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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 4  |  Page : 163-165

Laparoscopic resection of an adrenaline secreting para-aortic paraganglioma


1 Department of Surgery, Command Hospital, Pune, Maharashtra, India
2 Department of Oncosurgery, D. Y. Patil Medical College and Hospital, Pune, Maharashtra, India

Date of Submission07-Dec-2020
Date of Decision15-Jul-2021
Date of Acceptance18-Jul-2021
Date of Web Publication3-Dec-2021

Correspondence Address:
Prof. Samir Gupta
Department of Oncosurgery, D. Y. Patil Medical College and Hospital, Pune - 411 040, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JCRP.JCRP_18_21

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  Abstract 


Paragangliomas are rare neuroendocrine tumors arising from extra-adrenal chromaffin tissue. About half of paragangliomas occur in the para-aortic region, and only a few are functional. Although surgical resection remains the definitive treatment, only a few cases of laparoscopic resection of para-aortic paragangliomas have been reported. Here, we report a case of catecholamine-secreting left para-aortic paraganglioma resected laparoscopically. Laparoscopic resection of these tumors is associated with lesser blood loss, lesser postoperative pain, and shorter hospital stays. The close cooperation between the surgeon and anesthesiologist is highlighted.

Keywords: Functional, laparoscopic, paraganglioma


How to cite this article:
Badruddin B, Gupta S. Laparoscopic resection of an adrenaline secreting para-aortic paraganglioma. J Cancer Res Pract 2021;8:163-5

How to cite this URL:
Badruddin B, Gupta S. Laparoscopic resection of an adrenaline secreting para-aortic paraganglioma. J Cancer Res Pract [serial online] 2021 [cited 2022 Jun 30];8:163-5. Available from: https://www.ejcrp.org/text.asp?2021/8/4/163/331649




  Introduction Top


Paragangliomas are rare neuroendocrine tumors arising from a widely dispersed collection of specialized neural crest cells. It has been estimated that 25% of paragangliomas arise outside the adrenal glands.[1] Paragangliomas may be functional and result in symptoms of excess catecholamine production. Laparoscopic resection of these tumors has been infrequently reported. We report such a case which was successfully resected laparoscopically. This case highlights the skilled anesthesia care required for the management of such patients.


  Case Report Top


A 32-year-old female with no known comorbidities presented with headache, palpitation, and dyspnea on exertion. She was found to have hypertension and hence started on antihypertensives. Two-dimensional echo showed global hypokinesia, diastolic dysfunction, and ejection fraction of 40%. She was managed for left ventricular dysfunction and cardiogenic failure. During the evaluation, imaging revealed a mass lesion in the left para-aortic region. On a contrast-enhanced computerized tomography scan, a 37 mm × 40 mm well-defined hypoechoic round to oval lesion was seen in the left para-aortic region, anterior to the left ureter and psoas muscle, and inferior to the level of the lower pole of the kidney [Figure 1].
Figure 1: Computed tomography scan showing the location of the tumor

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Pheochromocytoma profile showed elevated epinephrine, metanephrines, and chromogranin-A. She had orthostatic hypotension and was started on both alpha-blockers and beta-blockers. Her symptoms improved within 2 weeks with the combined blockade.

She underwent complete laparoscopic excision of the mass under General Anesthesia(GA). She was placed in the right lateral decubitus position, and three-port laparoscopic mobilization was done. A 10-mm camera port was placed in the right paramedian position just above the level of the umbilicus. One 10-mm port was placed in the left hypochondrium and another 5-mm port in the midline in the suprapubic area. The mesocolon of the descending colon was mobilized away from the tumor. The left ureter was identified and safeguarded. Surrounding adherent tissue was separated from the tumor using an Ultracision Harmonic Scalpel (Ethicon Endosurgery, Johnson and Johnson). Multiple feeding vessels were clipped and divided [Figure 2]. The tumor was retrieved using an endobag after extending the left hypochondrium 10-mm port incision. The gross tumor was 6 cm × 4 cm in size [Figure 3]. The total operative time was 60 min, and the intraoperative blood loss was around 100 mL.
Figure 2: Laparoscopic dissection of the tumor with harmonic scalpel

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Figure 3: Photograph of the specimen

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This case was challenging for the anesthesia team. Adequate hydration was ensured in the preoperative period with normal saline. On-table preparation began with an arterial blood pressure (BP) line placed under local anesthesia. The baseline BP hovered around 110/70 mmHg. Immediately, after intubation, the BP increased to 240/120 mmHg. This was managed with continuously titrated doses of nitroglycerine and dexmedetomidine infusions and brought under control. Further BP spikes of up to 200 mmHg occurred during the creation of a pneumoperitoneum and also frequently during handling of the tumor. These were managed with added infusions of sodium nitroprusside titrated according to the BP. After division of the feeding vessels toward the end of dissection, the BP stabilized near baseline values. The feeding vessels could not be tackled early in the surgery due to surrounding adhesions. However, after complete excision of the tumor, the patient went into hypotension and hence was started on inotropic support with noradrenaline and moved to the intensive care unit (ICU).

Postoperatively, the patient continued to be hypotensive in the ICU for 48 h and was managed with inotropes. She was discharged on the fourth postoperative day once her BP had stabilized. The histopathology was consistent with paraganglioma.


  Discussion Top


Paragangliomas are neuroendocrine tumors arising from extra-adrenal chromaffin tissue, and account for 10%–25% of pheochromocytomas. The incidence of catecholamine-secreting functional paragangliomas is 15%–24% of all paragangliomas.[1] The most commonly secreted catecholamine is norepinephrine, and the classic triad of catecholamine excess is headache, sweating, and palpitations. However, this triad may be absent and patients can be asymptomatic or symptoms can be vague.[2] Paragangliomas are most commonly present in the organ of Zuckerkandl at the aortic bifurcation.[3]

Men are affected more frequently than women, and most patients are between the ages of 30 and 45 years. Paragangliomas synthesize and store catecholamines, which include norepinephrine (noradrenaline), epinephrine (adrenaline), and dopamine. Elevated plasma and urinary levels of catecholamines and the methylated metabolites, metanephrines are the cornerstone for the diagnosis.[1] Computed tomography, magnetic resonance imaging, and ultrasonography are useful in detecting a retroperitoneal mass and can delineate its location, outline, and its relationship with the surrounding organs.[4]

The possibility for malignant transformation of paragangliomas makes surgical excision the treatment of choice.[2],[4] There are only rare reports of laparoscopic excision of para-aortic paragangliomas. There are no randomized studies which have compared open to laparoscopic approaches for excision of paragangliomas. A series of cases done by laparoscopic approach mostly focused on adrenal paragangliomas,[5] and it reported lesser blood loss, postoperative pain, and duration of stay by the laparoscopic approach compared to open surgery. The two most commonly used laparoscopic approaches are the lateral transabdominal/transperitoneal approach and posterior retroperitoneal approach. The former allows for intra-abdominal evaluation and provides more space for dissecting larger tumors.[6],[7],[8]

A three- or four-port approach can be used with colonic mobilization or duodenal kocherization as required. The ureter and inferior mesenteric vessels need to be safeguarded, especially in left-sided tumors as was the case in our patient. Early clipping of the feeding vessels can lead to better intra-operative management of BP; however, this was not possible in our case. Seeding and recurrence of tumors in the tumor bed or throughout the abdominal cavity can occur if pheochromocytomas or paragangliomas are fractured during dissection, mandating precise, and gentle dissection.[9] Specimen bags used for tumor retrieval should not tear.

Nakao et al.[10] reported on the close association required between the surgeon and anesthesiologist to control changes during surgery. It is recommended to start preoperative medical treatment at least 7–14 days before surgery to normalize BP and heart rate. This should include high intake of fluid and salt to reverse the catecholamine-induced blood volume contraction. Alpha-adrenergic receptor blockers should be started 7 days preoperatively to prevent perioperative hemodynamic complications.

The main complication expected during surgery is hemodynamic instability during mobilization of the tumor. Sodium nitroprusside and nitroglycerine are the two most commonly used drugs for intraoperative control of hypertension, as they have established safety profiles. On removal of the tumor due to a fall in catecholamine levels hypotension may occur and vasopressor support may be needed to maintain normal BP.[11]

Laparoscopy helps in the delicate handling of the tumor. This has been shown to be useful in intra-operative management.[12] The excellent anesthesia team during the surgery in our case resulted in a favorable outcome. An experienced anesthesiologist who is well versed in handling such challenges is essential in managing these cases.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published, and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Melmed S, Polonsky KS, Larsen PR, Kronenberg HM. Williams Textbook ofEndocrinology .13th ed. Philadelphia: Elsevier; 2015.p. 556-73.  Back to cited text no. 1
    
2.
Goers TA, Abdo M, Moley JF, Matthews BD, Quasebarth M, Brunt LM. Outcomes of resection of extra-adrenal pheochromocytomas/paragangliomas in the laparoscopic era: A comparison with adrenal pheochromocytoma. Surg Endosc 2013;27:428-33.  Back to cited text no. 2
    
3.
Whalen RK, Althausen AF, Daniels GH. Extra-adrenal pheochromocytoma. J Urol 1992;147:1-10.  Back to cited text no. 3
    
4.
Sahdev A, Sohaib A, Monson JP, Grossman AB, Chew SL, Reznek RH. CT and MR imaging of unusual locations of extra-adrenal paragangliomas (pheochromocytomas). Eur Radiol 2005;15:85-92.  Back to cited text no. 4
    
5.
Yeo H, Roman S. Pheochromocytoma and functional paraganglioma. Curr Opin Oncol 2005;17:13-8.  Back to cited text no. 5
    
6.
Agarwal G, Sadacharan D, Aggarwal V, Chand G, Mishra A, Agarwal A, et al. Surgical management of organ-contained unilateral pheochromocytoma: Comparative outcomes of laparoscopic and conventional open surgical procedures in a large single-institution series. Langenbecks Arch Surg 2012;397:1109-16.  Back to cited text no. 6
    
7.
Lenders JW, Duh QY, Eisenhofer G, Gimenez-Roqueplo AP, Grebe SK, Murad MH, et al. Pheochromocytoma and paraganglioma: An endocrine society clinical practice guideline. J Clin Endocrinol Metab 2014;99:1915-42.  Back to cited text no. 7
    
8.
Salgaonkar H, Behera RR, Sharma PC, Chadha M, Katara AN, Bhandarkar DS. Laparoscopic resection of a large paraganglioma arising in the organ of Zuckerkandl: Report of a case and review of the literature. J Minim Access Surg 2016;12:378-81.  Back to cited text no. 8
    
9.
Li ML, Fitzgerald PA, Price DC, Norton JA. Iatrogenic pheochromocytomatosis: A previously unreported result of laparoscopic adrenalectomy. Surgery 2001;130:1072-7.  Back to cited text no. 9
    
10.
Nakao Y, Sakamoto Y, Miyamoto Y, Kurashige J, Imamura Y, Hiyoshi Y, et al. Laparoscopic resection of a subclinical functioning para-aortic paraganglioma. Int Surg 2019;104:9-12.  Back to cited text no. 10
    
11.
Tatsugami K, Eto M, Hamaguchi M, Yokomizo A, Harano M, Naito S. What affects the results of a laparoscopic adrenalectomy for pheochromocytoma? Evaluation with respect to intraoperative blood pressure and state of tumor. J Endourol 2009;23:101-5.  Back to cited text no. 11
    
12.
Hattori S, Miyajima A, Hirasawa Y, Kikuchi E, Kurihara I, Miyashita K, et al. Surgical outcome of laparoscopic surgery, including laparoendoscopic single-site surgery, for retroperitoneal paraganglioma compared with adrenal pheochromocytoma. J Endourol 2014;28:686-92.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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