Transperitoneal Laparoscopic Adrenalectomy in Children - Initial Experience in an Emerging Tertiary Center in Eastern India
Avilash Sahu, Aditya Arvind Manekar, Subrat Kumar Sahoo, Bikasha Bihary Tripathy, Manoj Kumar Mohanty
Department of Paediatric Surgery, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India
Submitted: 01-Oct-2024. Revised: 16-Dec-2024. Accepted: 31-Dec-2024.
Published: 03-Mar-2025.
ABSTRACT
Background: Adrenal tumors account for 0.2%-1% of all tumors in children. They can be benign or malignant, secretory or nonsecretory, and can arise either from the cortex or medulla. The common indication in children undergoing laparoscopic adrenalectomy (LA) is neuroblastic tumors (neuroblastoma, ganglioneuroma, and ganglioneuroblastoma).
Methodology: This is a retrospective observational study conducted from October 2021 to February 2024 (28 months) in the pediatric surgery unit at our institute. All children under 18 years of age referred to us for surgery of adrenal mass were included in this study. Any children with image-defined risk factors (IDRFs + ve) were excluded from the study. Children were worked up and managed according to the protocol of the Institute Pediatric Tumor Board. The laparoscopic transperitoneal approach was used for all. All adrenal lesions with IDRF +ve were excluded from the study and resected via open approaches.
Results: A total of six patients were included (three each in male and female groups). The mean age was 31 months (range 22 months-39 months). There were two cases of neuroblastoma, and one each of pheochromocytoma, adrenal adenoma, adrenocortical carcinoma, and paraganglioma. The median follow-up period was 24 months (range 7-32 months). On follow-up, there are no recurrences reported till date with zero mortality.
Conclusion: LA is a safe and feasible operation in children without any IDRF. It should be preferred in children with small lesion with good plane between the tumor and normal tissues. For larger lesion and ACC, it should be used cautiously to prevent spillage. Thought difficult, laparoscopic resection is a safe approach for these adrenal tumors.
KEYWORDS: Adrenalectomy, laparoscopy, neurogenic tumors
INTRODUCTION
A drenal pathologies are rare in children. Adrenal tumors account for 0.2%-1% of all tumors in children. They can be benign or malignant, secretory or nonsecretory, and can arise either from the cortex or medulla. The common indication in children undergoing laparoscopic adrenalectomy (LA) is neuroblastic tumors (neuroblastoma, ganglioneuroma, and ganglioneuroblastoma).[1] It was first described by Gagner et al. in 1992 and since then it has become a procedure of choice whenever anatomically and technically feasible.[2] In Indian literature, there are very
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few retrospective studies available on LA in children. Difficulties with LA in children are: less working space, large tumor masses in relation to body size, unavailability of small-sized instruments, infiltrative nature of tumors in children, and of course a long learning curve of the procedure.[1] Herein, we report a retrospective analysis of our initial six pediatric LA and
Address for correspondence: Dr. Bikasha Bihary Tripathy, Department of Paediatric Surgery, All India Institute of Medical Sciences, Bhubaneswar, Odisha, India. E-mail: bbtripathy.dr@gmail.com
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How to cite this article: Sahu A, Manekar AA, Sahoo SK, Tripathy BB, Mohanty MK. Transperitoneal laparoscopic adrenalectomy in children - Initial experience in an emerging tertiary center in eastern India. J Indian Assoc Pediatr Surg 2025;30:317-21.
compare it with previously available literature regarding the safety and technical feasibility.
METHODOLOGY
This is a retrospective observational study conducted from October 2021 to February 2024 (28 months) in the pediatric surgery unit at our institute. All children under 18 years of age referred to us for surgery of adrenal mass were included in this study. Any children with image-defined risk factors (IDRFs +ve) were excluded from the study. Children were worked up and managed according to the protocol of the Institute Pediatric Tumor Board. The laparoscopic transperitoneal approach was used for all. All IDRF-positive adrenal lesions were excluded from the study as they were resected via an open approach.
Procedure
The child was placed in the reverse Trendelenburg position with operating side up. The surgeon and the assistant surgeon stood in front of the patient with a video monitor placed on the back of patient. Head was elevated 10°-15°. We used all 5 mm ports, (3 in number, extra port for right-sided lesions for liver retraction), as shown in Figure la and b. The camera port was put at the umbilicus via open Hassan’s technique. Pneumoperitoneum was achieved with carbon dioxide at pressure 8-10 mm, and flow 2.5-3 l/min and was adjusted according to anesthetic consideration.
For the right-sided lesions, an extra liver retracting port was put lateral to the working port as shown in Figure la. The right triangular, ligament was divided and then a grasper was used to hold the liver margin for retraction. Duodenum was kocherized and reflected medially to expose the inferior vena cava. Clear dissection was done between the adrenal and renal vessels so as not to injure the renal vasculature. Dissection was done initially to isolate the adrenal vessels which are shorter on the right
a
b
Tumor mass
5 mm
5 mm
Tumor mass
5 mm
5 mm
5 mm
5 mm
5 mm
Working ports
Camera port
Liver retraction ports
side, making the procedure more challenging. Once the vessels were identified and dissected free, we used 5 mm hemoclips and scissors to cut the vessels. Dissection was done to free all the attachments of the lesion using LigaSure and hook electrocautery and then the specimen was removed through the umbilical port under vision.
For the left-sided lesions, dissection started by incising the lateral fold of the peritoneum (white line of Toldt). Dissection was done along the splenic flexure till the sigmoid to reflect the colon medially. Phrenicocolic and lienorenal ligaments were cut and the spleen was made to fall medially to expose the retroperitoneum. Adrenal vessels were secured and ligated first before dissecting the gland to minimize the risk of catecholamine release in case of pheochromocytoma. The left adrenal vein was identified as medial to gonadal termination. Gland dissection was done with LigaSure to ensure complete hemostasis. Then specimen was retrieved [Figure 2a-d].
Intraoperative events if any such as organ injury or excessive bleeding were noted. Total operating time was considered from port insertion to port closure. Any conversion to open procedure was also noted. The child was shifted to the ward and started on feeds on 1st postoperative day and discharged from the surgical ward once on full feeds.
Biopsy was followed up and follow-up treatment was given accordingly by the pediatric oncology team. Children were called after 7 days from discharge to examine the local wound site. Screening abdominal ultrasound was done at 3 months and then followed up only if symptoms recurred as per the oncology team.
RESULTS
A total of six patients were included in the 28-month study period. Three each in male and female groups.
a
c
21.22 23 24 25 28 21/ 218 2690
Right adrenal vein
b
d
Median weight and age were 11 kg (range 9 kg-21 kg) and 31 months (range 22 months-39 months). Detailed demographics and intraoperative events are given in Tables 1 and 2. There were two cases of neuroblastoma, and one each of pheochromocytoma, adrenal adenoma, adrenocortical carcinoma, and paraganglioma. Three children (one pheochromocytoma, one neuroblastoma, and one adrenocortical carcinoma) had hypertension and were managed medically. Median operating time was 137.5 min (120-160 min). As we can see in Table 2, the carcinoma case required a longer time as the procedure was done slowly and meticulously to prevent spillage and achieve a gross RO resection. Clear demarcation between the tumor and adjacent organ with no local infiltration and size of 6 cm helped us in achieving successful laparoscopic resection. A child with pheochromocytoma required conversion due to excessive intraoperative bleeding requiring transfusion of single unit blood. Drain was also put in the same child which was removed on 2nd postoperative day. All children were started on oral sips as early as possible on 1st postoperative day and slowly graded up. The median surgical ward stay was 4 days. None had any complications according to the Clavien- Dindo classification. The median follow-up period was 28 months (range 7-32 months). On follow-up, there are no recurrences reported till date with zero mortality.
DISCUSSION
There are very few articles in Indian literature available on pediatric LA. Since its first description in 1992, it has gained popularity and is currently the procedure of
choice in adults for benign and malignant lesions. Still, in pediatric patients, it has got a steep learning curve. The first pediatric LA was reported by Yamamoto et al. in 1996[3] and later a case series was published by Mirallié et al.[4] in 2001 showing the safety and feasibility of this approach.
Previously conducted multicenter studies by St Peter et al. and Romano et al. have suggested that there are no weight and age restrictions.[5,6] The youngest baby to undergo LA is 14 days’ old weighing 3.2 kg as reported by Sosnowska-Sienkiewicz et al. with an operating time of 70 min.[7] Due to less working space in the pediatric age group, ergonomics is always a concern for performing LA. But having said this, there is no upper size limit recommended above which LA is contraindicated. Initially, international pediatric endosurgery group (IPEG) recommended that the size of adrenal lesion >6 cm is a relative contraindication for laparoscopic surgery but they summarized that there is no absolute contraindication for LA in children as long as the principle of oncology is not violated.[8] Several pediatric studies by Nerli et al. have shown safe resection with lesion of 7 cm.[9] Miller et al. and Pampaloni et al. in their cohort have stated there is no size limit for safe LA in children.[10,11] Newer adult studies also have shown good outcomes in larger adrenal lesions. Our decision of excluding cases with positive IDRF is consistent with studies by Shirota et al. and Sosnowska-Sienkiewicz et al. who have suggested safe resections in children can be achieved if lesions with IDRF-positive lesions are excluded.[7,12] In 2022, Oesterreich et al. published
| Table 1: Details of each patient | |||||||
|---|---|---|---|---|---|---|---|
| Age (months) | Weight (kg) | Sex | Clinical features | Diagnosis | Laterality | Max size on imaging (cm) | BP |
| 30 | 20 | Male | Virilization | Adrenocortical carcinoma | Right | 5.5 | Normal |
| 22 | 11 | Male | Abdominal pain | Adrenal adenoma | Left | 3.8 | Normal |
| 33 | 13 | Male | Incidentally detected | Paraganglioma | Right | 4.1 | Normal |
| 28 | 10 | Female | Incidentally detected | Neuroblastoma INSS2 | Left | 5.1 | Normal |
| 39 | 15 | Female | Irritability with poor feeding | Pheochromocytoma | Right | 6.2 | Hypertensive |
| 36 | 9 | Female | Abdominal lump | Neuroblastoma INSS2 | Left | 3.2 | Normal |
BP: Blood pressure
| Table 2: Depicting intraoperative events and follow-up | ||||||||
|---|---|---|---|---|---|---|---|---|
| OTT (min) | Intraoperative complication | Conversion | Postoperative drain | Postoperative complications | Duration of NPO (h) | Hospital stays (days) | Follow-up (months) | Follow up status |
| 160 | Nil | No | No | Nil | 24 | 4 | 35 | Disease free |
| 120 | Nil | No | No | Nil | 14 | 3 | 32 | Disease free |
| 135 | Nil | No | No | Nil | 20 | 5 | 27 | Disease free |
| 144 | Nil | No | No | Nil | 16 | 4 | 29 | Disease free |
| 140 | Bleeding | Yes | Yes | Hyperkalemia | 36 | 6 | 12 | Disease free |
| 127 | Nil | No | No | Nil | 24 | 4 | 9 | Disease free |
NPO: Nil per oral, OTT: Operation theatre time
a selection criteria and algorithm for safe LA. Further, they stated size <6 cm, negative IDRF, no feature of adrenocortical carcinoma on radiology, L1 according to International neuroblastoma risk group (INRG) as selection criteria for safe LA. The limitation of their study was a single-center, retrospective study design with small sample size. Their algorithm requires more collaborative research for future validation.[13]
Every child with adrenal mass should be managed on case-to-case basis under the care of a multidisciplinary team which should include a pediatric surgeon, radiologist, and pediatric oncologist. The decision of surgery should always be made after discussing it in the tumor board. The child should be offered the safe approach for resection; may it be open or minimal access (laparoscopic over retroperitoneoscopic or robotic). One should take care preoperatively about the involvement of viscus or vascular structures or any IDRF. The mean size of the adrenal lesion in our study was 4 cm. Largest size and smallest size were 6.2 and 3.2 cm, respectively. Two children with neuroblastoma had received six cycles of neoadjuvant chemotherapy. None in our cohort had any positive IDRF, so all except one in our study had successful completion of LA. Our idea of excluding the IDRF-positive tumors was consistent with previously published study by Shirota et al. and Catellani et al.[12,14]
The common complications encountered are bleeding (most common), adjacent viscus injury, colonic perforation, diaphragmatic injury, and inadequate exposure.[15] In our study, we had one intraoperative complication of bleeding that required conversion to an open approach. Bleeding in our case was due to venous bleeding and fluctuating blood pressure (BP) in a pheochromocytoma child which required one packed cell transfusion. Bleeding as an intraoperative complication was also reported by de Barros et al. in their study of seven adrenal neuroblastoma without any IDRF.[16] They also attributed excessive bleeding from the adhesion-formed postchemotherapy but we did not encounter such bleeding in two of our postchemotherapy patients in whom the surgery was completed successfully via laparoscopy.
We required conversion in one case of pheochromocytoma. Conversion rate and local recurrences are reported higher in cases of IDRF-positive lesion than the negative ones.[17] Studies also have advised a minimal access approach for positive IDRF tumors and then subsequent conversion during difficult dissection. They found no statistical significance in the two groups: one being IDRF-positive tumors approached with laparoscopic surgery then converted to open
one and tumors directly treated with open surgery.[18] Successful Minimally invasive surgery (MIS) for Adreno cortical carcinoma (ACC) was also reported by Oesterreich et al. in their study on the laparoscopic approach to pediatric adrenal tumors in 2022.[13]
The median operating time in our study was 137.5 min. As in the results, we can see by gaining experience there was a reduction in operating time from 160 to 120 min. Our results were similar to de Barros et al. and Lopes et al. with a mean operating time of 138 min.[16,19] On contrary, Mandelia et al. and Miller et al. reported longer operating time than ours which might be due to larger lesions in their cohort.[1,10] The other thing that they found is that the surgical time depends on the tumor type and is more for pheochromocytoma. This prolonged time could be due to inadequate BP stabilization during surgery. Sosnowska-Sienkiewicz et al. in their cohort had prolonged timing of 230 min in two cases of pheochromocytoma.[7]
Laparoscopic resection has many advantages over conventional open surgeries. These are minimal requirements of analgesia, early start of feeding, short postoperative stay, and excellent cosmesis. On the other hand, there is always a chance of tumor spillage and port site recurrence during specimen retrieval especially in cases of ACC. It is also advised not to morcellate the gland as well as the lesion during surgery. In our case also, we used a specimen retrieval bag made from UROBAG® with a purse string suture tied above it for securing the specimen inside the bag. Till date, we do not have any recurrences due to spillage. If there is any difficulty a Pfannenstiel incision can be given to retrieve the specimen. Previous studies have reported that adrenocortical carcinoma is a contraindication for LA as there is a possibility of tumor spillage due to infiltrating nature of the tumor.[1]
The mean surgical ward stay in our study was 4 days. As our patients are from far-off tribal areas we keep them in the hospital for 1 day extra before sending them on their long journey back home. The mean hospital stay was 3.5 days in previous studies by Catellani et al. and Lopes et al.[14,19] There are also studies reporting a shorter mean hospital stay of 2 days by Nerli et al. and Eassa et al.[9,20] Postoperative stay can be prolonged sometimes due to delayed recovery because of electrolyte imbalance in Conn’s syndrome causing paralytic ileus and parental anxiety and apprehensiveness but we did not have any of those complications in postoperative period.[1]
Currently, there has been a trend toward retroperitoneoscopic adrenalectomy. The advantages of it are; no colon mobilization, no breech in the
peritoneum, and less risk of intraperitoneal viscus injury. The downside of this approach is limited working space, bilateral lesion that cannot be accessed, and poor bleeding control. The major drawback for learners is that it is technically challenging with a longer learning curve. Series published by Lee et al. in 2019 showed the safe and feasible execution of this approach with a short operating time and mean ward stay of 3 days. In their study, they measured the lumbar lordosis angle and stated that since this angle is <40° in children, the position required to flatten this angle is minimal and makes the procedure feasible in the less extreme operating position.[21]
Limitations in our study are small sample, retrospective study design, and single emerging tertiary care center with a short follow-up period. Small sample size is due to rarity of adrenal masses in children. Being an emerging center with learning experience, the operation theatre (OT) timing is slightly longer than usual which may depict the slowness of this procedure which can be enhanced with advanced skills.
CONCLUSION
LA is a safe and feasible operation in children without any IDRF. It should be preferred in children with a small lesion with a good plane between the tumor and normal tissues. For larger lesions and ACC, it should be used cautiously to prevent spillage. Thought difficult, but it is possible to safely resect a carcinoma laparoscopically.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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