair surgical visualization during laparoscopic surgery. Therefore, according to our study, in the surgical treatment of OL, the experience of the surgeon might be the main factor influencing the specific surgical approaches.
There are some significant limitations to the interpretation of our results. First, due to the retrospective nature of our study, the cyst characteristics of each patient cannot be guaranteed to be entirely correct, as they are summarized from surgical records by two surgeons rather than intraoperative photographs. Although a unified database was designed using Excel software before data collection, this still could induce some bias. In addition, our study did not further analyze surgical options and prognosis of ML and OL among different surgeons, which might be the main factor influencing the surgical methods. Furthermore, due to the lack of complete original images from the two centers, the imaging results of ML and OL in our study were not evaluated for preoperative distinguish, which was the focus of our future research.