Skip to main content
Log in

Micro- and macroscopic hematuria caused by renal vein entrapment: systematic review of the literature

  • Review
  • Published:
Pediatric Nephrology Aims and scope Submit manuscript

Abstract

Background

Hematuria secondary to renal vein entrapment is mentioned only passing in textbooks and reviews.

Methods

We performed a search of the National Library of Medicine database for peer-reviewed publications using the terms “renal vein” or “nutcracker” and “hematuria”.

Results

We identified 187 published reports/studies that covered 736 patients, of whom 288 had microscopic hematuria and 448 had macroscopic hematuria. The patient cohort comprised 159 patients aged ≤17 years. Abdominal pain was absent in  approximately 65 % of all patients, and a clinically relevant left-sided varicocele was observed in 29 % of the male patients. A normal pre-aortic left renal vein and an anomalous anatomy were noted in 680 and 56 patients, respectively. The body mass index (BMI) was lower in patients with renal vein entrapment than in the controls, with a regression of hematuria correlating with an increase in BMI. A surgical procedure was attempted in 34 % of the patients, of which the most common were endovascular stenting and transposition of the renal vein distally into the vena cava.

Conclusions

In cases of unexplained hematuria with or without abdominal pain, clinicians should consider the diagnosis of renal vein congestion, especially in males with varicocele. Ultrasonic Doppler flow scanning is the recommended initial diagnostic modality in these patients. Expectation management is advised in the great majority of cases.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2

Similar content being viewed by others

References

  1. Noorani A, Walsh SR, Cooper DG, Varty K (2009) Entrapment syndromes. Eur J Vasc Endovasc Surg 37:213–220

    Article  PubMed  CAS  Google Scholar 

  2. De Schepper A (1972) “Nutcracker”-fenomeen van de vena renalis en venueze pathologie van de linker nier. J Belg Radiol 55:507–511

    PubMed  Google Scholar 

  3. Moher D, Liberati A, Tetzlaff J, Altman DG, PRISMA Group (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. Ann Intern Med 151:264–269

    Article  PubMed  Google Scholar 

  4. Alaygut D, Bayram M, Soylu A, Cakmakcı H, Türkmen M, Kavukcu S (2013) Clinical course of children with nutcracker syndrome. Urology 82:686–690

    Article  PubMed  Google Scholar 

  5. Ali-El-Dein B, Osman Y, Shehab El-Din AB, El-Diasty T, Mansour O, Ghoneim MA (2003) Anterior and posterior nutcracker syndrome: a report on 11 cases. Transplant Proc 35:851–853

    Article  PubMed  CAS  Google Scholar 

  6. Allam SR, Livingston TS, Kalaria V, Diaz-Calderon W, Pumphrey JA, Patel L, Sankarapandian B (2014) Posterior nutcracker syndrome: an infrequent cause of hematuria. Kidney Int 85:985–986

    Article  PubMed  Google Scholar 

  7. Alper F, Tasar M, Yildiz H, Tasar A, Saglam M (2005) Unexplained hematuria in association with a rare renal vein anomaly: MDCT and MRA demonstration. Cardiovasc Intervent Radiol 28:847–849

    Article  PubMed  Google Scholar 

  8. Altugan FS, Ekim M, Fitöz S, Ozçakar ZB, Burgu B, Yalçınkaya F, Soygür T (2010) Nutcracker syndrome with urolithiasis. J Pediatr Urol 6:519–521

    Article  PubMed  Google Scholar 

  9. Andrianne R, Limet R, Waltregny D, de Leval J (2002) Hématurie causée par un “nutcracker syndrome” ou “syndrome du casse-noisettes”: confirmation peropératoire de sa réalité. Prog Urol 12:1323–1326

    PubMed  Google Scholar 

  10. Arima M, Hosokawa S, Ogino T, Ihara H, Terakawa T, Ikoma F (1990) Ultrasonographically demonstrated nutcracker phenomenon: alternative to angiography. Int Urol Nephrol 22:3–6

    Article  PubMed  CAS  Google Scholar 

  11. Ariyoshi A, Nagase K (1990) Renal hematuria caused by "nutcracker" phenomenon: a more logical surgical management. Urology 35:168–170

    Article  PubMed  CAS  Google Scholar 

  12. Baldi S, Rabellino M, Zander T, González G, Maynar M (2011) Endovascular treatment of the nutcracker syndrome: report of two cases. Minim Invasive Ther Allied Technol 20:356–359

    Article  PubMed  Google Scholar 

  13. Balduyck B, Van Den Brande F, Rutsaert R (2014) Abdominal aortic aneurysm rupture into a retro-aortic left renal vein. Acta Chir Belg 114:136–138

    PubMed  CAS  Google Scholar 

  14. Baril DT, Polanco P, Makaroun MS, Chaer RA (2011) Endovascular management of recurrent stenosis following left renal vein transposition for the treatment of Nutcracker syndrome. J Vasc Surg 53:1100–1103

    Article  PubMed  Google Scholar 

  15. Barnes RW, Fleisher HL 3rd, Redman JF, Smith JW, Harshfield DL, Ferris EJ (1988) Mesoaortic compression of the left renal vein (the so-called nutcracker syndrome): repair by a new stenting procedure. J Vasc Surg 8:415–421

    Article  PubMed  CAS  Google Scholar 

  16. Basile A, Tsetis D, Calcara G, Figuera M, Coppolino F, Patti MT, Midiri M, Granata A (2007) Nutcracker syndrome due to left renal vein compression by an aberrant right renal artery. Am J Kidney Dis 50:326–329

    Article  PubMed  Google Scholar 

  17. Basile A, Tsetis D, Calcara G, Figuera M, Patti MT, Ettorre GC, Granata A (2007) Percutaneous nitinol stent implantation in the treatment of nutcracker syndrome in young adults. J Vasc Interv Radiol 18:1042–1046

    Article  PubMed  Google Scholar 

  18. Bates CM, Adler BM, Schwaderer A, Coley BD (2007) Clinical quiz. A patient with recurrent episodes of red urine. Pediatr Nephrol 22:187–191

    Article  PubMed  Google Scholar 

  19. Ben Abdallah Chabchoub R, Chabchoub K, Maaloul I, Gargouri L, Ben Mahfoudh K, Nabil Mhiri M, Mhiri R, Mahfoudh A (2011) Le syndrome casse-noisette (Nutcracker): une cause rare d’hématurie. Arch Pediatr 18:1188–1190

    Article  PubMed  CAS  Google Scholar 

  20. Bhanji A, Malcolm P, Karim M (2010) Nutcracker syndrome and radiographic evaluation of loin pain and hematuria. Am J Kidney Dis 55:1142–1145

    Article  PubMed  Google Scholar 

  21. Bhimma R, Robbs J (2005) Nutcracker. Lancet 365:1280

    Article  PubMed  Google Scholar 

  22. Breitwieser C, Asbach P, Sachs M, Rogalla P, Rodenwaldt J (2005) Posterior "nutcracker" phenomenon: hemodynamic relevant aorto-retroaortal renal vein fistula leading to fatal right heart failure. Acta Radiol 46:193–195

    Article  PubMed  CAS  Google Scholar 

  23. Calado R, Braz M, Lobo L, Simão C (2011) Nutcracker syndrome. Síndrome de Nutcracker. Hematúria sem diagnóstico? Acta Med Port 24[Suppl 3]:695–698

    PubMed  Google Scholar 

  24. Ceriani E, Carrabba M, Lemos A, Fabrizi F, Messa P (2006) An unusual cause of gross hematuria, improved by recumbency. Kidney Int 70:978

    Article  PubMed  CAS  Google Scholar 

  25. Chang CT, Hung CC, Ng KK, Yen TH (2005) Nutcracker syndrome and left unilateral haematuria. Nephrol Dial Transplant 20:460–461

    Article  PubMed  Google Scholar 

  26. Chen HH, You ZH, Chuang SH, Wu TH (2011) Nutcracker syndrome. Int Med J 41:503–504

    Article  CAS  Google Scholar 

  27. Chen S, Zhang H, Shi H, Tian L, Jin W, Li M (2011) Endovascular stenting for treatment of Nutcracker syndrome: report of 61 cases with long-term followup. J Urol 186:570–575

    Article  PubMed  Google Scholar 

  28. Chen S, Zhang H, Tian L, Li M, Zhou M, Wang Z (2009) A stranger in the heart: LRV stent migration. Int Urol Nephrol 41:427–430

    Article  PubMed  Google Scholar 

  29. Chen S, Zhang H, Tian L, Li M (2012) Endovascular management of nutcracker syndrome after migration of a laparoscopically placed extravascular stent. Am J Kidney Dis 60:322–326

    Article  PubMed  Google Scholar 

  30. Chen W, Chu J, Yang JY, Li HP, Zhuang WQ, Huang YH, Guo WB (2005) Endovascular stent placement for the treatment of nutcracker phenomenon in three pediatric patients. J Vasc Interv Radiol 16:1529–1533

    Article  PubMed  Google Scholar 

  31. Chen YM, Wang IK, Ng KK, Huang CC (2002) Nutcracker syndrome: an overlooked cause of hematuria. Chang Gung Med J 25:700–705

    PubMed  Google Scholar 

  32. Cheon JE, Kim WS, Kim IO, Kim SH, Yeon KM, Ha IS, Cheong HI, Choi Y (2006) Nutcracker syndrome in children with gross haematuria: doppler sonographic evaluation of the left renal vein. Pediatr Radiol 36:682–686

    Article  PubMed  Google Scholar 

  33. Chi YW, Ehrensing C (2007) Images in vascular medicine. Nutcracker syndrome. Vasc Med 12:251–253

    Article  PubMed  Google Scholar 

  34. Chiesa R, Anzuini A, Marone EM, Briguori C, Moura MR, Melissano G, Colombo A, Rosanio S (2001) Endovascular stenting for the nutcracker phenomenon. J Endovasc Ther 8:652–655

    PubMed  CAS  Google Scholar 

  35. Choi SH, Kim JS, Shin TS, Lee YS, Kim HJ, Choi NG (2009) Nutcracker syndrome diagnosed with 3-dimensional computed tomography angiography. Korean J Urol 50:711–713

    Article  Google Scholar 

  36. Chuang CK, Chu SH, Lai PC (1997) The nutcracker syndrome managed by autotransplantation. J Urol 157:1833–1834

    Article  PubMed  CAS  Google Scholar 

  37. Chung BI, Gill IS (2009) Laparoscopic splenorenal venous bypass for nutcracker syndrome. J Vasc Surg 49:1319–1323

    Article  PubMed  Google Scholar 

  38. Cohen F, Amabile P, Varoquaux A, Piquet P, Bartoli JM, Vidal V, Jacquier A (2009) Endovascular treatment of circumaortic nutcracker syndrome. J Vasc Interv Radiol 20:1255–1257

    Article  PubMed  Google Scholar 

  39. Daily R, Matteo J, Loper T, Northup M (2012) Nutcracker syndrome: symptoms of syncope and hypotension improved following endovascular stenting. Vascular 20:337–341

    Article  PubMed  Google Scholar 

  40. Del Canto Peruyera P, Vaquero Lorenzo F, Vallina-Victorero Vazquez MJ, Alvarez Salgado A, Vicente Santiago M, Botas Velasco M, Alvarez Fernandez LJ (2014) Recurrent hematuria caused by nutcracker syndrome. Ann Vasc Surg 28:1036, e15-1036.e19

    PubMed  Google Scholar 

  41. Dever DP, Ginsburg ME, Millet DJ, Feinstein MJ, Cockett AT (1986) Nutcracker phenomenon. Urology 27:540–542

    Article  PubMed  CAS  Google Scholar 

  42. Di Palma AM, Netti GS, Papagno F, Antonica G, Ettorre GC, Prisco T, Gesualdo L (2004) Sindrome di nutcracker: una macroematuria intermittente di difficile diagnosi. G Ital Nefrol 21:288–292

    PubMed  Google Scholar 

  43. Dong W, Yao Y, Huang H, Han J, Zhao X, Huang J (2014) Surgical management of nutcracker phenomenon presenting as left varicocele in adolescents: a novel approach. J Pediatr Urol 10:424–429

    Article  PubMed  Google Scholar 

  44. Ekwenna O, Gorin MA, Castellan M, Casillas V, Ciancio G (2011) Inverted nutcracker syndrome: a case of persistent hematuria and pain in the presence of a left-sided inferior vena cava. Sci World J 11:1031–1035

    Article  Google Scholar 

  45. El Harrech Y, Jira H, Chafiki J, Ghadouane M, Ameur A, Abbar M (2009) Actitud expectante en el Síndrome del Cascanueces. Actas Urol Esp 33:93–96

    Article  PubMed  Google Scholar 

  46. Esquena Fernández S, Abascal Junquera JM, Trilla Herrera E, Martos Calvo R, Ramírez Sevilla C, Morote Robles J (2005) Síndrome del cascanueces como causa de hematuria. Actas Urol Esp 29:805

    Article  PubMed  Google Scholar 

  47. Fitoz S, Yalcinkaya F (2008) Compression of left inferior vena cava: a form of nutcracker syndrome. J Clin Ultrasound 36:101–104

    Article  PubMed  Google Scholar 

  48. Franco A, Matías J, Colom S, Muñoz J, López-Costea MA, Contreras J, Serrallach N (1994) Fenomeno nutcracker o cascanueces: una infrecuente causa de hematuria. Actas Urol Esp 18:826–828

    PubMed  CAS  Google Scholar 

  49. Fu WJ, Hong BF, Gao JP, Xiao YY, Yang Y, Cai W, Guo G, Wang XX (2006) Nutcracker phenomenon: a new diagnostic method of multislice computed tomography angiography. Int J Urol 13:870–873

    Article  PubMed  Google Scholar 

  50. Fu WJ, Hong BF, Xiao YY, Yang Y, Cai W, Gao JP, Guo G, Wang XX (2004) Diagnosis of the nutcracker phenomenon by multislice helical computed tomography angiography. Chin Med J (Engl) 117:1873–1875

    Google Scholar 

  51. Fujita K, Munakata A (1991) Left renal haematuria by compression of an unusual vein. Int Urol Nephrol 23:303–306

    Article  PubMed  CAS  Google Scholar 

  52. Gagnon LO, Ponsot Y, Benko A, le Tu M (2009) Nutcracker syndrome in a 20-year-old patient treated with intravascular stent placement: a case report. Can J Urol 16:4765–4769

    PubMed  Google Scholar 

  53. Genc G, Ozkaya O, Bek K, Acikgoz Y, Danaci M (2010) A rare cause of recurrent hematuria in children: nutcracker syndrome. J Trop Pediatr 56:275–277

    Article  PubMed  Google Scholar 

  54. Gibo M, Onitsuka H (1998) Retroaortic left renal vein with renal vein hypertension causing hematuria. Clin Imaging 22:422–424

    Article  PubMed  CAS  Google Scholar 

  55. Gorospe EC, Aigbe MO (2006) Nutcracker syndrome: a rare cause of hematuria. Sci World J 6:745–746

    Article  Google Scholar 

  56. Granata A, Figuera M, Basile A (2007) Nutcracker syndrome. Am J Kidney Dis 50:A43–A44

    PubMed  Google Scholar 

  57. Guerrero-Sánchez V, Hernández-Hernández JL, García-Montero A (2012) Síndrome del cascanueces - Caso clinic y revisión de la literature. Rev Med Inst Mex Seguro Soc 50:545–548

    PubMed  Google Scholar 

  58. Guo P, Belostotsky V (2013) Cracking haematuria in adolescent with negative work up. BMJ Case Rep 2013. doi:10.1136/bcr-2013-009924

  59. Hanna HE, Santella RN, Zawada ET Jr, Masterson TE (1997) Nutcracker syndrome: an underdiagnosed cause for hematuria? S D J Med 50:429–436

    PubMed  CAS  Google Scholar 

  60. Hartung O, Azghari A, Barthelemy P, Boufi M, Alimi YS (2010) Laparoscopic transposition of the left renal vein into the inferior vena cava for nutcracker syndrome. J Vasc Surg 52:738–741

    Article  PubMed  Google Scholar 

  61. Hartung O, Barthelemy P, Berdah SV, Alimi YS (2009) Laparoscopy-assisted left ovarian vein transposition to treat one case of posterior nutcracker syndrome. Ann Vasc Surg 23:413, e13-413.e16

    Article  PubMed  Google Scholar 

  62. Hartung O, Grisoli D, Boufi M, Marani I, Hakam Z, Barthelemy P, Alimi YS (2005) Endovascular stenting in the treatment of pelvic vein congestion caused by nutcracker syndrome: lessons learned from the first five cases. J Vasc Surg 42:275–280

    Article  PubMed  Google Scholar 

  63. Hendriks JJ, Schröder CH, Strijk SP, de Vries JD, Monnens LA (1991) Het syndroom van de ingeklemde linker niervene (notenkrakersyndroom). Tijdschr Kindergeneeskd 59:95–97

    PubMed  CAS  Google Scholar 

  64. Hohenfellner M, D'Elia G, Hampel C, Dahms S, Thüroff JW (2002) Transposition of the left renal vein for treatment of the nutcracker phenomenon: long-term follow-up. Urology 59:354–357

    Article  PubMed  Google Scholar 

  65. Hohenfellner M, Steinbach F, Schultz-Lampel D, Schantzen W, Walter K, Cramer BM, Thüroff JW, Hohenfellner R (1991) The nutcracker syndrome: new aspects of pathophysiology, diagnosis and treatment. J Urol 146:685–688

    PubMed  CAS  Google Scholar 

  66. Igari H (2001) Diagnosis of nutcracker syndrome of the left renal vein: value of the corticomedullary phase of helical CT. Nihon Igaku Hoshasen Gakkai Zasshi 61:292–297

    PubMed  CAS  Google Scholar 

  67. Inal M, Karadeniz Bilgili MY, Sahin S (2014) Nutcracker syndrome accompanying pelvic congestion syndrome; color Doppler sonography and multislice CT findings: a case report. Iran J Radiol 11:e11075

    PubMed  PubMed Central  Google Scholar 

  68. Ishidoya S, Chiba Y, Sakai K, Orikasa S (1994) Nutcracker phenomenon: a case with surgical treatment and its diagnostic criteria. Acta Urol Jpn 40:135–138

    CAS  Google Scholar 

  69. Itoh S, Yoshida K, Nakamura Y, Mitsuhashi N (1997) Aggravation of the nutcracker syndrome during pregnancy. Obstet Gynecol 90:661–663

    Article  PubMed  CAS  Google Scholar 

  70. Jang YB, Kang KP, Lee S, Kim W, Kwak HS, Park SK (2005) Posterior nutcracker phenomenon. Nephrol Dial Transplant 20:2573–2574

    Article  PubMed  Google Scholar 

  71. Jodorkovsky R, Milman E (2012) A child with recurrent gross hematuria caused by the nutcracker syndrome: lessons learned. Clin Pediatr (Phila) 51:291–293

    Article  Google Scholar 

  72. Kaneko K, Kiya K, Nishimura K, Shimizu T, Yamashiro Y (2001) Nutcracker phenomenon demonstrated by three-dimensional computed tomography. Pediatr Nephrol 16:745–747

    Article  PubMed  CAS  Google Scholar 

  73. Kaneko K, Ohtomo Y, Yamashiro Y, Obinata K, Kurokawa S, Aizawa S (1999) Magnetic resonance angiography in nutcracker phenomenon. Clin Nephrol 51:259–260

    PubMed  CAS  Google Scholar 

  74. Karaosmanoğlu D, Karcaaltincaba M, Akata D, Ozmen M (2010) Unusual causes of left renal vein compression along its course: MDCT findings in patients with nutcracker and pelvic congestion syndrome. Surg Radiol Anat 32:323–327

    Article  PubMed  Google Scholar 

  75. Kavukcu S, Kasap B, Göktay Y, Seçil M (2004) Doppler sonographic indices in diagnosing the nutcracker phenomenon in a hematuric adolescent. J Clin Ultrasound 32:37–41

    Article  PubMed  Google Scholar 

  76. Kawa G, Kawakita S, Fujita I, Matsuda T (2000) A simple treatment for gross haematuria caused by the 'nutcracker' phenomenon. BJU Int 86:908–909

    Article  PubMed  CAS  Google Scholar 

  77. Kawamoto S, Horton KM, Fishman EK (2008) Nutcracker phenomenon demonstrated by multidetector computed tomography with three-dimensional imaging. Urology 71:228–229

    Article  PubMed  Google Scholar 

  78. Kim JY, Joh JH, Choi HY, Do YS, Shin SW, Kim DI (2006) Transposition of the left renal vein in nutcracker syndrome. Eur J Vasc Endovasc Surg 31:80–82

    Article  PubMed  CAS  Google Scholar 

  79. Kim KW, Cho JY, Kim SH, Yoon JH, Kim DS, Chung JW, Park JH (2011) Diagnostic value of computed tomographic findings of nutcracker syndrome: correlation with renal venography and renocaval pressure gradients. Eur J Radiol 80:648–654

    Article  PubMed  Google Scholar 

  80. Kim SH, Cho SW, Kim HD, Chung JW, Park JH, Han MC (1996) Nutcracker syndrome: diagnosis with Doppler US. Radiology 198:93–97

    Article  PubMed  CAS  Google Scholar 

  81. Kim SJ, Kim CW, Kim S, Lee TH, Lee JW, Lee SH, Jeong YS (2005) Long-term follow-up after endovascular stent placement for treatment of nutcracker syndrome. J Vasc Interv Radiol 16:428–431

    Article  PubMed  Google Scholar 

  82. Kimura K, Araki T (1996) Images in clinical medicine. Nutcracker phenomenon. N Engl J Med 335:171

    Article  PubMed  CAS  Google Scholar 

  83. Kodama K, Noda T, Motoi I (2013) Nutcracker phenomenon: an unusual presentation of acute aortic dissection. Indian J Urol 29:67–69

    Article  PubMed  PubMed Central  Google Scholar 

  84. Koshimichi M, Sugimoto K, Yanagida H, Fujita S, Miyazawa T, Sakata N, Okada M, Takemura T (2012) Newly-identified symptoms of left renal vein entrapment syndrome mimicking orthostatic disturbance. World J Pediatr 8:116–122

    Article  PubMed  Google Scholar 

  85. Kumon H, Tsugawa M, Matsumura Y, Ohmori H (1990) Endoscopic diagnosis and treatment of chronic unilateral hematuria of uncertain etiology. J Urol 143:554–558

    PubMed  CAS  Google Scholar 

  86. Lang EK, Emery K, Macchia RJ (2007) Microscopic hematuria attributed to retroaortic left renal vein. J Urol 178:2164

    Article  PubMed  Google Scholar 

  87. Lang EK, Macchia RJ, Castle E, Earhart V (2006) Nutcracker phenomenon. J Urol 175:1120

    Article  PubMed  Google Scholar 

  88. Lau JL, Lo R, Chan FL, Wong KK (1986) The posterior "nutcracker": hematuria secondary to retroaortic left renal vein. Urology 28:437–439

    Article  PubMed  CAS  Google Scholar 

  89. Lee CC, Lin JT, Deng HH, Lin ST (1993) Hematuria due to nutcracker phenomenon of left renal vein: report of a case. J Formos Med Assoc 92:291–293

    PubMed  CAS  Google Scholar 

  90. Lee JH, Choi HW, Lee YJ, Park YS (2014) Causes and outcomes of asymptomatic gross haematuria in children. Nephrol (Carlton) 19:101–106

    Article  Google Scholar 

  91. Lee S, Kim W, Kang KP, Kwak HS, Park SK (2007) Microscopic hematuria and the posterior nutcracker phenomenon. Kidney Int 72:1037

    Article  PubMed  CAS  Google Scholar 

  92. Li H, Sun X, Liu G, Zhang Y, Chu J, Deng C, Zhou B, Chen W, Yang J (2013) Endovascular stent placement for nutcracker phenomenon. J Xray Sci Technol 21:95–102

    Article  PubMed  CAS  Google Scholar 

  93. Li H, Zhang M, Jiang Y, Zhang Z, Na W (2014) Microsurgical spermatic-inferior epigastric vein anastomosis for treating nutcracker syndrome-associated varicocele in infertile men: a preliminary experience. Urology 83:94–99

    Article  PubMed  Google Scholar 

  94. Li MF, Yang TL (2011) Nutcracker syndrome. Nephrol (Carlton) 16:118

    Article  CAS  Google Scholar 

  95. Li P, Shao P, Qin C, Ju X, Meng X, Li J, Lv Q, Cao Q, Yin C (2014) Retroperitoneal laparoscopic extravascular stent placement for renal nutcracker syndrome: initial experience. Urol Int 92:396–399

    Article  PubMed  Google Scholar 

  96. Li X, Ji C, Guo H (2012) Abdominal aortic transposition as a treatment alternative for posterior nutcracker syndrome. Int J Urol 19:1043–1044

    Article  PubMed  Google Scholar 

  97. Lidove O, Orozco R, Gucry B, Correas JM, Robino C, Méjean A (2001) A young woman with intermittent macroscopic haematuria. Nephrol Dial Transplant 16:853–855

    Article  PubMed  CAS  Google Scholar 

  98. Linares P, Vivas S, Dominguez A, Jorquera F, Muñoz F, Espinel J, Herrera A, Olcoz JL (2002) An uncommon association of abdominal vascular compression syndromes: Dumbar and Nutcracker. Eur J Gastroenterol Hepatol 14:1151–1153

    Article  PubMed  Google Scholar 

  99. LiLiu Y, Sun Y, Jin X (2012) Left renocaval venous bypass with autologous great saphenous vein for nutcracker syndrome. J Vasc Surg 55:1482–1484

    Article  Google Scholar 

  100. Liu Y, Sun Y, Wu XJ, Jiang Y, Jin X (2011) Endovascular stent placement for the treatment of nutcracker syndrome. Int Urol Nephrol 44:1097–1100

    Article  PubMed  Google Scholar 

  101. Luo XL, Qian GN, Xiao H, Zhao CL, Zhou XD (2012) Posterior nutcracker syndrome associated with interrupted left inferior vena cava with azygos continuation and retroaortic right renal vein. Korean J Radiol 13:345–349

    Article  PubMed  PubMed Central  Google Scholar 

  102. Lozuk B, Tanaskovic S, Radak D, Babic S, Kovacevic V, Matic P (2014) Infrarenal abdominal aorta aneurysm: a rare cause of anterior nutcracker syndrome with associated pelvic congestion. Ann Vasc Surg 28:263, e17-263.e20

    Article  PubMed  Google Scholar 

  103. Ma Z, Liu X, Ning Y, Shao J, Liu W, He X (2013) Nutcracker phenomenon in combination with glomerular nephritis in isolated hematuria patients. Int Urol Nepphrol 45:809–816

    Article  CAS  Google Scholar 

  104. Mansour MA, Rutherford RB, Metcalf RK, Pearce WH (1991) Spontaneous aorto-left renal vein fistula: the "abdominal pain, hematuria, silent left kidney" syndrome. Surgery 109:101–106

    PubMed  CAS  Google Scholar 

  105. Marone EM, Psacharopulo D, Kahlberg A, Coppi G, Chiesa R (2011) Surgical treatment of posterior nutcracker syndrome. J Vasc Surg 54:844–847

    Article  PubMed  Google Scholar 

  106. Marone EM, Psacharopulo D, Tshomba Y, Chiesa R (2011) A typical case of nutcracker phenomenon. J Vasc Surg 53:219

    Article  PubMed  Google Scholar 

  107. Marques GL, Carvalho JG, Nascimento MM, Marks SG, Olbertz LG (2012) Síndrome de quebra-nozes (nutcracker) em uma mulher jovem em investigação por quadro de hematúria recorrente: um relato de caso. J Bras Nefrol 34:195–198

    Article  PubMed  Google Scholar 

  108. Matsubara T, Ogawa O, Yanagita M (2013) Physical finding of nutcracker phenomenon. Kidney Int 83:335

    Article  PubMed  Google Scholar 

  109. Matsukura H, Arai M, Miyawaki T (2005) Nutcracker phenomenon in two siblings of a Japanese family. Pediatr Nephrol 20:237–238

    Article  PubMed  Google Scholar 

  110. Mendizábal S, Román E, Serrano A, Berbel O, Simón J (2005) Left renal vein hypertension syndrome. Nefrologia 25:141–146

    PubMed  Google Scholar 

  111. Mercieri A, Mercieri M, Armanini M, Raiteri M (2002) Exertional haematuria. Lancet 359:1402

    Article  PubMed  Google Scholar 

  112. Muller Arteaga C, Martín Martín S, Cortiñas González JR, González Fajardo JA, Fernández del Busto E (2009) Síndrome del cascanueces posterior: vena renal retroaórtica asociada a fístula arteriovenosa y carcinoma renal. A propósito de un caso y revisión de la literature. Actas Urol Esp 33:101–104

    Article  PubMed  Google Scholar 

  113. Murayama S, Shimoda Y, Kishikawa T (1989) Pyelovenous backflow in left renal vein hypertension: case report. Radiat Med 7:55–57

    PubMed  CAS  Google Scholar 

  114. Nam JK, Park SW, Lee SD, Chung MK (2010) The clinical significance of a retroaortic left renal vein. Korean J Urol 51:276–280

    Article  PubMed  PubMed Central  Google Scholar 

  115. Natale F, Granato C, Aronne L, Di Marco GM, Lo Priore E, Mocerino R, Cirillo C, Calabrò P, Golino P, Russo MG, Calabrò R (2009) Nutcracker syndrome. Eur J Echocardiogr 10:993

    Article  PubMed  Google Scholar 

  116. Neste MG, Narasimham DL, Belcher KK (1996) Endovascular stent placement as a treatment for renal venous hypertension. J Vasc Interv Radiol 7:859–861

    Article  PubMed  CAS  Google Scholar 

  117. Okada M, Tsuzuki K, Ito S (1998) Diagnosis of the nutcracker phenomenon using two-dimensional ultrasonography. Clin Nephrol 49:35–40

    PubMed  CAS  Google Scholar 

  118. Oteki T, Nagase S, Hirayama A, Sugimoto H, Hirayama K, Hattori K, Koyama A (2004) Nutcracker syndrome associated with severe anemia and mild proteinuria. Clin Nephrol 62:62–65

    Article  PubMed  CAS  Google Scholar 

  119. Ozel A, Tufaner O, Kaya E, Maldur V (2011) US and MDCT diagnosis of a rare cause of haematuria in children: posterior nutcracker syndrome. J Ultrasound 14:89–91

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  120. Ozono Y, Harada T, Namie S, Ichinose H, Shimamine R, Nishimawa Y, Hara K (1995) The "nutcracker" phenomenon in combination with IgA nephropathy. J Int Med Res 23:126–131

    PubMed  CAS  Google Scholar 

  121. Park YB, Lim SH, Ahn JH, Kang E, Myung SC, Shim HJ, Yu SH (2000) Nutcracker syndrome: intravascular stenting approach. Nephrol Dial Transplant 15:99–101

    Article  PubMed  CAS  Google Scholar 

  122. Pastershank SP (1974) Left renal vein obstruction by a superior mesenteric artery. J Can Assoc Radiol 25:52–54

    PubMed  CAS  Google Scholar 

  123. Peker A, Yagmurlu A, Ekim M, Gokcora H, Fitoz S (2011) Nutcracker syndrome in a patient with a history of inferior vena cava ligation. J Clin Ultrasound 39:418–421

    Article  PubMed  Google Scholar 

  124. Polaina-Rusillo M, Liébana-Carpio L, Borrego-Hinojosa J, Liébana-Cañada A (2012) Gross haematuria in patients with nutcracker syndrome. Nefrologia 32:537–538

    PubMed  Google Scholar 

  125. Preza Fernandes J, Amorim R, Gomes MJ, Oliveira V, Reis A, Ribeiro-Castro J (2012) Posterior nutcracker syndrome with left renal vein duplication: a rare cause of haematuria in a 12-year-old boy. Case Rep Urol 2012:849681

    PubMed  PubMed Central  CAS  Google Scholar 

  126. Puig S, Stühlinger HG, Domanovits H, Staudenherz A, Zebenholzer K, Rebhandl W, Prokop M (2002) Posterior "Nutcracker" phenomenon in a patient with abdominal aortic aneurysm. Eur Radiol 12[Suppl 3]:S133–S135

    PubMed  Google Scholar 

  127. Qin J, Zheng XY, Jiang H (2012) Posterior nutcracker phenomenon in patient with renal arteriovenous malformation. Urology 80:e73–e74

    Article  PubMed  Google Scholar 

  128. Radisic MV, Feldman D, Diaz C, Froment RO (2007) Unexplained hematuria during pregnancy: right-sided nutcracker phenomenon. Int Urol Nephrol 39:709–711

    Article  PubMed  Google Scholar 

  129. Rana MA, Oderich GS, Bjarnason H (2013) Endovenous removal of dislodged left renal vein stent in a patient with nutcracker syndrome. Semin Vasc Surg 26:43–47

    Article  PubMed  Google Scholar 

  130. Rao J, Yang J, Liu Z, Wang L, Liu L, Yin Z, Yang L (2008) Right retrocaval ureter and left nutcracker syndrome: a case report. Urology 71:1226, e9-1226.e11

    Article  PubMed  Google Scholar 

  131. Rassi I, Khabbaz Z, Chelala D, Jebara VA (2010) A new variant of the posterior nutcracker phenomenon. J Vasc Surg 51:1279

    Article  PubMed  Google Scholar 

  132. Reed NR, Kalra M, Bower TC, Vrtiska TJ, Ricotta JJ 2nd, Gloviczki P (2009) Left renal vein transposition for nutcracker syndrome. J Vasc Surg 49:386–394

    Article  PubMed  Google Scholar 

  133. Romero Coronado J (2009) Síndrome del cascanueces. Radiologia 51:537–538

    Article  PubMed  CAS  Google Scholar 

  134. Rudloff U, Holmes RJ, Prem JT, Faust GR, Moldwin R, Siegel D (2006) Mesoaortic compression of the left renal vein (nutcracker syndrome): case reports and review of the literature. Ann Vasc Surg 20:120–129

    Article  PubMed  Google Scholar 

  135. Russo D, Minutolo R, Iaccarino V, Andreucci M, Capuano A, Savino FA (1998) Gross hematuria of uncommon origin: the nutcracker syndrome. Am J Kidney Dis 32:E3

    Article  PubMed  CAS  Google Scholar 

  136. Sacks BA, Gomori J, Lerner M, Masters R, DeWolf W (1981) Left renal venous hypertension in association with the nutcracker phenomenon. Cardiovasc Intervent Radiol 4:253–255

    Article  PubMed  CAS  Google Scholar 

  137. Salehipour M, Rasekhi A, Shirazi M, Haghpanah A, Jahanbini S, Eslahi SA (2010) The role of renal autotransplantation in treatment of nutcracker syndrome. Saudi J Kidney Dis Transpl 21:237–241

    PubMed  Google Scholar 

  138. Salehipour M, Kazemi K, Shamsaeefar A, Hekmati P, Sanaei AK, Bahador A, Aliakbarian M, Malek Hosseini SA (2014) Nutcrackerlike phenomenon is an unusual cause for gross hematuria after a kidney graft. Exp Clin Transplant. doi:10.6002/ect.2013.0260

    PubMed  Google Scholar 

  139. Santos Arrontes D, Salgado Salinas R, Chiva Robles V, Gómez de Vicente JM, Fernández González I, Costa Subias J, Páez Borda A, Berenguer Sánchez A (2003) Síndrome del cascanueces. A propósito de un caso y revisión de la literatura. Actas Urol Esp 27:726–731

    Article  PubMed  CAS  Google Scholar 

  140. Sataa S, Chekib K, Riadh M, Hamza E (2012) Nutcracker syndrome as an incidental finding on computed tomography scan after blunt abdominal trauma. Tunis Med 90:192–195

    PubMed  Google Scholar 

  141. Scultetus AH, Villavicencio JL, Gillespie DL (2001) The nutcracker syndrome: its role in the pelvic venous disorders. J Vasc Surg 34:812–819

    Article  PubMed  CAS  Google Scholar 

  142. Segawa N, Azuma H, Iwamoto Y, Sakamoto T, Suzuki T, Ueda H, Yamamoto K, Shimizu T, Kichikawa K, Katsuoka Y (1999) Expandable metallic stent placement for nutcracker phenomenon. Urology 53:631–633

    Article  PubMed  CAS  Google Scholar 

  143. Shah D, Qiu X, Shah A, Cao D (2013) Posterior nutcracker syndrome with left renal vein duplication: an uncommon cause of hematuria. Int J Surg Case Rep 4:1142–1144

    Article  PubMed  PubMed Central  Google Scholar 

  144. Shao P, Li P, Ju X, Qin C, Yin C (2014) Retroperitoneal laparoscopic reimplantation of the left renal vein for nutcracker syndrome. Urol Int. doi:10.1159/000362423

    Google Scholar 

  145. Shaper KR, Jackson JE, Williams G (1994) The nutcracker syndrome: an uncommon cause of haematuria. Br J Urol 74:144–146

    Article  PubMed  CAS  Google Scholar 

  146. Shin JI, Lee SM, Park JM, Shin YH, Lee JS, Kim MJ (2005) Doppler ultrasonographic detection of nutcracker syndrome in a young child with intussusception: a case report. Acta Paediatr 94:1510–1513

    Article  PubMed  Google Scholar 

  147. Shin JI, Park JM, Lee JS, Kim MJ (2007) Doppler ultrasonographic indices in diagnosing nutcracker syndrome in children. Pediatr Nephrol 22:409–413

    Article  PubMed  Google Scholar 

  148. Shin JI, Park JM, Lee JS, Kim MJ (2007) Effect of renal Doppler ultrasound on the detection of nutcracker syndrome in children with hematuria. Eur J Pediatr 166:399–404

    Article  PubMed  PubMed Central  Google Scholar 

  149. Shin JI, Park JM, Lee JS, Kim MJ (2007) Morphologically improved nutcracker syndrome in an 11-year-old girl with hematuria. Pediatr Int 49:677–679

    Article  PubMed  Google Scholar 

  150. Shin JI, Park JM, Lee SM, Shin YH, Kim JH, Lee JS, Kim MJ (2005) Factors affecting spontaneous resolution of hematuria in childhood nutcracker syndrome. Pediatr Nephrol 20:609–613

    Article  PubMed  Google Scholar 

  151. Shokeir AA, el-Diasty TA, Ghoneim MA (1994) The nutcracker syndrome: new methods of diagnosis and treatment. Br J Urol 74:139–143

    Article  PubMed  CAS  Google Scholar 

  152. Sillo TO, Jones KE, Willetts IE (2012) Haematuria and the retro-aortic left renal vein. BMJ Case Rep 2012. doi: 10.1136/bcr.02.2012.5836

  153. Skeik N, Gloviczki P, Macedo TA (2011) Posterior nutcracker syndrome. Vasc Endovascular Surg 45:749–755

    Article  PubMed  Google Scholar 

  154. Song Y, Wu JY, Chen JH (2013) Haematuria and the nutcracker syndrome. QJM 106:879–880

    Article  PubMed  CAS  Google Scholar 

  155. Stassen CM, Weil EH, Janevski BK (1989) Left renal vein compression syndrome ("nutcracker phenomenon"). Röfo 150:708–710

    PubMed  CAS  Google Scholar 

  156. Stewart BH, Reiman G (1982) Left renal venous hypertension "nutcracker" syndrome. Managed by direct renocaval reimplantation. Urology 20:365–369

    Article  PubMed  CAS  Google Scholar 

  157. Strul N, Vaessen S, Collard L, Ghuysen MS, Khamis J, Brisbois D, Dondelinger RF, Bonnet P, Bricteux G, Krzesinski JM (2007) Le cas clinique du mois - L’anatomie au secours des cliniciens face à une douleur répétitive abdominale: le syndrome de la pince aorto-mésentérique. Rev Med Liege 62:73–76

    PubMed  CAS  Google Scholar 

  158. Sugimoto K, Fujita S, Miyazawa T, Nishi H, Okada M, Takemura T (2012) Pediatric left renal vein entrapment syndrome diagnosed by 99mTc-albumin-conjugate scintigraphy. Nephron Clin Pract 122:122–126

    Article  PubMed  CAS  Google Scholar 

  159. Takahashi Y, Akaishi K, Sano A, Kuroda Y (1988) Intra-arterial digital subtraction angiography for children with idiopathic renal bleeding: a diagnosis of nutcracker phenomenon. Clin Nephrol 30:134–140

    PubMed  CAS  Google Scholar 

  160. Takebayashi S, Ueki T, Ikeda N, Fujikawa A (1999) Diagnosis of the nutcracker syndrome with color Doppler sonography: correlation with flow patterns on retrograde left renal venography. AJR Am J Roentgenol 172:39–43

    Article  PubMed  CAS  Google Scholar 

  161. Takemura T, Iwasa H, Yamamoto S, Hino S, Fukushima K, Isokawa S, Okada M, Yoshioka K (2000) Clinical and radiological features in four adolescents with nutcracker syndrome. Pediatr Nephrol 14:1002–1005

    Article  PubMed  CAS  Google Scholar 

  162. Tanaka H, Waga S (2004) Spontaneous remission of persistent severe hematuria in an adolescent with nutcracker syndrome: seven years' observation. Clin Exp Nephrol 8:68–70

    Article  PubMed  Google Scholar 

  163. Tsumura K, Yoshida K, Yamamoto S, Takahashi S, Iida K, Enomoto Y (2014) A case of the nutcracker syndrome developed after delivery. Case Rep Urol 2014:503017

    PubMed  PubMed Central  Google Scholar 

  164. Ullery BW, Itoga NK, Mell MW (2014) Transposition of the left renal vein for the treatment of nutcracker syndrome in children: a short-term experience. Ann Vasc Surg 28:1938, e5-e8

    Article  PubMed  Google Scholar 

  165. Ulusan Ş, Koç Z (2007) Left inferior vena cava associated with nutcracker phenomenon. Fırat Tıp Derg 12:151–153

    Google Scholar 

  166. Uzu T, Ko M, Yamato M, Takahara K, Yamauchi A (2002) A case of nutcracker syndrome presenting with hematuria in pregnancy. Nephron 91:764–765

    Article  PubMed  Google Scholar 

  167. van der Laan L, Vos JA, de Boer E, van den Berg JC, Moll FL (2004) Het centraalveneuze-compressiesyndroom: zeldzaam, maar goed endovasculair te behandelen. Ned Tijdschr Geneeskd 148:433–437

    PubMed  Google Scholar 

  168. Wang L, Yi L, Yang L, Liu Z, Rao J, Liu L, Yang J (2009) Diagnosis and surgical treatment of nutcracker syndrome: a single-center experience. Urology 73:871–876

    Article  PubMed  Google Scholar 

  169. Wang X, Zhang Y, Li C, Zhang H (2012) Results of endovascular treatment for patients with nutcracker syndrome. J Vasc Surg 56:142–148

    Article  PubMed  Google Scholar 

  170. Waseem M, Upadhyay R, Prosper G (2012) The nutcracker syndrome: an underrecognized cause of hematuria. Eur J Pediatr 171:1269–1271

    Article  PubMed  Google Scholar 

  171. Wei SM, Chen ZD, Zhou M (2003) Intravenous stent placement for treatment of the nutcracker syndrome. J Urol 170:1934–1935

    Article  PubMed  Google Scholar 

  172. Weiner SN, Bernstein RG, Morehouse H, Golden RA (1983) Hematuria secondary to left peripelvic and gonadal vein varices. Urology 22:81–84

    Article  PubMed  CAS  Google Scholar 

  173. Wendel RG, Crawford ED, Hehman KN (1980) The "nutcracker" phenomenon: an unusual cause for renal varicosities with hematuria. J Urol 123:761–763

    PubMed  CAS  Google Scholar 

  174. Wenwei C, Xiangzhi Y, Huazhi X, Yirong Y, Chengdi L (2014) Left nutcracker syndrome and right ureteropelvic junction obstruction. Asian J Surg 37:167–169

    Article  PubMed  Google Scholar 

  175. Wilson Denham SL, Hester FA, Weber TM (2013) Abdominal pain of vascular origin: nutcracker syndrome. Ultrasound Q 29:263–265

    Article  PubMed  Google Scholar 

  176. Wolfish NM, McLaine PN (1985) Idiopathic unilateral gross hematuria in the pediatric patient. Am J Nephrol 5:59–62

    Article  PubMed  CAS  Google Scholar 

  177. Wolfish NM, McLaine PN, Martin D (1986) Renal vein entrapment syndrome: frequency and diagnosis. A lesson in conservatism. Clin Nephrol 26:96–100

    PubMed  CAS  Google Scholar 

  178. Xu D, Gao Y, Chen J, Wang J, Ye J, Liu Y (2013) Laparoscopic inferior mesenteric-gonadal vein bypass for the treatment of nutcracker syndrome. J Vasc Surg 57:1429–1431

    Article  PubMed  Google Scholar 

  179. Xu D, Liu Y, Gao Y, Zhang L, Wang J, Che J, Zhu Y (2009) Management of renal nutcracker syndrome by retroperitoneal laparoscopic nephrectomy with ex vivo autograft repair and autotransplantation: a case report and review of the literature. J Med Case Rep 3:82

    Article  PubMed  PubMed Central  Google Scholar 

  180. Yagci B, Tavasli B, Karabulut N, Kiroglu Y (2008) Clinical significance and renal haemodynamics of incidentally detected retroaortic left renal vein: assessment with venous Doppler sonography. Br J Radiol 81:187–191

    Article  PubMed  CAS  Google Scholar 

  181. Yang BZ, Li Z, Wang ZG (2012) Br J Nutcracker syndrome due to left-sided inferior vena cava compression and treated with superior mesenteric artery transposition. J Vasc Surg 56:816–818

    Article  PubMed  Google Scholar 

  182. Yang WJ, Liu YP, Yang FS (2007) Mesoaortic compression of left renal vein revealed by multidetector computed tomography: nutcracker syndrome. Emerg Med J 24:636

    Article  PubMed  PubMed Central  Google Scholar 

  183. Yun SJ, Nam DH, Ryu JK, Kim JS (2014) The roles of the liver and pancreas in renal nutcracker syndrome. Eur J Radiol 83:1765–1770

    Article  PubMed  Google Scholar 

  184. Yu G, Bo S (2003) The nutcracker syndrome. J Urol 169:2293–2294

    Article  PubMed  Google Scholar 

  185. Zapardiel I, Sanfrutos L, Perez-Medina T, Godoy-Tundidor V, Delafuente-Valero J, Zanagnolo V (2010) Clinical management of Nutcracker's syndrome during pregnancy. J Matern Fetal Neonatal Med 23:589–592

    Article  PubMed  Google Scholar 

  186. Zhang H, Li M, Jin W, San P, Xu P, Pan S (2007) The left renal entrapment syndrome: diagnosis and treatment. Ann Vasc Surg 21:198–203

    Article  PubMed  Google Scholar 

  187. Zhang H, Zhang N, Li M, Jin W, Pan S, Wang Z, Feng H (2003) Treatment of six cases of left renal nutcracker phenomenon: surgery and endografting. Chin Med J (Engl) 116:1782–1784

    Google Scholar 

  188. Zhang Q, Zhang Y, Lou S, Liu F, Ye Z, Zhang D (2010) Laparoscopic extravascular renal vein stent placement for nutcracker syndrome. J Endourol 24:1631–1635

    Article  PubMed  Google Scholar 

  189. Zhong J, Yuan J, Chong V, Wang Z, Xu J, Ding Z (2013) The clinical application of one-stop examination with 640-slice volume CT for Nutcracker syndrome. PLoS One 8:e74365

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  190. Mazzoni MB, Kottanatu L, Simonetti GD, Ragazzi M, Bianchetti MG, Fossali EF, Milani GP (2011) Renal vein obstruction and orthostatic proteinuria: a review. Nephrol Dial Transplant 26:562–565

    Article  PubMed  Google Scholar 

  191. Fretz PC, Sandlow JI (2002) Varicocele: current concepts in pathophysiology, diagnosis, and treatment. Urol Clin N Am 29:921–937

    Article  Google Scholar 

  192. Milani G, Bianchetti MG, Bozzani S, Bettinelli A, Fossali EF (2010) Body mass index modulates postural proteinuria. Int Urol Nephrol 42:513–515

    Article  PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Mario G. Bianchetti.

Additional information

Federica A. Vianello and Marta B. M. Mazzoni contributed equally to this work.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Vianello, F.A., Mazzoni, M.B.M., Peeters, G.G.A.M. et al. Micro- and macroscopic hematuria caused by renal vein entrapment: systematic review of the literature. Pediatr Nephrol 31, 175–184 (2016). https://doi.org/10.1007/s00467-015-3045-2

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00467-015-3045-2

Keywords

Navigation