This 13-year-old boy was admitted at the age of 1.5 years in June 1990 to the Department of Paediatric and Oncological Surgery in Szczecin with suspected Wilms
28 PIOTR JUSZKIEWICZ, ANDRZEJ BRODKIEWICZ, ANNA WALECKA ET AL.
tumour of the left kidney. On admission, a palpable mass on the left side of the abdomen was found. Abdominal ultra sonography revealed a tumour measuring 10.0 × 9.0 cm in the lower pole of the left kidney. A four-week course of chemotherapy (AMD, VCR) was administered accor
ding to the SIOP 6 protocol followed by left nephrectomy.
Histopathology revealed clinical stage II of Wilms tumour.
After nephrectomy, the patient received a 9-month course of chemotherapy (AMD, VCR).
Follow-up after chemotherapy continued at the Outpa-tient Department of Paediatric Oncology with abdominal ultrasound and chest X-ray. Abdominal ultrasonography performed three years after surgery disclosed a 3.0 × 2.5 cm cystic lesion at the site of the removed kidney. Fine-needle biopsy of the lesion revealed bloody content. Doppler ultra-sonography of the abdomen was rescheduled due to tech-nical reasons. Unfortunately, the child did not reappear for follow-up until 2002 when a clear systolic murmur in
Fig. 1. USG. Arteriovenous fistula of left renal vessels (arrow) Ryc. 1. USG. Przetoka tętniczo-żylna naczyń nerkowych po stronie lewej (strzałka)
Fig. 2. Computer tomography. Arteriovenous fistula of left renal vessels (arrow) Ryc. 2. Tomografia komputerowa. Przetoka tętniczo-żylna naczyń nerkowych
po stronie lewej (strzałka)
Fig. 3. Abdominal aortogram. Arteriovenous fistula of left renal vessels (arrow) Ryc. 3. Arteriografia. Przetoka tętniczo-żylna naczyń nerkowych po stronie
lewej (strzałka)
Fig. 4. State after embolization of the left renal artery (arrow) Ryc. 4. Stan po embolizacji lewej tętnicy nerkowej (strzałka)
the left lumbar region was heard at physical examination.
Doppler ultrasound detected an arteriovenous fistula with turbulent flow between stumps of the left renal vessels (fig. 1). Echocardiography and ECG were unrevealing and there were no signs of circulatory failure. Blood pressure monitored for 24 hours was normal. CT of the abdomen disclosed an anastomosis between the dilated inferior vena cava, cystic lesion, and abdominal aorta (fig. 2). Arterio-graphy of the renal vessels confirmed the diagnosis of AVF (fig. 3).
Embolization of the fistula was performed with two MReye coils (length 4 cm, diameter 4 mm) with transverse fibres (Cook, USA) placed in the distal part of the renal artery stump. The procedure passed without complications and aortography confirmed complete disappearance of the fistula (fig. 4). To date, follow-up examinations (abdominal Doppler ultrasound was last done in December 2007) did not reveal any re-canalisation.
ARTERIOVENOUS FISTULA: A RARE COMPLICATION OF NEPHRECTOMY IN WILMS TUMOUR 29
Discussion
To our knowledge, there are no reports in medical lit-erature of AVF after nephrectomy in children with Wilms tumour. We found only one report of post-nephrectomy fistula in a patient with pyonephrosis caused by nephro-lithiasis and this is the first case of fistula embolization after nephrectomy in a child [2]. Coppes et al. described a case of paediatric AVF in a girl with suspected bilat-eral Wilms tumour, who underwent surgical renal biopsy [7]. Van der Zee et al. described a girl with post-traumatic renal AVF [8]. According to our e-mail correspondence with representatives of the National Wilms Tumour Study (Prof. G.J. D’Angio and Prof. M. Ritchey), post-nephrectomy AVF associated with Wilms tumour has not been reported in the American literature. Moreover, textbooks of paediatric oncology and surgery do not mention the possibility of AVF after nephrectomy in children with Wilms tumour.
Symptoms of AVF include hypertension and murmur or tremor detected at the lumbar region. Large, undiagnosed fistulas may lead to cardiomegaly. Circulatory failure may ensue [2]. Arteriovenous fistula formation is possible when the renal artery and vein are ligated in one block, occa-sionally together with surrounding vessels near the renal hilum. However, this may be the only way to perform safe excision of a kidney with tumour after initial ligation of the vessels. Arteriovenous fistula formation may be caused by simultaneous damage of the arterial or venous wall with a needle and/or necrosis or inflammation of the wall at the site of ligation of the renal vascular pedicle [6]. Fistulas usually form between renal vessels but an anastomosis may appear between the renal artery and the inferior vena cava.
Vigilant long-term postoperative follow-up with Doppler ultrasonography is crucial for the diagnosis of postopera-tive complications. There is not only the risk of local re-lapse in the form of a solid mass but also of a cystic lesion with intense arterial flow. In such cases, colour Doppler ultrasonography is recommended [3]. Arteriovenous fis-tulas appear more often on the right side (70%) probably because right renal vessels are shorter and their isolation is performed with a different technique.
Arteriovenous fistula formation in adults takes from a few months to even 50 years after surgery [9]. In our patient, the time was approximately nine years. Ultrasono-graphy without Doppler detected abnormalities three years after surgery. Bloody content was demonstrated with fine-needle biopsy drawing attention to a fistula. Nine years later, the diagnosis of AVF was made. Following arteriography, successful percutaneous transluminal embolization of the fistula via the left femoral artery was performed [10].
Conclusions
It is essential to ligate renal vessels individually during nephrectomy. Joint ligation of vessels of the renal pedicle may result in AVF formation. It is therefore of great im-portance to include Doppler ultrasonography during post-operative follow-up. Percutaneous transluminal embolization of AVF is an effective method that spares the patient the burden of a surgical procedure.
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Komentarz
Podwiązanie tętnicy nerkowej i żyły nerkowej w jednym
„en block”, niezależnie od powodów wykonania nefrektomii niesie ryzyko powstania przetoki tętniczo-żylnej.
Oryginalność przedstawionej pracy wynika z zastoso-wanej metody naprawczej. Uniknięcie powtórnej operacji naprawczej – poprzez drogę klasyczną i zastąpienie jej tech-niką małoinwazyjną podkreśla znaczenie ww. pracy.
prof. dr hab. n. med. Marek Ostrowski
A N N A L E S A C A D E M I A E M E D I C A E S T E T I N E N S I S
R O C Z N I K I P O M O R S K I E J A K A D E M I I M E D Y C Z N E J W S Z C Z E C I N I E 2009, 55, 2, 30–34
JANUSZ DOŚ, PIOTR gUTOWSKI1, MAgDALENA gÓRSKA-DOŚ