A symptomatic adrenal mass diagnosed during pregnancy is a rare clinical scenario. Two primary considerations are the timing of intervention and determining the appropriate surgical approach. Here, we present the case of a young female patient with flank pain, whom on diagnostic imaging, was found to have a large cystic adrenal mass. She was taken for a robotic-assisted resection of her left-sided adrenal mass during the second trimester of pregnancy. Preoperative and postoperative assessment demonstrated a viable intrauterine pregnancy. She tolerated the procedure well and was discharged 5 days later with resolution of her symptoms on follow-up. As one of only three reported cases of robotic adrenal surgery during pregnancy, we demonstrate that a robotic-assisted surgical approach is a viable and durable surgical option under the given clinical circumstances.
- Adrenal disorders
- General surgery
- Surgical oncology
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An adrenal cystic mass may be diagnosed clinically or discovered incidentally on imaging for an unrelated problem. This is a rare clinical diagnosis, with only 500 cases reported in the current literature.1 Pseudocyst is the most common of the four subtypes of adrenal cystic masses.2 An adrenal pseudocyst is defined as a non-neoplastic, non-parasitic cyst of the adrenal gland in which an epithelial or endothelial lining is not demonstrated.3 The majority of these lesions are asymptomatic, however, there is an association with pregnancy and haemorrhage into the cyst.4 These patients may present with severe abdominal or flank pain, and in severe circumstances, in haemorrhagic shock. Adrenal cystic lesions have also been associated with adrenocortical carcinoma and pheochromocytoma, and these need to be ruled out. Because imaging and fine-needle aspiration biopsy can sometimes be misleading, excisional biopsy may be required for a definitive diagnosis.1 5
Here, we present a pregnant patient that was found to have a cystic adrenal mass that, on workup with image-guided biopsy through a multidisciplinary approach, demonstrated benign pathology. She was taken to the operating room during the second trimester and was successfully managed with a minimally invasive surgical approach. This case presents a unique diagnosis and demonstrates the utility for robotic-assisted surgical management for pregnant patients with symptomatic, benign adrenal masses.
A female patient in her 20s, 8 weeks pregnant by last menstrual period, Gravida 3, para 2, with one prior abortion, presented to our institution with complaints of left-sided flank and lower back pain radiating to the left mid-abdomen for 2 days duration. She was afebrile and tender on examination in the left upper abdomen and flank area, with no rebound tenderness or guarding. Her vital signs were normal. The labs were significant only for a mild leukocytosis and a normal lactate.
Initial investigations included imaging with renal ultrasound, demonstrating a heterogeneous, hypoechoic-appearing left suprarenal lesion measuring 7.8×6.4×7.5 cm. She was admitted to the surgical service for pain control and underwent an abdominal magnetic resonance angiogram, which demonstrated a left upper quadrant (LUQ) cystic lesion of either mesenteric or adrenal origin, as depicted in figure 1.
The obstetrics team was consulted on admission and determined that she had a viable intrauterine pregnancy roughly 8 weeks gestational age on pelvic ultrasound. The advanced endoscopy service was consulted for endoscopic ultrasound and transgastric fine-needle aspiration biopsy of her LUQ cystic mass. The final pathology report determined there were few atypical cells, and therefore, malignancy could not definitively be ruled out. The patient continued to experience severe abdominal pain requiring multiple doses of intravenous opioids until discharge on hospital day 10.
One month later, the patient presented to the emergency department with severe left-sided abdominal and flank pain, associated decreased appetite and nausea. A repeat MRI demonstrated interval increase in the size of the adrenal mass, now measuring 7.5×7.4 ×7 cm. After multidisciplinary review, the interventional radiology service was consulted for CT-guided core needle biopsy. Imaging findings on CT angiography are depicted in figure 2. A 15 mL of bloody fluid was obtained and sent for cytology, demonstrating inflammatory cells and no malignancy, atypia or necrosis. She was discharged on hospital day seven and followed in her surgeon’s office the following week for preoperative planning. She was offered surgical excision based on the persistence of her severe pain requiring multiple hospital admissions for pain control and to rule out underlying malignancy.
At 18 weeks of pregnancy, the patient presented electively for a robotic-assisted left adrenal cystic mass excision. The obstetrics team evaluated the patient preoperatively to establish a fetal heart rate by ultrasound. She was placed in the right lateral decubitus position after marking her abdomen for port placement, with care taken to avoid the gravid uterus. Pneumoperitoneum was established with a varess needle placed at Palmer’s point and set to an insufflation pressure of 12 mm Hg. Entry into the abdomen was accomplished with a robotic zero-degree scope using an OPTIVIEW trocar (Ethicon US, Bridgewater, New Jersey, USA) placed in the right upper quadrant (RUQ). Four 8 mm robotic ports were placed under direct visualisation with a 30° robotic scope in the right upper, supraumbilical and left mid abdomen using the DaVinci Xi system (Intuitive Surgical, Sunnyvale, California, USA). The 12 mm laparoscopic assistant port was placed between robotic arms 2 and 3, which housed the camera and robotic endoshears, respectively. Arms 1 and 4 housed the fenestrated bipolar and tip up bowel grasper, respectively. Her target anatomy was set to the LUQ, just inferior to the spleen and lateral to the greater curvature of the stomach.
Initial inspection of the abdomen demonstrated a large retroperitoneal bulge, which was assessed with the use of an endoscopic ultrasound (figure 3A). Initially, the descending colon was mobilised followed by the splenic flexure; and then the gastrocolic ligament was divided to gain access to the lesser sac (figure 3B). This assisted in reflecting the omentum superiorly towards the RUQ and the colon inferiorly towards the pelvis. The retroperitoneal mass was approached initially from the lateral aspect to avoid the renal structures at the hilum of the kidney (figure 4A). The spleen was mobilised laterally at the splenophrenic ligament. The body and tail of the pancreas had been pushed cephalad by the mass and were carefully dissected free. Dissection of the superior portion of the cyst followed until normal appearing adrenal gland tissue was identified (figure 4B).
The mass was densely adherent to the superior aspect of the adrenal gland and an adrenalectomy was accomplished using the robotic Ligasure device and endoshears on arm 3 (figure 4C,D). After completing lateral, superior and inferior dissection, the left adrenal vein was identified anteromedial to the cystic mass. The vessel was clipped and divided with the Ligasure (figure 5A,B). The cystic mass was dissected free and decompressed with an endoscopic needle yielding about 60 mL of bloody fluid. The specimen was then extracted using a large endobag through the laparoscopic assistant port, after extending the incision 2 cm in each direction horizontally. Next, haemostasis was achieved and the abdomen was desufflated and closed.
Outcome and follow-up
Overall, the patient underwent an uncomplicated procedure and had an uneventful hospital stay. The obstetrics team evaluated the patient in the postanaesthesia recovery unit and established a fetal heart rate by ultrasound. She was then sent to the surgical intensive care unit for 24 hours of postoperative monitoring. The final pathology following her surgery was intraluminal organising adrenal haemorrhage with pseudocyst formation as depicted in figure 6. The peripheral gland appeared unremarkable and negative for tumour with one benign lymph node. Figure 7 depicts microscopic tissue sections of the mass.
On follow-up in the surgeon’s office, the patient remained free of her left flank and abdominal pain. She maintained a healthy pregnancy and delivered a baby girl at full term without complications.
Our patient’s workup demonstrated a symptomatic, unilateral, non-functioning adrenal mass that was cystic in nature. She denied any episodic hypertension, headaches, dizziness, flushing or palpitations. She was not taking any medications at the time of her presentation. There was a low suspicion for a functional adrenal mass under these circumstances, and this was ruled out early in her management. Initial screening included normal serum sodium, potassium, aldosterone, renin activity and plasma metanephrine levels. Additionally, fasting AM cortisol, adrenocorticotrophic hormone and dehydroepiandrosterone sulfate levels were within normal limits.
Tissue biopsy was required to rule out the remote possibility of adrenal malignancy. Considering that solid adrenal tumours>6 cm in diameter carry a 25% risk of potential malignancy, cystic adrenal masses carry a much lower malignancy potential of roughly 6%.6 7 Initial fine-needle aspiration biopsy was inconclusive and was repeated with core needle biopsy and cyst aspiration. Because the final pathology results demonstrated atypical cells and malignancy could not definitively be ruled out, surgical excision was the next best step in establishing a diagnosis while also potentially alleviating her complaints of persistent pain.
A multispecialty approach was established to facilitate the diagnosis and management of our patient’s surgical complaint. The obstetrics team evaluated the pregnancy throughout her care, from initial presentation at 8 weeks gestation to discharge after her surgery at 19 weeks gestation. The gastroenterology and interventional radiology teams were consulted for imaging and diagnostic procedures. The case was reviewed at a multidisciplinary tumour board meeting and it was determined that interval adrenalectomy and mass excision following entry into the second trimester was the optimal intervention.
Adrenalectomy for a haemorrhagic adrenal cystic mass has been performed both during or following pregnancy and the timing of the procedure remains a difficult dilemma.3 8 9 The current recommendations as outlined by the American College of Obstetricians and Gynecologists indicate that patients should undergo non-emergent, non-gynaecological surgery in the second trimester when feasible.10 Urgent surgery should be performed regardless of trimester, while completely elective surgery may be postponed until after the completion of pregnancy.
Although laparoscopic adrenalectomy in pregnant patients has been performed for Cushing syndrome, hyperaldosteronism and pheochromocytoma,11–14 this is the first case of robotic-assisted adrenal mass excision reported in the literature for an indication other than pheochromocytoma and paraganglioma.15 16 There are multiple publications since 2007 that demonstrate the safety and utility of robotic-assisted abdominal surgical procedures in pregnant patients.13 17–21 Even though many of these publications are presented as case studies or case series, there are no large, randomised studies comparing robotic with laparoscopic and open surgical procedures in pregnant patients. This is likely attributable to the case numbers and availability of robotic surgery systems as well as the limited number of trained and experienced robotic surgeons available at various institutions worldwide.
A recent meta-analysis comparing outcomes from robotic-assisted and laparoscopic adrenalectomies performed on 1162 patients (747 patients treated with robotic assisted adrenalectomy and 415 patients treated with laparoscopic adrenalectomy) found no significant differences in intraoperative and postoperative complications, mortality, conversion to open and haemorrhage.22 There was, however, longer operative time and shorter hospital stay for patients undergoing robotic adrenalectomy. Another meta-analysis comparing robotic-assisted adrenalectomy with laparoscopic adrenalectomy in 600 patients (277 robotic assisted adrenalectomies and 323 laparoscopic adrenalectomies) determined that there was no significant difference in operative time and conversion rate, but that laparoscopic adrenalectomy was associated with longer hospital stay and significantly higher estimated blood loss.23 Both studies were limited by the quality of evidence that was reviewed and conclude that robotic assisted adrenalectomy is safe and feasible.
The decision to perform this case using a robotic approach rather than a laparoscopic approach was primarily based on the surgeon’s prior operative experience and his preference for this patient given the clinical circumstances. With consideration to the position of the patient’s gravid uterus, operative exposure was of primary concern and thought to be advantageous using the robotic scope. Fine dissection was assisted by the articulating instruments and three working arms of the robotic system.
A symptomatic adrenal cystic mass during pregnancy is an exceedingly rare clinical circumstance with limited precedence, as demonstrated by a review of the current literature.
The differential diagnosis is broad, requires cross-sectional imaging and tissue biopsy to make a diagnosis, and is generally managed by excision or adrenalectomy.
The timing of surgical intervention is a key factor to be considered: this patient underwent surgical intervention during the second trimester of pregnancy, which is in accordance with the current guidelines for non-emergent non-gynacological surgery during pregnancy.
Robotic-assisted minimally invasive surgical management for symptomatic, benign adrenal pathology is a viable option during pregnancy.
Patient consent for publication
Contributors Supervised by AP and IM. Patient was under the care of AP, IM and NTC. Report was written by NTC and DM.
Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
Competing interests None declared.
Provenance and peer review Not commissioned; externally peer reviewed.