Surgical Treatment of Peripheral Vascular Malformations: A Systematic Review and Meta-Analysis.

BACKGROUND: Surgical treatment of peripheral vascular malformations is widely performed as primary and secondary treatments. Excellent results have been reported; however, it is thought that complications are likely to occur because of damage to adjacent structures. This systematic review aimed to elucidate the indications and outcomes of surgical treatment of vascular malformations. METHODS: PubMed, EMBASE, and the Cochrane Central Register of Controlled Trials were searched for studies reporting outcomes of surgery in at least 15 patients with a single type of peripheral soft-tissue vascular malformation. The authors extracted data on patient and lesion characteristics, treatment characteristics, and outcomes (including complications). Meta-analysis was conducted on recurrence and complication rates. RESULTS: A total of 3042 articles were found, of which 24 were included: nine studies on arteriovenous malformations, seven on venous malformations, and eight on lymphatic malformations, totaling 980 patients. Meta-analyses showed pooled proportions for recurrences of 11 percent in arteriovenous malformations, 5 percent in venous malformations, and 9 percent in lymphatic malformations. Pooled proportions of major complications were 9 percent for arteriovenous malformations, 3 percent for venous malformations, and 1 percent for lymphatic malformations. The authors found a 5 percent pooled recurrence proportion in total resections, compared with 28 percent in subtotal resections. The pooled odds ratio for recurrence in total and subtotal resections showed a significant lower recurrence rate after total resection (odds ratio, 0.14, p = 0.02). CONCLUSIONS: Surgical treatment of vascular malformations appears to be effective and safe in many cases. However, it seems that surgery is performed predominantly in small lesions, and subtotal resection has a higher risk of recurrence than total resection.

Electrosclerotherapy as a Novel Treatment Option for Hypertrophic Capillary Malformations: A Randomized Controlled Pilot Trial.

BACKGROUND: Bleomycin sclerotherapy is ineffective for treating capillary malformations (CMs) because bleomycin cannot adequately be injected into the small-diameter capillary lumina. Electrosclerotherapy (EST) might be a new treatment modality for CMs, as it combines bleomycin sclerotherapy and "electroporation"-an electric field applied to the tissue. Electroporation disrupts the transmembrane potential, facilitating bleomycin transportation across the vessel wall, hypothetically leading to targeted drug delivery and increased effectiveness of bleomycin in CMs. OBJECTIVE: To explore the efficacy, safety, and feasibility of EST for CMs in a randomized within-patient controlled pilot study. MATERIALS AND METHODS: Fifteen regions of interest (ROI) within the hypertrophic CMs of 5 patients were randomly allocated to EST, bleomycin injection, or no treatment. Outcome was assessed after 7 weeks by the patient and a blinded outcome assessor using the patient-observer scar assessment score (POSAS), global assessment of change (GAC), colorimetry, and laser speckle contrast imaging. RESULTS: Color and hypertrophy of all ROIs treated with EST significantly improved, based on the POSAS (medians patient -11; observer -13), GAC, and colorimetry (ΔE 3.4-16.5) scores. CONCLUSION: This pilot study demonstrates the first proof of concept for electrosclerotherapy as a new treatment modality for CMs. Further research is warranted.

Oral sildenafil as a treatment option for lymphatic malformations in PIK3CA-related tissue overgrowth syndromes.

Patients with extensive lymphatic malformations associated with tissue overgrowth syndromes (such as Klippel-Trenaunay syndrome and CLOVES) often pose a therapeutic challenge for physicians. In recent years, it has been suggested that oral sildenafil therapy might be used to treat congenital lymphatic malformations. However, this possible new therapy has not yet been used in patients with lymphatic malformations associated with tissue overgrowth syndromes. A 30-year-old man with extensive capillary-lymphatic malformations of the right leg and thorax, and a tissue overgrowth syndrome caused by a somatic mutation in the PIK3CA gene, was treated with oral sildenafil due to symptoms of pain, dyspnea, and functional impairment. Several weeks after the start of the treatment, the patient reported softening of the lymphatic malformation and a significant improvement of his symptoms and physical condition. So far, sildenafil is still considered a last resort in the treatment of complex treatment-resistant lymphatic malformations. With this case report, we demonstrate that sildenafil could also be an alternative treatment option for lymphatic malformations in patients with syndromes belonging to the PIK3CA-related overgrowth spectrum.

Pathological findings in gynecomastia: analysis of 5113 breasts.

OBJECTIVE: This study aimed to analyze the histopathology results of surgically excised breast specimens with the diagnosis of gynecomastia (GM). SUMMARY BACKGROUND DATA: Gynecomastia is a term used to describe benign hypertrophy of the breast in men; it is a common, mostly transient, phenomenon in adolescents, but may also be seen in older men. Breast enlargement can lead to psychological problems; if it persists it can be surgically corrected. The obtained breast tissue specimens are routinely submitted for pathological examination. We performed this study to assess the prevalence of pathological findings after surgical management of GM. METHODS: Pathology reports were obtained from the nationwide network and registry of histopathology and cytopathology in the Netherlands (PALGA). The reports of 5113 breasts were analyzed for the prevalence of pathologies in different age groups. RESULTS: The average age of the patients was 35.3 ± 18.3 years (range, 1-88 years). The most common finding was GM followed by pseudo-GM. The overall prevalence of invasive carcinomas was 0.11% and of in situ carcinomas was 0.18%. The youngest patient with invasive cancer was 65 years old and the youngest patient with carcinoma in situ was 24 years old. The overall prevalence of atypical ductal hyperplasia was 0.4%; in patients younger than 20 years, it was 0.23%. The youngest patient with atypical ductal hyperplasia was 16 years old. Pathological findings were found more often in unilateral procedures. CONCLUSIONS: The prevalence of malignancies in GM resection specimens is low; however, it increases with patient age. Unilateral cases have a statistically nonsignificant higher prevalence of pathologies. LEVEL OF EVIDENCE: Prognostic/risk II.