A large congenital splenic pseudocyst in an adolescent treated surgically after failed percutaneous drainage

A large congenital splenic pseudocyst in an adolescent treated surgically after failed percutaneous drainage

Journal of Pediatric Surgery Case Reports 35 (2018) 14–16 Contents lists available at ScienceDirect Journal of Pediatric Surgery Case Reports journa...

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Journal of Pediatric Surgery Case Reports 35 (2018) 14–16

Contents lists available at ScienceDirect

Journal of Pediatric Surgery Case Reports journal homepage: www.elsevier.com/locate/epsc

A large congenital splenic pseudocyst in an adolescent treated surgically after failed percutaneous drainage

T

Ahad Anjuma, Jeremy T. Neumanb, Richard Sidlowc,∗ a

Department of Pediatrics, Staten Island University Hospital—Northwell Health, Staten Island, NY, USA Division of Pediatric Radiology, Department of Radiology, Staten Island University Hospital— Northwell Health, Staten Island, NY, USA c Division of Pediatric Hospitalist Medicine, Department of Pediatrics, Staten Island University Hospital—Northwell Health, Staten Island, NY, USA b

A R T I C LE I N FO

A B S T R A C T

Keywords: Spleen Pseudocyst Percutaneous drainage Marsupialization

Symptomatic splenic cysts have historically been addressed using different surgical approaches. Percutaneous drainage, while desirable due to being less invasive than open or laparoscopic surgery, may not provide definitive treatment for every patient. We describe below an adolescent patient with a congenital splenic pseudocyst in which percutaneous drainage failed requiring subsequent laparoscopic marsupialization. The merits of each approach are discussed.

1. Introduction In the pediatric population, cystic lesions of the spleen are uncommon. In underdeveloped countries they are true cysts most commonly due to parasitic infections, whereas in developed countries they are usually either congenital or traumatic pseudocysts [1,2]. Splenic pseudocysts (SP) are usually incidentally found and are asymptomatic. When symptomatic, they tend to be large, with the most common symptom being left upper quadrant abdominal pain. Nausea, vomiting, or early satiety may accompany the pain if the SP compresses the stomach or other gastrointestinal organs. Cough, pleuritic chest pain, or dyspnea may occur if the SP compresses the diaphragm or phrenic nerve [3]. The most serious of complications of a SP is rupture with resulting peritonitis [4]. To diagnose SP, ultrasound is used first to define the number, dimensions, and volume of the cyst(s), in addition to elucidating its hyper/hypoechoic nature and the presence/absence of calcifications. Computed tomography is also useful for displaying trabeculations, septations, and better characterization of calcifications [5]. Definitive diagnosis of a SP is made when fluid and parenchymal contents of the cyst are collected analyzed pathologically [3]. Much of the recent literature concerned with SP uses the Martin system to classify these cysts histologically, with the presence of an epithelial versus a fibrous lining determining whether the cyst is true or false, respectively [1–3]. Management of SP depends on the size of the cyst and severity of symptoms. Cysts less than five centimeters in size in their greatest dimension are typically managed conservatively, whereas those larger than five centimeters in size in their greatest dimension or patients who



experience significant symptoms as those mentioned above are managed surgically [6]. Treatment options include percutaneous drainage with or without sclerotherapy, marsupialization, cyst decapsulation, and partial or total splenectomy that is performed open or laparoscopically. We present below a case of a SP occurring in an adolescent that required surgical intervention following prior percutaneous drainage. 2. Case report A 17-year-old female with a past medical history of sickle cell trait, asthma, and anxiety disorder presented to the Emergency Room with left upper quadrant abdominal pain. The patient stated that the pain started four months ago and had been progressively worsening. The pain was described as sharp, crampy, intermittent, with radiation to the back. Upon review of systems the patient admitted to a dry cough and nausea. The patient denied trauma, fever, chills, night sweats, weight loss, vomiting, early satiety, dysuria, as well as any relevant travel history or animal exposures. The patient's mother had a history of polycystic kidney/liver disease. Her physical examination was normal, including vital signs, except for moderate left upper quadrant tenderness to light palpation. No splenomegaly was noted. The patient's laboratory values were within normal limits as were anteroposterior and lateral chest X-rays. Ultrasound of the abdomen showed a 9.1 cm by 5.1 cm by 5.3 cm spleen with a solitary 4.8 cm by 4.9 cm by 5.7 cm hypoechoic simple splenic cyst with an estimated volume of 69 cubic centimeters (Fig. 1). The findings on computed

Corresponding author. Department of Pediatrics, Staten Island University Hospital—Northwell Health, 475 Seaview Avenue, 10305, Staten Island, NY, USA. E-mail addresses: [email protected], [email protected] (R. Sidlow).

https://doi.org/10.1016/j.epsc.2018.05.009 Received 6 May 2018; Received in revised form 15 May 2018; Accepted 18 May 2018 Available online 23 May 2018 2213-5766/ © 2018 Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).

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appeared on CT imaging—retroperitoneal adhesions other than splenocolic and phrenicosplenic ligaments were found and taken down. The cyst itself was bulging out from splenic tissue and was discolored compared to the normal splenic parenchyma. Intra-operative ultrasound was used to confirm the cyst's location and cautery was used to create a hole in the cyst which was then drained, widened, followed by wall sampling and electrocoagulation for hemostasis. Once marsupialized, omental packing of the cystic cavity was performed. Pathologic analysis of the surgical specimens confirmed that the splenic lesion was a pseudocyst. The patient has had no recurrence of the SP following the marsupialization procedure. 3. Discussion While splenic cystic lesions can be due to parasitic or bacterial infections, neoplasms, hemangiomas, or be congenital in nature, SP on a worldwide basis are most often acquired traumatically [2]. The splenic adhesions noted intraoperatively, in addition to the pseudocyst itself, may have been post-traumatic in nature—however, the lining of the pseudocyst was noted intraoperatively to be trabeculated, arguing a congenital origin [2]. Despite the patient's family history of polycystic kidney disease, no reports of splenic cysts have been reported in association with this entity. The decision to initially drain the SP was made due to her severe pain. This was with the foreknowledge of the high risk of rapid reaccumulation and was a temporizing measure prior to a performing a definitive cystectomy/marsupialization procedure [3]. Percutaneous ethanol sclerotherapy after drainage has been used with variable success in both children and adults for both congenital and post-traumatic splenic cysts of varying sizes [7–12]. This procedure often must be repeated many times to be successful, however. In patients with multiple comorbidities or for those with contraindications to surgery, drainage with or without sclerotherapy may be the only option [13]. One other case of pediatric SP that was managed with percutaneous drainage also experienced rapid re-accumulation of fluid. That patient eventually received surgical decapsulation with permanent resolution of symptoms [14]. There are also cases in the literature of SP's recurring following laparoscopic decapsulation of the cyst [15]. With a more aggressive approach towards de-roofing the cyst, this can potentially be avoided. However, limitations to this approach are excessive size of the cyst, the cyst being completely covered by splenic parenchyma, its location near the splenic hilum, or if there are multiple cysts present [3]. In these situations, partial or total splenectomy must be considered despite however the immunologic and hematopoietic role that the spleen plays in the pediatric population.

Fig. 1. Transverse sonographic image of the spleen shows a well-defined intrasplenic cyst with internal debris (star). Note lack of internal vascularity.

Fig. 2. Axial computer tomographic image with intravenous contrast shows the same intrasplenic cyst (star).

4. Conclusion

tomography (CT) of the abdomen and pelvis with intravenous contrast were consistent with the abdominal ultrasound, with the solitary cyst's contents having simple fluid density (Fig. 2). Management options were discussed with the patient and she opted for percutaneous drainage of the cyst. Sixty milliliters of fluid was drained and upon analysis was negative for malignant cells, red blood cells, white blood cells, with subsequent microbiologic cultures being negative. A drainage catheter was placed and monitored for output while pain control was provided. The catheter was to be removed when the output was less than 20 cubic centimeters for two consecutive days. One day after catheter removal, the patient returned to the Emergency Room complaining of recurrence of the left upper quadrant pain that she had experienced prior to the drainage procedure. An ultrasound revealed a 4.5 cm by 4.8 cm by 3.9 cm complex splenic cyst without vascular flow. It was then decided to laparoscopically remove the cyst. Intraoperatively the spleen was more lateral and cephalad than it

The choice of surgical approach for the treatment of large splenic cystic lesions requires careful anatomic/radiographic evaluation and individualization based on a patient's comorbidities and the presumed etiology of the cyst. Percutaneous drainage for this entity remains an option for those whom surgery is not an option. However, in light of our case and others in the medical literature, when a surgical approach is possible, it should be pursued. Patient consent Consent to publish the case report was not obtained. This report does not contain any personal information that could lead to the identification of the patient. Funding No funding or grant support. 15

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Authorship

[6] Delforge X, Chaussy Y, Borrego P, Abbo O, Sauvat F, Ballouhey Q, Irtan S, Arnaud A, Ibtissam K, Panait N, Rodesch G, Steyaert H, Schneider A, Dubois R, Mesureur S, Haraux E, Buisson P. Management of nonparasitic splenic cysts in children: a French multicenter review of 100 cases. J Pediatr Surg 2017;52(9):1465–70. http://dx.doi. org/10.1016/j.jpedsurg.2017.01.054. [7] Mun SW, Lim TJ, Hwang EH, Lee YJ, Jeon UB, Park JH. A case of post-traumatic pseudocyst in the spleen successfully treated with alcohol sclerotherapy. Pediatr Gastroenterol Hepatol Nutr 2015;18(4):276–9. [8] Anon R, Guijarro J, Amoros C, Gil J, Bosca MM, Palmero J, Benages A. Congenital splenic cyst treated with percutaneous sclerosis using alcohol. Cardiovasc Intervent Radiol 2006;29:691–3. [9] De Caluwe D, Phelan E, Puri P. Pure alcohol injection of a congenital splenic cyst: a valid alternative? J Pediatr Surg 2003;38(4):629–32. [10] Wu HM, Kortbeek JB. Management of splenic pseudocysts following trauma: a retrospective case series. Am J Surg 2006;191(5):631–4. [11] Volk M, Rogler G, Strozer M, Lock G, Manke C, Feuerback S. Post-traumatic pseudocyst of the spleen: sclerotherapy with ethanol. Cardiovasc Intervent Radiol 1999;22:246–8. [12] Akhan O, Baykan Z, Oguzkurt L, Sayek I, Ozmen MN. Percutaneous treatment of a congenital splenic cyst with alcohol: a new therapeutic approach. Eur Radiol 1997;7:1067–70. [13] Moir C, Guttman F, Jequier S, Sonnino R, Youssef S. Splenic cysts: aspiration, sclerosis, or resection. J Pediatr Surg 1989;24(7):646–8. http://dx.doi.org/10. 1016/s0022-3468(89)807110. [14] Lappin J, Corbally MT, Guiney EJ. Splenic pseudocyst: aspiration or cyst decapsulation. Ir J Med Sci 1992;161(2). 48-48 https://doi.org/10.1007/BF02942082. [15] Fisher JC, Gurung B, Cowles RA. Recurrence after laparoscopic excision of nonparasitic splenic cysts. J Pediatr Surg 1989;24(7):646–8https://doi.org/10.1016/ S0022-3468(89)80711-0.

All authors attest that they meet the current ICJME criteria for authorship. Conflict of interest The following authors have no financial disclosures: (AA, JT, RS). References [1] Juma US, Ayubu MF. Giant benign non-traumatic splenic pseudo cyst: an atypical cause of left upper quadrant abdominal pain. Int J Med Med Sci 2013;5(12):510–3. http://dx.doi.org/10.5897/IJMMS2013.0996. [2] Sinha CK, Agrawal M. Nonparasitic splenic cysts in children: current status. Surgeon J Roy Coll Surg Edinb Ireland 2011;9(1):49–53. http://dx.doi.org/10.1016/j.surge. 2010.08.005. [3] Galyfos G, Touloumis Z, Palogos K, Stergios K, Chalasti M, Nikolaos K, Lavant L. Oversized pseudocysts of the spleen: report of two cases. Int J Sur Case Rep 2014;5(2):104–7. http://dx.doi.org/10.1016/j.ijscr.2013.11.006. [4] Martinaityte I, Pivoriunas J, Jurgaitis J, Paskonis M, Ostapenka A, Strupas K. Posttraumatic splenic pseudocyst: a case report and review of the literature. Lithuanian surgery 2009;7(3–4):69–73. [5] Senol M, Ozdemir H, Sahiner IT, Ozdemir ZU. Non-traumatic pseudocyst of the spleen: a case report. Kafkas J Med Sci 2014;4(2):79–83. http://dx.doi.org/10. 5505/kjms.2014.24008.

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