Pham Hien Duy, Tran Duy Le Dinh, Dang Thom Hoang, Nguyen Luan Vo Mac, Dang Trang Thu, Nguyen Linh Vu Thuy, Nguyen Quang Thanh
Department of Pediatric Surgery, National Hospital of Pediatrics, Hanoi, Vietnam.
College of Health Science, Vin University, Hanoi, Vietnam.
Front Med (Lausanne). 2024 Oct 21;11:1453312. doi: 10.3389/fmed.2024.1453312. eCollection 2024.
Vascular malformations, including lymphatic-venous malformations (LVMs), are complex and can lead to significant morbidity. This case report details the management of a rare multifocal, combined LVM in a 3-month-old male, misdiagnosed as a right inguinal hernia prenatally. Postnatal imaging revealed multiloculated cystic masses extending from the mons pubis to the right groin, scrotum, and thigh. Doppler ultrasound and MRI demonstrated the extent of the masses, with the largest cyst measuring 4 × 4 × 2 cm. Initial surgical excision and debulking were performed; however, the lesion recurred 6 months later. Given the complexity and involvement of truncal vessels, complete excision was not feasible, and sclerotherapy with intralesional Bleomycin injections was initiated. After the first Bleomycin injection, a significant reduction in lesion size was observed. However, 6 months later, the lesion recurred and enlarged, necessitating a second Bleomycin injection. Subsequent follow-ups showed further reduction in lesion size. Unfortunately, 2 years later, the patient experienced diffuse cystic infiltration of the pelvis and right lower extremity due to treatment delays caused by the COVID-19 pandemic. Imaging at this stage revealed a cystic mass in the right pelvic cavity measuring 10 × 6 × 5 cm. Two additional sessions of Bleomycin injections were performed, resulting in a successful resolution of the cystic masses. Regular follow-ups have shown that the LVMs remain under control, with no symptoms and no concerns from the family. This case underscores the complexities involved in diagnosing and managing multifocal, combined LVMs. It suggests that Bleomycin sclerotherapy can be a valuable, minimally invasive alternative to more extensive surgical procedures, particularly when critical structures are involved. The patient's condition has been successfully managed with a combination of surgical and sclerotherapy interventions, ultimately resulting in symptomatic relief and cosmetic improvement.
血管畸形,包括淋巴管静脉畸形(LVMs),情况复杂,可导致严重的发病情况。本病例报告详细介绍了一名3个月大男婴罕见的多灶性、复合型LVM的治疗过程,该患儿在产前被误诊为右侧腹股沟疝。产后影像学检查显示,多房性囊性肿块从耻骨联合延伸至右侧腹股沟、阴囊和大腿。多普勒超声和磁共振成像(MRI)显示了肿块的范围,最大囊肿尺寸为4×4×2厘米。最初进行了手术切除和减瘤;然而,病变在6个月后复发。鉴于病变累及主干血管且情况复杂,完全切除不可行,遂开始采用病灶内注射博来霉素进行硬化治疗。首次注射博来霉素后,观察到病变大小显著缩小。然而,6个月后,病变复发并增大,需要再次注射博来霉素。随后的随访显示病变大小进一步缩小。不幸的是,2年后,由于新冠疫情导致治疗延误,患者出现盆腔和右下肢弥漫性囊性浸润。此时的影像学检查显示右侧盆腔有一个10×6×5厘米的囊性肿块。又进行了两次博来霉素注射,成功消除了囊性肿块。定期随访显示LVMs得到控制,无任何症状,家属也无担忧。本病例强调了诊断和治疗多灶性、复合型LVMs所涉及的复杂性。这表明博来霉素硬化治疗对于更广泛的外科手术而言可能是一种有价值的、微创的替代方法,尤其是在涉及关键结构时。通过手术和硬化治疗相结合的干预措施,成功控制了患者的病情,最终缓解了症状并改善了外观。