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      Spontaneous Retroperitoneal Hemorrhage (Wunderlich Syndrome) due to Large Upper Pole Renal Angiomyolipoma: Does Robotic-Assisted Laparoscopic Partial Nephrectomy Have a Role in Primary Treatment?

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          Abstract

          Spontaneous rapture with consequent retroperitoneal hemorrhage (Wunderlich's syndrome) is the complication mostly feared from large renal angiomyolipomas (RAMLs). In hemodynamic stable patients, minimal invasive therapies have superseded open surgery as the mainstay of treatment, with contemporary cases mostly treated by selective arterial embolization. Robotic-assisted laparoscopic partial nephrectomy (RALPN) is an established minimal access treatment that has been used in the past for benign and malignant lesions of the kidney in the elective setting, but rarely in urgent situations as primary treatment. We present a case of a ruptured RAML in a young female treated effectively by RALPN.

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          The management of renal angiomyolipoma.

          J E Oesterling, E Fishman, S Goldman … (1986)
          Article 0 comments Cited 71 times – based on 0 reviews     Review now
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            Contemporary diagnosis and management of renal angiomyolipoma.

            Caleb P Nelson, Martin G. Sanda (2002)
            We present a comprehensive resource that summarizes contemporary advances relevant to the clinical management of renal angiomyolipoma. A MEDLINE search was done using the key words angiomyolipoma, hemangioma or lipoma and kidney neoplasm, and therapeutic embolization. References from these articles were reviewed to identify additional relevant source material. We identified 13 series published since 1986 comprising 336 patients as well as 24 reports of therapeutic embolization for angiomyolipoma, comprising 76 patients. To our knowledge we report the largest compilation series to date. Angiomyolipoma is generally benign, although an uncommon subtype (epithelioid angiomyolipoma) may behave more aggressively. Tuberous sclerosis associated angiomyolipoma tends to be larger, multiple and more likely to cause spontaneous hemorrhage than the sporadic entity. Tumors that hemorrhage tend to be larger. Computerized tomography or magnetic resonance is usually sufficient for diagnosis. Biopsy is rarely useful. Primary indications for intervention include symptoms such as pain or bleeding or suspicion of malignancy. Prophylactic intervention is justifiable for large tumors, in females of childbearing age or in patients in whom followup or access to emergency care may be inadequate. Recent advances that have affected management include improved understanding of tuberous sclerosis complex and angiomyolipoma genetics, the identification of molecular markers that facilitate histopathological diagnosis, and the refinement of embolization and partial nephrectomy techniques. Although some cases of angiomyolipoma may require complete nephrectomy, most can be managed by conservative nephron sparing approaches. Ongoing research into the molecular biology and clinical behavior of angiomyolipoma may improve our ability to manage these lesions.
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              Renal angiomyolipoma: long-term results after arterial embolization.

              Nishita Kothary, Michael C Soulen, Timothy Clark … (2005)
              Selective arterial embolization of renal angiomyolipomas (AMLs) was performed to prevent hemorrhage in patients with AMLs larger than 4 cm. This study was conducted to evaluate the long-term efficacy of AML embolization. Nineteen patients underwent embolization for 30 renal AMLs between July 1991 and June 2002. Of these, 10 patients had tuberous sclerosis (TS) with multiple AMLs and nine patients had a solitary sporadic AML. Embolization was performed with use of ethanol mixed with iodized oil (Ethiodol) in 29 tumors; coils were used in addition to the ethanol/Ethiodol mixture in one case. All tumors were completely embolized according to angiographic criteria including vascular stasis and absence of arterial feeders. The efficacy of embolization was determined over a mean follow-up period of 51.5 months (range, 6-132 months). Recurrence was defined as an increase in tumor size of greater than 2 cm on follow-up imaging and/or recurrent symptoms that required repeat embolization. An institutional review board exemption was obtained to perform this retrospective study. Embolization of the renal AMLs was technically successful in all 19 patients and for all 30 lesions. AML recurrence was noted in 31.6% of patients (n = 19) and for 30% of lesions overall (n = 9). Six of 10 patients in the TS group had AML recurrences. No recurrences occurred in the patients with sporadic AML. In the TS group of 10 patients, there was a total of 21 AMLs and the overall tumor recurrence rate was 42.9% (nine of 21). Six lesions in four patients had to be reembolized because of recurrent symptoms, including one hemorrhage, and three lesions in two patients required repeat embolization because of a greater than 2 cm increase in size. The median time interval from embolization to recurrence was 78.7 months (range, 13-132 months). Statistical testing with use of the Fisher exact test demonstrated that patients with TS were significantly more likely to develop recurrence than those without TS (P = .01). Transarterial embolization is effective in preventing hemorrhage in patients with renal AMLs. However, long-term follow-up revealed a high AML recurrence rate in patients with TS. Lifelong surveillance for recurrence after AML embolization is essential in patients with TS.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Case Rep Urol
                Journal ID (iso-abbrev): Case Rep Urol
                Journal ID (publisher-id): CRIM.UROLOGY
                Title: Case Reports in Urology
                Publisher: Hindawi Publishing Corporation
                ISSN (Print): 2090-696X
                ISSN (Electronic): 2090-6978
                Publication date (Print): 2013
                Publication date (Electronic): 11 September 2013
                Volume: 2013
                Electronic Location Identifier: 498694
                Affiliations
                1Department of Urology, Athens Medical Center, 15125 Athens, Greece
                2Department of Urology, Laiko General Hospital, University of Athens, 18535 Athens, Greece
                Author notes
                *Achilles Ploumidis: aploumidis@ 123456yahoo.gr

                Academic Editors: S. K. Hong, A. Marte, T. J. Murtola, and J. Park

                Article
                DOI: 10.1155/2013/498694
                PMC ID: 3784227
                SO-VID: 21e9c8ea-543a-4bef-b662-b1c9cacf9a9d
                Copyright © Copyright © 2013 Achilles Ploumidis et al.
                License:

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 30 June 2013
                Date accepted : 12 August 2013
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Urology
                Data availability:
                ScienceOpen disciplines: Urology

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