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CASE REPORT Table of Contents  
Ahead of print publication
Uncommon cause of pain abdomen in an end-stage renal disease patient on hemodialysis


1 Department of Nephrology, Command Hospital Air Force, Bengaluru, Karnataka, India
2 Department of Radiology, Command Hospital Air Force, Bengaluru, Karnataka, India
3 Department of Pathology, Command Hospital Air Force, Bengaluru, Karnataka, India

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Date of Submission07-Feb-2022
Date of Decision22-Feb-2022
Date of Acceptance04-Mar-2022
Date of Web Publication25-Jun-2022
 

  Abstract 


Pain abdomen is an unusual symptom in an end-stage renal disease (ESRD) patient, and the causes of the same have not been clearly defined. The causes may be related to advanced azotemia causing esophagitis, gastritis, and peptic ulcer disease or pressure-related pain due to polycystic kidney disease. The causes may be totally unrelated to chronic kidney disease (CKD) and may present due to surgical causes such as cholelithiasis, appendicitis, diverticulitis, acute mesenteric ischemia, and others. We report an unusual case of pain abdomen in an 11-year-old child of CKD on maintenance hemodialysis (MHD) who presented with recent onset left-sided hypochondriac pain and swelling. His contrast-enhanced computed tomography abdomen showed a large hydatid cyst in the spleen, and the patient underwent partial cystectomy for the same. Histopathological examination confirmed the diagnosis of splenic hydatid cyst. The postoperative course was uneventful. There are only a few case reports of isolated splenic hydatid cyst reported from India, but to the best of our knowledge, the same has never been reported in a child with CKD on MHD making it an unusual cause of pain abdomen in ESRD patients.

Keywords: End-stage renal disease, pain abdomen, partial cystectomy, splenic hydatid cyst


How to cite this URL:
Akal RS, Jha VK, Yadav D, Sikarwar RS. Uncommon cause of pain abdomen in an end-stage renal disease patient on hemodialysis. APIK J Int Med [Epub ahead of print] [cited 2022 Oct 6]. Available from: https://www.ajim.in/preprintarticle.asp?id=348295





  Introduction Top


Pain abdomen is an unusual symptom in an end-stage renal disease (ESRD) patient, and the causes of the same have not been clearly defined. The cause may be related to advanced azotemia manifesting as acute esophagitis, gastritis, or peptic ulcer disease. The pain may be due to pressure-related symptoms of polycystic kidney disease or cyst hemorrhage. Other causes may be totally unrelated to chronic kidney disease (CKD) and are surgical causes such as cholelithiasis, appendicitis, diverticulitis, uremic colitis, and acute mesenteric ischemia.[1] Hydatid disease is a common health problem prevalent in endemic regions of sheep- and cattle-raising areas of the world, which has been reported frequently as an incidental cause of pain abdomen.[2] The most common organ involved by hydatid cysts is liver (50%–70%), followed by lungs (20%–30%). Other organs such as brain, kidney, muscles, heart, pancreas, ovaries, and orbit are infrequently involved, but practically, any organ can be affected.[3] The splenic involvement is rare and seen in <3% of total hydatid cyst cases.[4] Splenic hydatid cyst is a rare entity, and its presence in ESRD patients presenting as acute pain abdomen has never been reported.


  Case Report Top


This 11-year-old boy was a known case of dilated cardiomyopathy (ejection fraction: 25%) and CKD on maintenance hemodialysis (3/week) for the last 1 year. His vascular access was left brachiocephalic Arteriovenous fistula. He had no residual urinary function. He was admitted with complaints of pain abdomen and swelling in the left flank of 10-day duration. He had no gastrointestinal or lower urinary tract symptoms. Pain was continuous, localized, and severe. His examination revealed pallor and mild pedal edema. Blood pressure was 136/88 mmHg (on 3 antihypertensives) and pulse rate was 120/min. Per-abdomen examination revealed 8 cm × 6 cm tender swelling in the left hypochondriac region extending into the lumbar region with moderate ascites. His investigations revealed anemia with hemoglobin– 9.2 g/dl with normal Total leukocyte count (TLC) and platelets. His creatinine was 3.25 mg/dL with normal electrolytes and liver function tests. His serum albumin was low (2.8 g/dL), procalcitonin was raised (3.5 ng/mL), and viral markers were negative. His ultrasonography (USG) abdomen revealed multiloculated peritoneal collection in the left lumbar region with ascites and no evidence of chronic liver disease. His contrast-enhanced computed tomography (CT) chest and abdomen revealed a well-defined, septated, oval-shaped cystic lesion measuring 10.5 cm × 9.0 cm × 10.8 cm (anterior-posterior × transverse × cranial-caudal) with a thick heterogeneously enhancing wall (postcontrast 78 Hounsfield units) at the inferolateral aspect of spleen along with right pleural effusion and ascites [Figure 1]a and [Figure 1]b. The cyst was of recent onset as the previous USG done 6 months back had only ascites and no evidence of any cyst. His Echinococcus immunoglobulin G (IgG) enzyme-linked immunosorbent assay (ELISA) was positive (15 U/mL). Although he had no contact with animals such as sheep and dogs in the past, in view of suggestive imaging, positive serology, and raised procalcitonin levels, the diagnosis of infected splenic hydatid cyst was made, and the patient was treated with broad-spectrum intravenous antibiotics (vancomycin and meropenem) in renal modified doses. His diagnostic cyst aspiration revealed necrotic material mixed with few lymphocytes and occasional hooklets. His ascitic fluid evaluation revealed albumin of 2.1 g/dl, low serum ascites albumin gradient, total cell count of 150 cells (lymphocyte predominant), and Mycobacterium tuberculosis (MTB) polymerase chain reaction negative. His urea reduction ratio (URR) was low (54%), hence his HD duration was increased to 5 h (3/week) with increased ultrafiltration removal to treat his nephrogenic ascites. The patient underwent partial cystectomy of the splenic cyst under general anesthesia after 10 days of antibiotic use. Histopathological examination of cystectomy tissue revealed a benign hydatid cyst with middle lamellated hyaline layer [Figure 2]a and inner germinal layer having daughter cysts, calcified scolices, and refractile hooklets [Figure 2]b. Postoperative course remained uneventful. He was given albendazole 15 mg/kg/day in two divided doses for 3 months after completion of 2 weeks of antibiotics. A review at 3 months with USG did not reveal any recurrence of hydatid cyst and was pain free.
Figure 1: Contrast-enhanced computed tomography abdomen: (a) Coronal view, (b) Sagittal view) showing a well-defined thick-walled, oval-shaped cystic lesion (white arrows) with nonenhancing septations noted at the inferolateral aspect of the spleen

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Figure 2: (a) Cyst wall showing the middle lamellated hyaline layer of a hydatid cyst (black arrow) with germinal layer having daughter cysts and calcified scolices (×40; H and E); (b) Inner germinal layer with refractile hooklets (black arrow) (×400; H and E)

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  Discussion Top


Hydatid disease is a cyst-forming disease caused by infection with larval form of dog tapeworm of genus Echinococcus. The majority of the human diseases are caused mainly by two species, i.e., by Echinococcus granulosus and Echinococcus multilocularis. It is commonly seen in sheep- and cattle-raising areas of various parts of the world and is a significant health problem in India.[2] The adult worm lives in the small intestine of dogs and other canines which are definitive hosts. The ungulates such as sheep, goats, horses, and pigs are intermediate hosts, harboring the larval stage. Humans are the accidental hosts of the larval stage which form hydatid cysts.[5] The most common site of hydatid disease is liver followed by lungs. The rarer sites include brain, kidney, and thyroid, and practically, any organ can be infested. Hydatid disease of the spleen is uncommon even in endemic areas and is <3% of total cases.[3] Hydatid cysts of the spleen are predominantly asymptomatic and are diagnosed incidentally during evaluation of unrelated symptoms. Some patients may present with left hypochondriac mass and pain. In larger cysts, pressure symptoms or rupture of the cyst may occur.[6],[7] Our patient also presented with pain and mass in the left hypochondriac region with features suggestive of infected hydatid splenic cyst.

Echinococcus IgG ELISA has good sensitivity and specificity (75%–100%) for diagnosis. CT imaging also has a sensitivity of 95%–100% in diagnosing and analyzing the site, size, and number of cysts. CT can also be used for monitoring the lesion as well as detection of recurrence. Combining serology with imaging can successfully diagnose more than 90% of cases.[2],[3] Due to the risk of spontaneous rupture, these patients are managed surgically in which splenectomy (total or partial), cyst enucleation, partial cystectomy, unroofing with omentoplasty, and external drainage are the preferred modalities. The study by Ran et al. published in the International Journal of Infectious Diseases in 2014 justified spleen saving surgery over splenectomy as it is safe and preserves immune-related functions of the spleen.[7] Our patient underwent partial cystectomy as the definitive therapy as the cyst was superficial in location, recurrence rate is similar to more radical procedures like splenectomy, and it preserves immune function specifically in childhood by preserving spleen. During the surgery, special precautions were taken and hemodialysis was done 1 day prior to surgery to avoid hyperkalemia and fluid overload along with atracurium was used as an induction agent; the metabolism of the same is renal independent. Postoperatively, he was put on albendazole for 3 months to prevent recurrence.

Hydatid cyst in a known case of CKD has been rarely reported, and hence, we report this unique case. Renal hydatid cyst, though rare, can be seen in 1%–5% hydatid cysts.[8] The case report by Yadav et al. in 2016 has described the case of hydatiduria due to a communicating renal hydatid cyst managed with renal sparing approach including cystopericystectomy, closure of pelvicalyceal system, and double-J stenting in CKD Stage III.[9] Case reports of hydatid cysts with secondary glomerular involvement in the form of mesangioproliferative glomerulonephritis, membranous glomerulopathy, and minimal change disease have had been well-documented.[10] To the best of our knowledge, splenic hydatid cyst as a cause of acute abdomen in ESRD patients has not been reported earlier. The reason of its rarity in ESRD is not postulated, though one of the reasons could be acidic milieu of body fluids seen in ESRD patients.


  Conclusion Top


The hydatid cyst as a cause of pain abdomen is rare, and it is rarely documented in ESRD patients. The splenic hydatid cyst is even rarer in endemic areas in general population and has not been reported as a cause of acute abdomen in ESRD patients which was incidentally diagnosed in our patient and was appropriately treated.

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 his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Osorio MS, Giraldo GC. Gastrointestinal manifestations of chronic kidney disease. Rev Colomb Nefrol 2017;4:3-12.  Back to cited text no. 1
    
2.
Rasheed K, Zargar SA, Telwani AA. Hydatid cyst of spleen: A diagnostic challenge. N Am J Med Sci 2013;5:10-20.  Back to cited text no. 2
    
3.
Gautam S, Patil PL, Sharma R, Darbari A. Simultaneous multiple organ involvement with hydatid cyst: Left lung, liver and pelvic cavity. BMJ Case Rep 2021;14:e241094.  Back to cited text no. 3
    
4.
Pukar MM, Pukar SM. Giant solitary hydatid cyst of spleen – A case report. Int J Surg Case Rep 2013;4:435-7.  Back to cited text no. 4
    
5.
Eckert J, Deplazes P. Biological, epidemiological, and clinical aspects of echinococcosis, a zoonosis of increasing concern. Clin Microbiol Rev 2004;17:107-35.  Back to cited text no. 5
    
6.
Karabicak I, Yurtseven I, Yuruker SS, Ozen N, Kesim M. Splenic hydatid cyst. Can J Surg 2009;52:E209-10.  Back to cited text no. 6
    
7.
Ran B, Shao Y, Yimiti Y, Aji T, Shayiding P, Jiang T, et al. Spleen-preserving surgery is effective for the treatment of spleen cystic echinococcosis. Int J Infect Dis 2014;29:181-3.  Back to cited text no. 7
    
8.
Rexiati M, Mutalifu A, Azhati B, Wang W, Yang H, Sheyhedin I, et al. Diagnosis and surgical treatment of renal hydatid disease: A retrospective analysis of 30 cases. PLoS One 2014;9:e96602.  Back to cited text no. 8
    
9.
Yadav P, Alpana, Sharma V, Srivastava D, Hiralal. Communicating (Open) renal hydatid cyst managed successfully with renal sparing approach. J Clin Diagn Res 2016;10:D16-7.  Back to cited text no. 9
    
10.
Aziz F, Pandya T, Patel HV, Ramakrishna P, Goplani KR, Gumber M, et al. Nephrotic presentation in hydatid cyst disease with predominant tubulointerstital disease. Int J Nephrol Renovasc Dis 2009;2:23-6.  Back to cited text no. 10
    

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Correspondence Address:
Ramanjit Singh Akal,
Department of Nephrology, Command Hospital Air Force Agram Post, Bengaluru - 560 007, Karnataka
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ajim.ajim_28_22



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