Is Nutcracker Syndrome Innocent?

Öz

Objective: Nutcracker syndrome (NS) is defined by the compression of the left renal vein by the superior mesenteric artery. The incidence of NS and the exact treatment protocols are not well known. The objective of this study is to analyze children with NS and evaluate the prognosis in follow-up. Methods: All patients were referred to the pediatric nephrology clinic by pediatricians because of proteinuria. The study includes 74 patients (52 girls, 22 boys) with a mean age of 13.15±2.32 and records the presenting symptoms, degree of proteinuria, and radiologic findings. The diameter ratios of the left renal vein before and after stenosis and peak systolic velocity (PSV) ratios were calculated with a Doppler ultrasound, with a peak systolic ratio of 3.0:1 or greater was accepted as the diagnostic criterion for NS. Results: The mean age was 13.15±2.32 (5.33-17.42) years, while the mean follow-up duration was 14.6±9.1 months. Proteinuria was not correlated with the degree of compression. Most patients had a good prognosis, and none of them required medical or surgical treatment. A significant reduction or complete normalization of proteinuria was seen in more than half of the patients under conservative management. Conclusions: Based on our findings, NS seems to have a good prognosis in children who present with isolated proteinuria.

Anahtar Kelimeler

• Proteinuria
• nutcracker syndrome
• renal Doppler ultrasound

Kaynakça

1. Wolfish NM, McLaine PN, Martin D . Renal vein entrapment syndrome: frequency and diagnosis. A lesson in conservatism. Clin Nephrol 1986;26(2):96-100. google scholar
2. Pardinhas C, do Carmo C, Gomes C, Escada L, Alves R . Nutcracker Syndrome: A Single-Center Experience. Case Rep Nephrol Dial 2021;11(1):48-54. doi:10.1159/000512589. google scholar
3. K.W. Kim, J.Y. Cho, S.H. Kim, et al. Diagnostic value of computed tomographic findings of nutcracker syndrome: correlation with renal venography and renocaval pressure grandients, Eur. J. Radiol. 2010;80(3):648-54. doi: 10.1016/j.ejrad.2010.08.044. google scholar
4. Avgerinos ED, McEnaney R, Chaer RA. Surgical and endovascular interventions for nutcracker syndrome. Semin Vasc Surg 2013;26(4):170-7. doi: 10.1053/j.semvascsurg.2014.06.014. Epub 2014 Jun 25. google scholar
5. Zhang H, Li M, Jin W.et al. The left renal entrapment syndrome: diagnosis and treatment. Ann Vasc Surg 2007;21(2):198-203. doi: 10.1016/j.avsg.2006.10.021. google scholar
6. Shin JI, Lee JS, Kim MJ . The prevalence, physical characteristics and diagnosis of nutcracker syndrome. Eur J Vasc Endovasc Surg 2006;32(3):335-6. doi: 10.1016/j.ejvs.2006.04.030 google scholar
7. Granata A, Distefano G, Sturiale A, Figuera M, Foti PV, Palmucci S, et al. From Nutcracker Phenomenon to Nutcracker Syndrome: A Pictorial Review. Diagnostics (Basel) 2021;11(1):101. doi: 10.3390/ diagnostics11010101. google scholar
8. Bhanji A, Malcolm P, Karim M. Nutcracker syndrome and radiographic evaluation of loin pain and hematuria. Am J Kidney Dis 2010;55(6):1142-5. doi: 10.1053/j.ajkd.2009.10.010. google scholar

9. He Y, Wu Z, Chen S, Tian L, Li D, Li M, et al. Nutcracker syndrome--how well do we know it? Urology 2014;83(1):12-7. doi: 10.1016/j. urology.2013.08.033. google scholar
10. Vianello FA, Mazzoni MB, Peeters GG, Fossali EF, Camozzi P, Bianchetti MG, et al. Micro- and macroscopic hematuria caused by renal vein entrapment: Systematic review of the literature. Pediatr. Nephrol 2016; 31(2):175-184. doi: 10.1007/s00467-015-3045-2. google scholar
11. Okada M, Tsuzuki K, Ito S. Diagnosis of the nutcracker phenomenon using two-dimensional ultrasonography. Clin Nephrol 1998;49(1)35-40. google scholar
12. Alaygut D, Bayram M, Soylu A.et al. The clinical course of children with Nutcracker syndrome. Urology 2013;82(3):686-90. doi: 10.1016/j.urology.2013.03.048. google scholar
13. Agarwal A, Litra F, Barr LL. A Rare Cause of Abdominal and Flank Pain in Children: Nutcracker Syndrome. Cureus 2021;16;13(7) e16422. doi: 10.7759/cureus.16422. google scholar
14. Nutcracker Waldrop RD, Henning P . Nutcracker Syndrome Masquerading as Renal Colic in an Adolescent Athlete: A Case Report. Clin. Pract. Cases Emerg Med 2021;5:415-8. doi: 10.5811/ cpcem.2021.6.52140. google scholar
15. Gulleroglu K, Gulleroglu B, Baskin E. Nutcracker syndrome. World J. Nephrol 2014;3(4):277-281. doi: 10.5527/wjn.v3.i4.277. google scholar
16. Takebayashi S, Ueki T, Ikeda N, Fujikawa A . Diagnosis of the nutcracker syndrome with color Doppler sonography: correlation with flow patterns on retrograde left renal venography. AJR 1999;172(1):39-43. doi: 10.2214/ajr.172.1.9888735. google scholar
17. Hangge P, Gupta N, Khurana A, et al. Degree of Left Renal Vein Compression Predicts Nutcracker Syndrome. J Clin Med 2018;7(5):107. doi: 10.3390/jcm7050107. google scholar
18. Ananthan K, Onida S, Davies AH. Nutcracker Syndrome: An Update on Current Diagnostic Criteria and Management Guidelines. Eur J Vasc Endovasc Surg 2017;53(6):886-94. doi: 10.1016/j. ejvs.2017.02.015. google scholar
19. Takahashi Y, Sano A, Matsuo M. An ultrasonographic classification for diverse clinical symptoms of pediatric nutcracker phenomenon. Clin Nephrol 2005;64(1):47-54. doi: 10.5414/ cnp64047. google scholar
20. Park SJ, Lim JW, Cho BS, et al. Nutcracker syndrome in children with orthostatic proteinuria: diagnosis on the basis of Doppler sonography. J Ultrasound Med 2002;21(1):39-45; quiz 46. doi:10.7863/jum.2002.21.1.39 google scholar
21. Shin JI, Park JM, Lee JS, et al. Morphologically improved nutcracker syndrome in an 11-year-old girl with hematuria. Pediatr Int 2007; 49(5):677-9. Doi:10.1111/j.1442-200X.2007.02435. google scholar
22. Shin JI, Park JM, Lee JS, Kim MJ. Effect of renal Doppler ultrasound on the detection of nutcracker syndrome in children with hematuria. Eur J Pediatr 2007;166(5):399-404. doi: 10.1007/ s00431-006-0252-7. google scholar
23. Cheon JE, Kim WS, Kim IO, Kim SH, Yeon KM, Ha IS,et al. Nutcracker syndrome in children with gross haematuria: Doppler sonographic evaluation of the left renal vein. Pediatr Radiol 2006;36(7):682-6. doi: 10.1007/s00247-006-0145-y. google scholar
24. Meyer J, Rother U, Stehr M, Meyer A. Nutcracker syndrome in children: Appearance, diagnostics, and treatment - A systematic review. J Pediatr Surg. 2022;57(11):716-722. doi:10.1016/j. jpedsurg.2021.12.019. google scholar
25. Ananthan K, Onida S, Davies AH. Nutcracker Syndrome: An Update on Current Diagnostic Criteria and Management Guidelines. Eur J Vasc Endovasc Surg 2017;53(6):886-94. doi: 10.1016/j. ejvs.2017.02.015. google scholar
26. Kargın Çakıcı E, Yazılıtaş F, Çınar HG. Nutcracker syndrome in children: the role of Doppler ultrasonography in symptomatic patients. Turkish J Pediatr Dis 2019;5: 348-52. google scholar