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Partially thrombosed aneurysm of the abdominal aorta: Unusual cause of chronic inflammation and resistance to recombinant human erythropoietin.
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MedLine Citation:
PMID:  24574630     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Diagnosis of chronic inflammatory syndrome is often a challenge. In dialysis patients, this condition leads to resistance to recombinant human erythropoietin (rHuEPO). We here report a case of a 72-year-old male undergoing chronic hemodialysis and developed rHuEPO resistance without any obvious etiology. Investigations showed a partially thromosed aneurysm of the infrarenal aorta. Antiplatelet therapy was started with a satisfactory outcome.
Authors:
M El Amrani; A El Kharras; M Asserraji
Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Indian journal of nephrology     Volume:  24     ISSN:  0971-4065     ISO Abbreviation:  Indian J Nephrol     Publication Date:  2014 Jan 
Date Detail:
Created Date:  2014-02-27     Completed Date:  2014-02-27     Revised Date:  2014-03-03    
Medline Journal Info:
Nlm Unique ID:  8914356     Medline TA:  Indian J Nephrol     Country:  India    
Other Details:
Languages:  eng     Pagination:  38-40     Citation Subset:  -    
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Journal Information
Journal ID (nlm-ta): Indian J Nephrol
Journal ID (iso-abbrev): Indian J Nephrol
Journal ID (publisher-id): IJN
ISSN: 0971-4065
ISSN: 1998-3662
Publisher: Medknow Publications & Media Pvt Ltd, India
Article Information
Copyright: © Indian Journal of Nephrology
open-access:
Print publication date: Season: Jan-Feb Year: 2014
Volume: 24 Issue: 1
First Page: 38 Last Page: 40
PubMed Id: 24574630
ID: 3927190
Publisher Id: IJN-24-38
DOI: 10.4103/0971-4065.125069

Partially thrombosed aneurysm of the abdominal aorta: Unusual cause of chronic inflammation and resistance to recombinant human erythropoietin
M. El Amraniaff1
A. El Kharras1
M. Asserrajiaff1
Department of Nephrology and Dialysis, First Medical and Surgical Center, Agadir, Morocco
1Department of Radiology, First Medical and Surgical Center, Agadir, Morocco
Correspondence: Address for correspondence: Dr. El Amrani Mohamed, Unit of Dialysis, Military Teaching Hospital, Rabat, Morocco. E-mail: meders@hotmail.com

Introduction

Diagnosis of chronic inflammatory syndrome (CIS) is often a challenge in patients with chronic kidney disease. This condition also increases the risk of resistance to recombinant human erythropoietin (rHuEPO). We here report an uncommon etiology of CIS in hemodialysis and discuss its particularities.


Case Report

A 72-year-old male with nephroangiosclerosis was on maintenance hemodialysis (three sessions per week) for 10 years. He was a chronic smoker and had dyslipidemia treated with diet alone. His medications included: furosemide 500 mg in non-dialysis day, oral folic acid and vitamin B12, and monthly 75-150 μg of methoxy-polyethylene-glycol epoetin-beta. Biological monitoring revealed episodic elevations of C-reactive protein (CRP) ranging from 1 mg/l to 60 mg/l associated with a decrease of hemoglobin (Hb) levels without any other obvious cause of resistance to rHuEPO [Figure 1]. Serum ferritin, intact parathyroid hormone and aluminum levels were within the normal ranges. The mean Kt/v was 1.15. The patient had no malnutrition and his native fistula showed no malfunction.

Physical examination found no evidence for any local or systemic cause of this inflammation. His vital signs were: Temperature 37°C, pulse 74 beats/min, blood pressure 128/68 mmHg and respiratory rate 20 breaths/min. Biological tests including liver parameters and serum protein electrophoresis were normal. Serology for hepatitis B virus showed negative hepatitis B surface (Hbs) antigen, positive anti-Hbs, and anti-hepatitis B core antibodies with no current biological or morphological signs of chronic hepatitis. Serology for syphilis, hepatitis C, and HIV viruses were negative. Chest radiography and radiography of the sinuses were normal. Sputum examination for acid-fast bacilli and tuberculin skin test was negative. The ear-nose-throat and stomatological examination were normal. Tumor markers were normal. Main laboratory tests are summarized in Table 1.

Transthoracic echocardiogram, blood culture, abdominal and pelvic ultrasound, eso-gastric endoscopy, and colonoscopy showed no abnormalities. As part of a screening study of vascular calcifications in hemodialysis, our patient underwent a lateral abdominal X-ray, which demonstrated diffuse calcifications of the abdominal aorta and a large aneurysm extending from the second to the fourth lumbar vertebra [Figure 2]. Multislice spiral computed tomography-angiography with 3D-reconstruction showed a saccular dilatation of the infrarenal segment of the abdominal aorta measuring 70 mm in height, 41 mm of anteroposterior diameter and 40 mm in transverse diameter with diffuse calcification of both anterior and posterior wall of the aorta extending to the primitive iliac arteries [Figure 3]. There was no evidence of dissection or rupture. Cross sections showed a partial thrombosis of the aorta wall [Figure 4]. The diagnosis of CIS complicating a partially thrombosed aneurysm of the abdominal aorta was then made. Antiplatelet therapy by lysine acetylsalicylate 160 mg/day was introduced to prevent embolic complications. Follow-up over last 6 months showed regression of inflammation, improvement of Hb level, and reduction of rHuEPO doses [Figure 1].


Discussion

Anemia is a common complication of chronic renal failure (CRF). Most patients with CRF achieve the desired target Hb level when supplemented with rHuEPO and parenteral iron.[1] About a quarter of the dialysis patients; however, have a poor response and need higher doses to reach the target Hb level.[2] Iron deficiency, underdialysis, infection and inflammatory conditions may all play a significant role in causing a poor response to rHuEPO therapy. Less common causes of resistance to rHuEPO include blood loss, hyperparathyroidism, aluminum toxicity, vitamin B12 or folic acid deficiency, hemolysis, marrow dysfunction, hemoglobinopathies, concomitant angiotensin converting enzyme inhibitor therapy, carnitine deficiency, and antibodies against the erythropoietin molecule.[3, 4]

There is a well-demonstrated relationship between resistance to rHuEPO therapy and the inflammatory response.[5, 6, 7] Activation of the immune system during the inflammatory process diverts iron traffic from erythropoiesis to storage sites within the reticuloendothelial system, inhibits erythroid progenitor proliferation and differentiation, suppresses erythropoietin production, and blunts response to erythropoietin.[8] In clinical practice, measurement of CRP levels is widely used to monitor inflammation.[9] Our patient showed CIS without any evident etiology. Fortuitously, the diagnosis of partially thrombosed aneurysm of the abdominal aorta was made during a screening study and provided an explanation to this CIS.

Long duration on hemodialysis, advanced age, hypertension, dyslipidemia, and smoking are significant risk factors in our patient for atherosclerosis and vascular calcifications as reported in hemodialysis population.[10] The infrarenal abdominal aorta is prone to the development of atherosclerotic plaque and its complications including obstruction, ulceration, thrombus formation, and potentially, aneurysmal degeneration.[11] The spontaneous thrombosis of aortic aneurysm is rare.[12] In our patient, extensive atherosclerotic and calcified plaques may explain the thrombosis rather than the aneurysm itself.

Thrombosis and inflammation are intertwined in vascular pathology. Inflammation can beget local thrombosis, and thrombosis can amplify inflammation.[13] Indeed, the smooth muscle cell in the artery wall can undergo inflammatory activation when exposed to thrombin and products of thrombosis. For example, thrombin stimulation causes smooth muscle cells to produce interleukin (IL)-6 abundantly.[14] Platelet-derived growth factor, released from platelet alpha granules during thrombosis, can also markedly augment IL-6 production by smooth muscle cells.[15] IL-6, in turn, can induce the cute inflammatory phase response and increase plasma concentrations of fibrinogen, and the inflammatory marker CRP as in our patient. Thereby, antithrombotic treatment may suppress inflammation and help break the vicious cycle by limiting the local and systemic amplification loops between inflammation and thrombosis.[13]

Accelerated atherosclerosis and vascular calcifications are frequent, but ominous, complications in hemodialysis patients. The development of an aneurysm of the abdominal aorta is highly favored in this particularly exposed population. Functional signs are rare and often confused with digestive complaints. A CIS may accompany thrombotic complications and lead to resistance to rHuEPO. Our case alerts physicians on this rare etiology of CIS and highlights the importance of screening for vascular calcification in the early stages of kidney disease and during the hemodialysis follow-up.


Notes

Source of Support: Nil

Conflict of Interest: None declared.

References
1. European best practice guidelines for the management of anemia in patients with chronic renal failureNephrol Dial TransplantYear: 199914Suppl 5150
2. Bárány P. Inflammation, serum C-reactive protein, and erythropoietin resistanceNephrol Dial TransplantYear: 200116224711158392
3. Macdougall IC. Poor response to erythropoietin: Practical guidelines on investigation and managementNephrol Dial TransplantYear: 199510607147566570
4. Fishbane S. Hyporesponsiveness to recombinant human erythropoietin in dialysis patientsDial TransplantYear: 2000295458
5. Hörl WH,Jacobs C,Macdougall IC,Valderrábano F,Parrondo I,Thompson K,et al. European best practice guidelines 14-16: Inadequate response to epoetinNephrol Dial TransplantYear: 200015Suppl 4435011052148
6. Bárány P,Divino Filho JC,Bergström J. High C-reactive protein is a strong predictor of resistance to erythropoietin in hemodialysis patientsAm J Kidney DisYear: 19972956589100046
7. Kalantar-Zadeh K,McAllister CJ,Lehn RS,Lee GH,Nissenson AR,Kopple JD. Effect of malnutrition-inflammation complex syndrome on EPO hyporesponsiveness in maintenance hemodialysis patientsAm J Kidney DisYear: 2003427617314520627
8. Bratosin D,Mazurier J,Tissier JP,Estaquier J,Huart JJ,Ameisen JC,et al. Cellular and molecular mechanisms of senescent erythrocyte phagocytosis by macrophages. A reviewBiochimieYear: 199880173959587675
9. Smrzova J,Balla J,Bárány P. Inflammation and resistance to erythropoiesis-stimulating agents – What do we know and what needs to be clarified?Nephrol Dial TransplantYear: 200520Suppl 8827
10. Balla S,Nusair MB,Alpert MA. Risk factors for atherosclerosis in patients with chronic kidney disease: Recognition and managementCurr Opin PharmacolYear: 2013 Epub ahead of print, in press.
11. Glagov S. Wissler RW,Geer JCHemodynamic risk factors: Mechanical stress, mural architecture, medial nutrition and the vulnerability of arteries to atherosclerosisThe Pathogenesis of AtherosclerosisYear: 1972BaltimoreWilliams and Wilkins164
12. Dalal S,Dohlon M,Beard JD. Thrombosed abdominal aortic aneurysms. Do they need surveillance to prevent late rupture?Eur J Vasc Endovasc SurgYear: 200122570211735212
13. Libby P,Simon DI. Inflammation and thrombosis: The clot thickensCirculationYear: 200110317182011282900
14. Kranzhöfer R,Clinton SK,Ishii K,Coughlin SR,Fenton JW 2nd,Libby P. Thrombin potently induces cytokine production by human vascular smooth muscle cells but not in mononuclear phagocytesCirc ResYear: 199679286948756006
15. Loppnow H,Libby P. Proliferating or interleukin 1-activated human vascular smooth muscle cells secrete copious interleukin 6J Clin InvestYear: 19908573182312724

Figures

[Figure ID: F1]
Figure 1 

History of inflammation, hemoglobin, and recombinant human erythropoietin doses



[Figure ID: F2]
Figure 2 

Lateral abdominal X-ray: Aneurysm of the abdominal aorta



[Figure ID: F3]
Figure 3 

3D reconstruction of multislice computed tomography angiography: Extensive calcification of the abdominal aorta extending to the primitive iliac arteries



[Figure ID: F4]
Figure 4 

Contrast enhanced multislice computed tomography angiography cross section: Partial thrombosis of the abdominal aorta wall



Tables
[TableWrap ID: T1] Table 1 

Laboratory tests




Article Categories:
  • Case Report

Keywords: Aneurysm, atherosclerosis, chronic inflammatory syndrome, resistance to recombinant human erythropoietin, thrombosis.

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