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UNUSUAL CASE
Year : 2021  |  Volume : 17  |  Issue : 3  |  Page : 379-381
 

Refractory lymphatic ascites following laparoscopic management of renal lymphangiectasia: An unusual presentation


Department of Urology, All India Institute of Medical Sciences, New Delhi, India

Date of Submission26-Nov-2020
Date of Decision31-Jan-2021
Date of Acceptance04-Mar-2021
Date of Web Publication08-Apr-2021

Correspondence Address:
Sumit Saini
Department of Urology, All India Institute of Medical Sciences, 5030, Teaching Block, Ansari Nagar, New Delhi - 110 029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jmas.JMAS_306_20

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 ¤ Abstract 

Renal lymphangiectasia characterised by either unilocular or multilocular cystic lesion in and around the kidney is an uncommon condition. Presentation of these lesions is quite varied, which along with its uncommon occurrence adds to the challenges in the management of this condition. Most of these cases are managed conservatively and very rarely need any intervention. We present an unusual complication of refractory lymphatic ascites following laparoscopic deroofing of a unilocular renal lymphangiectasia in a 21-year-old gentleman who presented with left flank pain. The ascitic fluid analysis suggested non-chylous lymphatic ascites. The surgical outcome was rather exasperating for the patient than the disease itself. Hence, in the interest of the patient with minimal symptoms, if the imaging is highly suggestive of renal lymphangiectasia, either no intervention or the least invasive procedures should be attempted, whenever possible.


Keywords: Laparoscopic deroofing, non-chylous ascites, refractory lymphatic ascites, renal lymphangiectasia


How to cite this article:
Saini S, Nayak B, Singh P, Seth A. Refractory lymphatic ascites following laparoscopic management of renal lymphangiectasia: An unusual presentation. J Min Access Surg 2021;17:379-81

How to cite this URL:
Saini S, Nayak B, Singh P, Seth A. Refractory lymphatic ascites following laparoscopic management of renal lymphangiectasia: An unusual presentation. J Min Access Surg [serial online] 2021 [cited 2021 Aug 4];17:379-81. Available from: https://www.journalofmas.com/text.asp?2021/17/3/379/313394



 ¤ Introduction Top


Renal lymphangiectasia characterised by either uni/multilocular cystic lesion in and around the kidney is an uncommon condition. We present an unusual complication of refractory lymphatic ascites following laparoscopic management of unilocular renal lymphangiectasia.


 ¤ Case Report Top


A 21-year-old otherwise healthy male with no significant medical or surgical history presented elsewhere with mild left flank pain. Initial ultrasonogram followed by contrast-enhanced computed tomography (CT) scan suggested left peri-renal hypodense (8–10 HU) cystic lesion/well-marginated collection with no contrast extravasation in delayed films. It was misinterpreted initially as idiopathic left peri-nephric urinoma. Pigtail was placed which persistently drained large volumes. This was followed by retrograde pyelogram and double-J stenting which too did not help. He later underwent laparoscopic deroofing at another centre with the diagnosis of left renal lymphangiectasia [Figure 1]. The drain was removed after 2 days as its output was minimal (<30 ml/day). Following drain removal, he developed debilitating ascites with respiratory distress which progressed over 3 weeks as depicted [Figure 2]. An intra-peritoneal drain was then placed. The drain output was quite high (1500–2200 ml) for more than 8 weeks and the patient presented to our centre at this stage. Analysis of clear, straw-coloured ascitic fluid included the fluid cytology which was negative for atypical/malignant cells and had triglycerides of <12 mg/dl even after consumption of high-fat diet, creatinine level of 0.8 mg/dl, serum ascites albumin gradient >1.1 mg/dl and normal serum and ascitic fluid amylase levels. A peritoneovenous shunt with a reversed saphenous vein was attempted, which was unsuccessful. Fortunately for the patient, after around 12 weeks, the drain output started declining and the drain was removed after 4 weeks. Follow-up imaging with ultrasonography (USG) at 6 months following drain removal revealed no ascites with bilateral normal kidneys and no peri-nephric collection.
Figure 1: Pre-operative computed tomography scan pictures of (L) renal lymphangiectasia (a) Axial cuts, (b) Saggital cuts

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Figure 2: Post-operative computed tomography scan pictures of lymphatic ascites (a) Axial cuts, (b) Saggital cuts

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 ¤ Discussion Top


With varied presentations and nomenclature, renal lymphangiectasia is an uncommon benign condition. These lesions, which may either be unilateral or bilateral, tend to occur more frequently in males, without any age predilection. Renal lymphangiectasia occurs secondary to congenital or very rarely acquired miscommunication between the renal and retroperitoneal lymphatic channels.[1],[2] As the name implies, lymphangiectasia or dilatation of lymphatic vessels occurs due to the lymph accumulation in and around the kidneys, which is seen radiographically as unilocular or multilocular cysts.[1],[2]

In majority of cases, this condition is diagnosed incidentally but can present with flank mass, pain, hypertension, haematuria, chyluria, renal vein thrombosis, ureteral obstruction, lower limb oedema, ascites, pleural effusion or even chylothorax.[2] Similar to its aetiopathogenesis, to date, no consensus has been reached regarding its optimal management. Management includes conservative options such as follow-up when asymptomatic, aspiration with or without sclerotherapy or pigtail placement to more invasive options such as laparoscopic cyst deroofing, marsupialisation, excision or very rarely even nephrectomy.[2]

Before any intervention, the certainty with which this condition is diagnosed provides the clinician as well as the patient an opportunity to assess the associated risks/benefits. There is an emerging role of contrast-enhanced ultrasound (CEUS), in addition to CT scan and magnetic resonance imaging (MRI) for the diagnosis of this condition. CEUS using gas microbbubles provides a dynamic study with characteristics between greyscale USG and CECT/MRI. It can rule out other renal parenchymal pathologies and help establish the diagnosis of renal lymphangiectasias in equivocal cases.[3]

Laparoscopic deroofing for renal lymphangiectasia is a well-described procedure and works on the basic principle of lymph absorption from the peritoneal surface.[4] Unfortunately, our patient developed debilitating ascites following this procedure which necessitated abdominal drain placement. Although the exact reason could not be ascertained, the very basic principle of peritoneal absorption on which this procedure relied likely got overwhelmed and probably could have led to this unusual complication.

Although intervention was justified for this large symptomatic renal lymphangiectasia, the famous aphorism primum non nocere (first, do no harm) should always be borne in mind.[5]

In conclusion, with a diligent interpretation of imaging as a pre-requisite, for any suggestion of renal lymphangiectasia in a patient with minimal symptoms, either no intervention or the least invasive procedures should be attempted whenever possible, in the interest of the patient. This unusual and debilitating complication of refractory lymphatic ascites must be taken into consideration for a seemingly straightforward procedure like laparoscopic cyst deroofing.

Consent for publication

Written and informed consent was obtained from the patient.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
 ¤ References Top

1.
Chaabouni A, Rebai N, Fourati M, Rekik S, Chabchoub K, Slimen MH, et al. Cystic lymphangioma of the kidney: Diagnosis and management. Int J Surg Case Rep 2012;3:587-9.  Back to cited text no. 1
    
2.
Leite AF, Venturieri B, de Araújo RG, Silva EJ, Elias Junior J. Renal lymphangiectasia: Know it in order to diagnose it. Radiol Bras 2016;49:408-9.  Back to cited text no. 2
    
3.
Guadagni S, Gianardi D, Palmeri M, Furbetta N, Di Franco G, Bianchini M, et al. Contrast enhanced ultrasound for the diagnosis of bilateral renal lymphangiectasia: Literature review and contrast enhanced ultrasound findings. J Ultrasound 2020. doi: 10.1007/s40477-020-00529-9. Epub ahead of print. PMID: 32886346..  Back to cited text no. 3
    
4.
Meyyappan RM, Ravikumar S, Gopinath M. Laparoscopic management in a rare case of bilateral perirenal lymphangiomatosis. Indian J Urol 2013;29:73-4.  Back to cited text no. 4
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5.
Smith CM. Origin and uses of primum non nocere–above all, do no harm! J Clin Pharmacol 2005;45:371-7.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2]



 

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