Nijmegen breakage syndrome (NBS) is clinically characterized by a high predisposition to lymphoid malignancies [1]. Lymph node biopsy is a standard diagnostic method where there is a suspicion of lymphoma. In recent years, fluorescence-guided techniques using tracers such as indocyanine green (ICG) have been introduced as an effective sentinel lymph node biopsy method to enhance tissue visualization. ICG imaging is a real-time method to evaluate organ perfusion based on direct visualization of the fluorescence emitted by ICG under near-infrared (NIR) light after injecting the tracer solution [2].
Improving intraoperative visualization of lymph nodes has become an important clinical challenge [3]. When pathological lymph nodes are deeply located in the abdomen or pelvis, blind dissection often leads to unnecessary invasiveness or a potentially increased risk of perioperative complications. Casaccia et al. and Gilbert et al. [4, 5] have reported laparoscopic biopsy as a method of choice. According to the literature, indocyanine green visualization has been established as a well-known tracer. In various oncological pathologies with secondary involvement of lymph nodes, ICG has already been established as a tracer of choice [6, 7]. The range of possible causes of abdominal and pelvic lymphadenopathy in the pediatric population is wide. Distinguishing between benign, atypical, and malignant lesions is crucial, especially in children with a genetic predisposition for malignancy. There is no consensus regarding the best diagnostic method of a minimally invasive way to obtain material for a histopathological examination in lymphoproliferative disorders in pediatric oncology.
This report aims to present the effectiveness of ICG-guided biopsy as an additional method of targeted surgical lymphatic tissue biopsy in pediatric patients.
The authors present the case of a 16-year-old girl with NBS and abdominal lymphadenopathy. In her medical history, bilateral surgical treatment of vesicoureteral reflux and right nephrectomy of the hypoplastic kidney had been performed. Screening ultrasound examination revealed enlarged lymph nodes in the hypogastric region. Magnetic resonance imaging (MRI) showed a pathological lymphatic mass spreading along the inferior vena cava and right iliac vessels. In addition to the high risk of lymphoproliferative disorders in NBS and the risk of postoperative adhesions (taking into consideration the right nephrectomy in the past), the patient was qualified for laparoscopic lymph node biopsy assisted with ICG fluorescence imaging. During the induction of general anesthesia, subcutaneous and intradermal injection of 2 mL of ICG solution in the bilateral inguinal region was performed. Intraoperatively, we detected clear and strong visualization of lymphatic tissue based on fluorescence. This allowed the surgeon to guide directly to the pathological lymph nodes (Fig. 1). Also, delayed ICG flow through the pathological lymphatic mass and heterogenous enhancement patterns were observed compared to normal-looking other lymphatic tissue (according to the preoperative MRI). Pathomorphological examination of the material revealed Hodgkin’s lymphoma (mixed-cellular subtype).
The postoperative course was uneventful, and the patient was transferred to the Department of Pediatric Oncology and Hematology three hours after the operation. She was qualified for chemotherapy.
Lymphomas need to be diagnosed by biopsy of lymphadenopathies with localizations that are often difficult to access, such as the retroperitoneum, neck, or mediastinum [8]. Initial reports of ICG application to identify pathological lymphatic tissue in lymphoproliferative diseases confirm that this is a feasible and safe diagnostic method. Excellent visualization of the local lymphatic system results in improved patient perioperative safety, a shorter operating time, and a reduced risk of both short- and long-term complications [6]. The pattern of fluorescence and tropism of ICG in pathological lymphatic mass is a promising area for further investigation. This intraoperative feature has been observed by Casaccia et al. [5], who reported a stop of the tracer flow in enlarged lymph nodes with a probability of obstruction of lymph drainage induced by pathological tissue.
Evaluation of ICG-guided biopsy of lymph nodes and its effect on the reduction of false negative results is an attractive option for pediatric patients. According to the FLABILY study, using ICG could simplify the lymph node search and help in the selection of a more accurate place for a biopsy [9]. More prospective studies with larger patient samples are needed to reach conclusions regarding the optimal method of use of fluorescent staining in lymphoproliferative disorders in the pediatric population.
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Authors declare that informed consent for publication was not obtained, as published data does not allow for patient identification.
Authors’ contributions
All authors wrote, reviewed and agreed to the final version of the manuscript.
Funding
This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.
Conflict of interest
The authors declare no conflict of interest.
Acknowledgements
We thank Prof. Przemysław Gałązka for critical revision of this manuscript.
