Interventional Therapy for a Child With Tracheal Lobular Capillary Hemangioma: A Case Report and Literature Review

Tracheal lobular capillary hemangioma (TLCH), a rare vascular tumor histologically defined by lobular aggregates of capillary-sized vessels within vascularized granulation tissue [1, 2], poses significant diagnostic and therapeutic challenges. Clinically, TLCH manifests with nonspecific respiratory symptoms—including cough, wheezing, hemoptysis [3], and progressive airway obstruction—that overlap with common pulmonary disorders, frequently leading to misdiagnosis. The condition's extreme rarity, particularly in pediatric populations, further complicates timely recognition. Diagnosis currently depends on imaging examination (e.g., computed tomography [CT]) and bronchoscopic evaluation, confirmed by histopathology. While bronchoscopic intervention remains the primary treatment, standardized protocols are lacking due to limited pediatric case reports (Table 1). We present the first documented pediatric TLCH case successfully managed through a staged bronchoscopic approach: preoperative bronchial artery embolization followed by sequential snare polypectomy, biopsy forceps resection, Neodymium-doped Yttrium Aluminum Garnet (Nd-YAG) laser ablation, and argon plasma coagulation for hemostasis. This novel approach confirms the feasibility of minimally invasive partial lesion resection in TLCH management and establishes an intraoperative emergency protocol for massive hemorrhage control, offering a clinically relevant framework for managing this rare pediatric condition. A 9-year-old boy presented with a 4-day history of cough, hemoptysis, and chest tightness after blunt chest trauma. Physical exam revealed left lung dullness and reduced breath sounds. Initial labs were unremarkable, with negative infectious workup. Empirical azithromycin, fluids, and oxygen improved hemoptysis but worsened dyspnea and chest pain, prompting pituitrin administration. Thoracic CT showed leftward mediastinal/tracheal deviation, partial left main bronchial obstruction, and a 0.8 cm vascular-enhancing para-bronchial nodule. Diffuse left lung consolidation with volume loss and homogeneous enhancement was noted, without lymphadenopathy (Figure 1A). History included recurrent respiratory infections and eczema. The findings suggest traumatic bronchovascular injury with secondary pulmonary hemorrhage and consolidation, though underlying structural anomalies require exclusion given the enhancing nodule. Acute clinical deterioration despite initial stabilization highlights the need for urgent multidisciplinary evaluation to address potential airway compromise and hemorrhagic complications. A multidisciplinary team (MDT) concluded the left para-bronchial vascularized nodule likely represented a benign lesion despite its hypervascularity. Angiography with embolization was prioritized to mitigate bleeding risks. Preoperative planning included tumor resection via bronchoscopy under general anesthesia with bronchial blocker (BB-7) and ECMO standby. Bronchial artery angiography revealed contrast extravasation in left bronchial artery branches (Figure 1D1), prompting targeted embolization using gelatin sponge particles (350–560 µm) via microcatheter. Post-procedural angiography confirmed vascular occlusion with a “stump sign” (Figure 1D2). Bronchoscopy under laryngeal mask airway exposure identified a 8 × 10 × 15 mm broad-based, smooth-surfaced mass occluding the mid-left main bronchus at 9 o'clock (Figure 2A). A staged approach using snare resection, biopsy forceps, Nd-YAG laser ablation, and argon plasma coagulation (APC) was initiated. Histopathology samples were obtained (Figure 2B–E). Intraoperatively, massive hemorrhage ensued after 2/3 resection, necessitating emergency left bronchus occlusion via preset bronchial blocker to maintain right lung ventilation (Figure 2F). Failed hemostasis with pituitrin, topical epinephrine, and batroxobin prompted urgent repeat angiography, identifying a small aberrant bronchial artery branch. Coil embolization at the left bronchial artery origin achieved definitive bleeding control (Figure 1E). The patient demonstrated marked clinical improvement after the intervention. Postoperative enhanced CT of the lung showed partial re-expansion of left lung with significant tumor volume reduction (Figure 1B). Histopathological analysis confirmed the diagnosis of TLCH. MDT consultation recommended complete endoscopic resection under general anesthesia, however, the family chose alternative conservative therapy with two courses (half-month) of oral sirolimus. Bronchoscopy 6 months later revealed substantial tumor regression. The bronchial lumen exhibited marked dilated, permitting 4.0 mm bronchoscopic passage under minimal pressure (Figure 2G–I). Twelve-month follow-up CT confirmed complete left bronchial patency without residual lesions (Figure 1C). Serial bronchoscopic and radiographic evaluations until the submission of the manuscript demonstrated sustained remission without recurrence. LCH, a histologically benign vascular proliferation alternatively termed pyogenic granuloma (PG), demonstrates predilection for cutaneous and mucosal surfaces, particularly within the upper aerodigestive tract [1]. TLCH represents an exceptionally rare subset, with fewer than 20 documented cases globally and only five pediatric instances reported in medical literature [4-8] (Table 1). The etiopathogenesis of TLCH remains unclear but is hypothesized to involve localized vascular hyperplasia or chronic inflammation. In this case, the patient's recurrent respiratory infections may have contributed to endothelial proliferation. Although TLCH exhibits negligible malignant potential and limited invasiveness delayed management risks severe complications like life-threatening airway obstruction or hemorrhage, necessitating early intervention. TLCH management requires individualized strategies prioritizing bronchoscopic resection for airway preservation, with surgery reserved for anatomically complex or endoscopically inaccessible lesion. ECMO provides vital intraoperative support in high-risk resections when endoscopic/surgical approaches are insufficient. Pediatric TLCH management typically achieves resolution via single bronchoscopic interventions. Among five reported cases [4-8], four achieved resolutions through monotherapy. Technical variations included endoscopic forceps resection with oxymetazoline irrigation and electrocautery for hemostatic optimization in one case, while another employed preoperative prophylactic embolization. These lesions typically presented as small pedunculated masses with low hemorrhagic risk. Surgical resection was performed in only one pediatric case due to lesion size and rich of blood supply. This case represents the first reported pediatric TLCH successfully managed through multimodal bronchoscopic intervention, incorporating a therapeutic sequence of electrocautery snare resection, forceps debulking, Nd-YAG laser ablation, and APC. The 8 × 10 × 15 mm sessile, hypervascular mass necessitated staged resection due to broad anatomical attachment. Preoperative angiographic mapping proved critical given bronchial arterial variability; initial gelatin sponge embolization incompletely occluded feeding vessels, leading to intraoperative hemorrhage. Salvage coil embolization of a missed aberrant artery achieved definitive hemostasis. While polyvinyl alcohol (PVA) particles remain the embolization gold standard, gelatin sponges' transient efficacy and recanalization risks underscore their limitations. Though nonabsorbable, coils were justified here for absolute vascular exclusion during crisis. Successful outcomes relied on multidisciplinary coordination, preoperative embolization, and contingency planning. Key insights include: (1) temporary embolization agents necessitate backup strategies due to recanalization risks; (2) incomplete resection remains clinically acceptable when coupled with pharmacologic adjuncts like sirolimus; (3) intraoperative angiography is vital for salvaging hemorrhagic crises. These findings underscore the need for protocol refinement through multicenter collaboration to establish evidence-based guidelines. Future studies should prioritize long-term outcome monitoring and comparative analysis of embolization materials to optimize therapeutic algorithms for this rare pediatric condition. Lijun Wang: writing – review and editing, methodology, investigation, formal analysis. Haoran Jing: methodology, writing – review and editing, investigation, formal analysis. Yuhuan Liu: investigation. Keling Ma: formal analysis. Hao Wu: resources. Yang Li: data curation. Mingwei Wang: data curation. Fanzheng Meng: conceptualization; validation, project administration. Man Gao: conceptualization, validation, writing – review and editing. This study was a retrospective analysis of a single clinical case. According to the institutional policy of the Ethics Committee of the First Hospital of Jilin University, research involving individual case reports that have obtained informed consent from the patient and do not compromise privacy or ethical principles may be exempt from formal ethical review. Written informed consent was obtained from the patient's legal guardian before data collection and publication, including consent for the use of medical images and video materials. All patient identifiers have been removed to ensure confidentiality. The study was conducted in accordance with ethical standards, and the signed informed consent form is available upon request. The authors declare no conflicts of interest. The data that support the findings of this study are available from the corresponding author upon reasonable request.