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J Case Rep Images Urol 2021;6:100013Z15LG2021. Galloway et al. 1 www.ijcriurology.com

CASE REPORT PEER REVIEWED OPEN| OPEN ACCESS ACCESS Dysautonomia following radical cystectomy and intracorporeal orthotopic neobladder: A case report

Lan Anh S Galloway, Christopher P Dall, Lambros Stamatakis

ABSTRACT Article ID: 100013Z15LG2021

Orthostatic and dysautonomia occur commonly after , but there is little data regarding ********* these complications in the genitourinary surgery population. This report presents the case of a 74-year-old doi: 10.5348/100013Z15LG2021CR male diagnosed with muscle-invasive bladder carcinoma who underwent radical cystectomy and creation of an intracorporeal orthotopic neobladder. Following surgery, he developed significant lightheadedness and was found INTRODUCTION to have of unclear etiology. We postulate that this is likely multifactorial and due to a Dysautonomia and orthostatic hypotension are myriad of factors including postoperative weight loss, common postoperative complications found in up to extensive lymph node dissection, presence of a hiatal 49% of patients undergoing a wide range of procedures, hernia, and dietary contributions. This case report is the including orthopedic, bariatric, abdominal, and thoracic first of its kind to investigate and report on orthostatic surgery. It can result in delayed ambulation, increased hypotension as a postoperative complication of radical length of hospitalization, and falls [1–3]. However, cystectomy and creation of an orthotopic ileal neobladder. there are few reported cases in the literature regarding dysautonomia following major genitourinary surgery. In Keywords: Cystectomy, Dysautonomia, Orthostatic hypo- this study, we present a profound case of dysautonomia tension, Postoperative complication and orthostatic hypotension following robotic-assisted laparoscopic radical cystectomy and creation of an How to cite this article intracorporeal orthotopic neobladder.

Galloway LAS, Dall CP, Stamatakis L. Dysautonomia following radical cystectomy and intracorporeal CASE REPORT orthotopic neobladder: A case report. J Case Rep Images Urol 2021;6:100013Z15LG2021. A 74-year-old male with a history of hypertension, type II mellitus, hypothyroidism, hiatal hernia, and Agent Orange exposure presented to the Lan Anh S Galloway1, Christopher P Dall2,3, MD, Lambros clinic with gross hematuria. The patient underwent a Stamatakis2,3, MD computerized tomography (CT) scan, which revealed Affiliations: 1Medical Student, Georgetown University School asymmetric bladder wall thickening. Transurethral of , Washington, DC, USA; 2Resident , resection of bladder tumor (TURBT) was performed Department of Urology, MedStar Georgetown University and demonstrated high grade urothelial carcinoma 3 Hospital, Washington, DC, USA; Director of Urologic On- with invasion of the detrusor muscle (clinical stage T2). cology, Department of Urology, MedStar Washington Hospi- Subsequent staging revealed a lung mass and biopsy tal Center, Washington, DC, USA. demonstrated a concurrent right lung adenocarcinoma. Corresponding Author: Lan Anh S Galloway, MedStar After multidisciplinary discussion, he underwent Washington Hospital Center, 106 Irving St. NW, POB South lobectomy with four cycles of adjuvant gemcitabine and Room 315, Washington, DC 20010, USA; Email: Lsg56@ georgetown.edu cisplatin chemotherapy. Following this, he underwent repeat cystoscopy and bladder biopsy which demonstrated no evidence of residual carcinoma. Radical cystectomy Received: 17 December 2020 and urinary diversion were discussed and recommended Accepted: 05 February 2021 as the gold standard treatment for muscle-invasive Published: 14 March 2021 bladder carcinoma (MIBC), but the patient opted for

Journal of Case Reports and Images in Urology 6, 2021. J Case Rep Images Urol 2021;6:100013Z15LG2021. Galloway et al. 2 www.ijcriurology.com close imaging and cystoscopic surveillance in light of his Therefore, he was started on fludrocortisone and noted age and recent treatment for lung cancer. significant improvement in his symptoms, described by Surveillance cystoscopy six months later revealed the patient as a “miraculous” resolution. a new left lateral wall bladder tumor. was was consulted and believed his orthostasis may have consistent with MIBC with micropapillary features. been due to adrenal insufficiency. However, his work-up Restaging CT demonstrated pelvic lymphadenopathy, was negative and no clear cause was identified. He did suggestive of possible nodal metastasis. The likelihood note a weight loss of 8 kilograms since his cystectomy. of nodal metastasis was discussed extensively with Nutrition was consulted and recommended nutritional the patient and it was explained to him that a radical supplements and a high-protein diet. Following his cystectomy alone would likely not be curative, but that hospitalization, salvage pembrolizumab was initiated. it could be considered as part of a multimodal approach He continued to follow with the urology clinic to manage his advanced bladder cancer. He met with and reported slow improvement in his and radiation and medical oncology for further lightheadedness on ambulation. However, 11 months management recommendations and elected to proceed after surgery, he developed a small bowel obstruction and with a radical cystectomy with plans to proceed with upon surgical exploration he was found to have metastatic salvage immunotherapy. He was seen and evaluated bladder cancer implants on his small bowel. He aspirated preoperatively by the anesthesia team and noted to have on extubation and was admitted to the Surgical Intensive diabetic neuropathy and hypertension but was deemed Care Unit (SICU). His family ultimately decided to optimized for surgery. withdraw care and the patient expired shortly thereafter. The patient was taken to the operating room for a robotic-assisted laparoscopic radical cystoprostatectomy, extended lymph node dissection, and creation of an DISCUSSION intracorporeal orthotopic neobladder. The neobladder was made from a section of ileum 15 cm from the ileocecal Orthostatic hypotension is defined as a decrease valve and consisted of approximately 30 cm of bowel. in systolic by 20 mmHg or decrease in The small bowel anastomosis was performed in a side- diastolic blood pressure by 10 mmHg within 3 minutes of to-side fashion using the Endo GIA stapler (Medtronic, standing [4]. Normally, standing activates a physiologic Minneapolis, MN). The patient’s postoperative course was response in carotid baroreceptors, which increases unremarkable, and he was discharged on postoperative sympathetic tone and decreases parasympathetic tone day six. Surgical pathology revealed pT3aN3 urothelial via the vagus [5]. Maintenance of blood pressure is carcinoma, predominantly micropapillary pattern (90%). dysfunctional in those with orthostatic hypotension and 24/46 sampled lymph nodes were positive for carcinoma. blood pressure does not increase upon standing, impairing Six weeks after surgery, the patient presented to the cerebral perfusion. This can lead to lightheadedness emergency department with fever, nausea, and vomiting. and dizziness, and loss of consciousness if cerebral He complained of dizziness and lightheadedness hypoperfusion is severe [5]. Orthostatic hypotension is exacerbated by movement and sitting up. Admission often mediated via cardiac, neurologic, or drug-related vitals were notable for a fever to 38.5°C, a rate of 97 pathways. Diagnostic work-up includes autonomic beats per minute (bpm), and a blood pressure of 169/76. testing, investigations into patient drug regimens, and the Labs were significant for a leukocytosis to 24 × 103 cells/ head-up tilt test if the patient demonstrates symptoms μL and a lactatemia of 3.7 mmol/L. He was admitted for but no difference in blood pressure upon standing [5]. a urinary tract infection and started on vancomycin and The incidence of orthostatic hypotension has been found piperacillin/tazobactam. Subsequent blood and urine to be as high as 49% following any surgery and roughly cultures grew Escherichia coli and the patient began to 33% of abdominal [1]. Although there has been improve clinically. extensive research in other fields examining orthostatic Despite improvement in his fevers, leukocytosis, and hypotension, there is little data reported on the condition clinical appearance, the patient continued to complain of in the genitourinary surgical population. lightheadedness and dizziness. Upon further questioning, Orthostatic hypotension has been well documented in he reported these symptoms had started soon after surgery the bariatric surgery population [6, 7]. In one retrospective and had not improved. He complained of worsening chart review of 741 patients undergoing bariatric surgery, symptoms with distension of his neobladder prior to researchers found postoperative catheterization and relief with catheter placement. He to be a chronic and persistent issue in up to 4.2% of reported difficulty ambulating secondary to the dizziness these patients [8]. Researchers hypothesized that and several episodes of pre-. decreased sympathetic nervous system (SNS) activity Orthostatic vitals were notable for a sitting blood occurred following weight loss, manifesting as orthostatic pressure of 128/73 and of 70 bpm and a standing intolerance or hypotension. In the same study of bariatric blood pressure of 88/52 and heart rate of 85 bpm. This patients, 21 of the 31 patients experiencing orthostatic did not resolve after fluid resuscitation and resolution of intolerance underwent specialized autonomic testing that his sepsis and continued following his hospitalization. demonstrated impaired SNS activation with a Valsalva

Journal of Case Reports and Images in Urology 6, 2021. J Case Rep Images Urol 2021;6:100013Z15LG2021. Galloway et al. 3 www.ijcriurology.com maneuver [8]. The authors suggested that these patients after his infection improved, making this infection an may have had a pre-existing unlikely cause of his orthostatic hypotension. unmasked by significant weight loss and given our In , patients with spinal cord patient’s significant weight loss, this may have been a experience hypertension with significant contributor to his symptoms. and bladder irritation due to increased sympathetic Urinary diversion with ileum is associated with many nervous system stimulation. While the clinical scenarios metabolic complications including a hyperchloremic, are very different, the triggering of autonomic dysreflexia hypokalemic, metabolic acidosis [9]. Our patient was with bladder overdistention could provide a corollary to acidotic with a pH of 7.29 during his postoperative explain our patient’s symptoms. In our case, the patient admission. Nearly nine months later, he continued to insisted that his hypotensive symptoms worsened with have a metabolic acidosis with a pH of 7.30. Additionally, neobladder distention and improved with catheterization. the ileum is responsible for absorption of water, bile While speculative, perhaps some of his symptoms are salts, and B12. Although there is little literature on the related to similar neural pathways with parasympathetic relationship of these metabolic sequelae, they may be nerve involvement. Further investigation with formal associated with postoperative orthostatic hypotension. urodynamic testing and concomitant blood pressure Another study of bariatric surgery patients monitoring could have helped further define this demonstrated that high protein diets can result in proposed etiology of his symptoms. decreased norepinephrine levels, which can decrease venous return to the heart and affect cardiac output. This decrease in norepinephrine can lead to orthostatic CONCLUSION hypotension [10]. Patients undergoing radical cystectomy and ileal neobladder creation, including our patient, Literature searches have not identified other are often instructed to eat a protein-rich diet following instances of similar cases or an obvious explanation for surgery to help combat the catabolism seen after this our patient’s symptoms following radical cystectomy complex surgery. It is possible that a high-protein diet and intracorporeal neobladder creation. In summary, and the resulting changes in autonomic signaling may the etiology of this patient’s dysautonomia is likely have contributed to our patient’s symptoms. multifactorial and could be related to postoperative Lymph node dissection may be associated with weight loss with decreased SNS response, surgical postoperative dysautonomia. One recent case series disruption of important autonomic neural pathways described two cases of patients with testicular and during the lymphadenectomy, and unusual autonomic renal masses that underwent retroperitoneal lymph dysregulation related to distention of the ileal neobladder. node dissection and subsequently developed orthostatic Awareness of this case could help future clinicians who hypotension [11]. These cases were thought to be due to encounter similar scenarios and perhaps lead to further the extent of sympathetic denervation that occurred with investigation of this unusual phenomenon related to significant dissection of the superior mesenteric ganglia orthotopic continent urinary diversions. and fibers near the spinal muscular atrophy (SMA). Authors theorized that the sympathetic denervation of the SMA ganglia led to an inability of the bowel to REFERENCES vasoconstrict, thus impairing venous return when a patient stands or sits up. Our patient did not have a 1. Hanada M, Tawara Y, Miyazaki T, et al. Incidence of significant superior mesenteric ganglion dissection but orthostatic hypotension and cardiovascular response did have an extended pelvic and iliac lymphadenectomy to postoperative early mobilization in patients undergoing cardiothoracic and abdominal surgery. 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********* Conflict of Interest Authors declare no conflict of interest. Author Contributions Data Availability Lan Anh S Galloway – Conception of the work, Design All relevant data are within the paper and its Supporting of the work, Acquisition of data, Analysis of data, Information files. Interpretation of data, Drafting the work, Revising the work critically for important intellectual content, Final Copyright approval of the version to be published, Agree to be © 2021 Lan Anh S Galloway et al. This article is distributed accountable for all aspects of the work in ensuring that under the terms of Creative Commons Attribution questions related to the accuracy or integrity of any part License which permits unrestricted use, distribution of the work are appropriately investigated and resolved and reproduction in any medium provided the original Christopher P Dall – Conception of the work, Design of the author(s) and original publisher are properly credited. work, Acquisition of data, Analysis of data, Interpretation Please see the copyright policy on the journal website for of data, Drafting the work, Revising the work critically more information. for important intellectual content, Final approval of the version to be published, Agree to be accountable for all

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