METHODS: Medical records of patients who were treated for LM in the Paediatric Surgical Centre Universitas Gadjah Mada from January 2015 to January 2019 were reviewed. Scoring systems were used to assess the outcomes, including reduction of size, problems of aesthetics, functional problems, complications, necessity of further interventions, and interventions' frequencies.
RESULTS: During the four-year study, we included 31 children, consisting of 6, 5, and 20 patients in Groups I, II, and III, respectively. The total score did not significantly differ between Groups I, II, and III (14.67±2.80 vs. 13.40±2.07 vs. 12.50±1.47, respectively; p=0.056). Group II scored better in aesthetic problems than other groups (p=0.001), Group III scored higher in necessity of further interventions compared to the other groups (p=0.026), and Group I was higher in interventions' frequencies than the other groups (p<0.001). However, there were no significant differences in reduction of size, functional problems, and complications among groups (p=0.554, 0.151, and 0.076, respectively).
CONCLUSIONS: There is no significant different effect of the three modalities treatment for LM, although one group might have more beneficial effects compared with the other groups due to different scoring system parameters. Further multicentre and prospective cohort studies with a larger number of patients are necessary to establish the existence and extent of our findings.
PRESENTATION OF CASE: We report the first case of Blue Rubber Bleb Nevus Syndrome in Malaysia, a 23 years old Malay girl who suffers from multiple cutaneous venous malformation and gastrointestinal bleeding episodes.
DISCUSSION: The typical morbidity for this syndrome is symptomatic anemia due to secondary iron deficiency due to the gastrointestinal venous malformation bleeding. In managing the gastrointestinal bleeding, it mainly depends on the severity of gastrointestinal bleeding, some may resolve spontaneously, while the others may be needing blood transfusion, and some may require GIT resections. As for cutaneous lesions, normally it is innocuous depending on the region and size. Large or problematic cutaneous venous malformation might benefit from sclerotherapy or excision.
CONCLUSION: Multidisciplinary approach is crucial in managing BRBNS case due to its complexity and the spectrum of multiple organ involvement to ensure the best outcome to the patient.