Article Text
Abstract
The authors report on a rare case of neonatal scrotal oedema occurring concurrently with pain upon palpation of the spermatic cord on the first day of life. An ultrasound examination showed poor perfusion of the left testicle and a thrombosis of the left renal vein; intraoperative exploration indicated necrosis of the left testicle without signs of torsion. Gorged vessels with paravasal bleeding were found in the spermatic cord. The authors hypothesise that necrosis of the testicle may result from haemorrhagic infarction caused by renal venous thrombosis. Acute scrotal discolouration with pain upon palpation in neonates is usually attributed to testicular torsion. The authors report a case where these symptoms had a different cause.
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Case report
The infant was born at 36 weeks' gestational age via caesarean delivery, following an abnormal cardiotocogram. The birth weight was 2510 g, and the Apgar score was 10/10. Due to respiratory distress, he was immediately transferred to the neonatal intensive care unit, where laboured breathing improved with continuous positive airway pressure treatment.
A livid discolouration of the left scrotum and swelling of the spermatic cord with negative transillumination were observed upon admission (figures 1 and 2). Colour duplex sonography demonstrated poor perfusion of the left testicle; additionally, an enhanced echogenicity of the left testicle was evident (figure 3). Postoperative ultrasound examination of the upper urinary tract was conducted because haematuria occurred, revealing an enlarged, poorly perfused kidney on the left side caused by renal venous thrombosis. The vena cava and the right kidney were found to be unremarkable. There was also an adrenal haemorrhage on the left. Further ultrasound examination of the retroperitoneum did not show any pathology.
Livid discolouration of the left scrotum.
Transillumination of the scrotum.
Hyperechogenic left testicle in ultrasound imaging.
The finding of an acute scrotum led to immediate surgical exploration, which showed the patterns of left-sided testicular necrosis (figure 4). Careful intraoperative examination did not show any evidence of intravaginal or extravaginal torsion of the spermatic cord. As there was no improvement of testicular perfusion during surgical exploration, the left necrotic testicle was removed (inguinal orchiectomy). Histological analysis showed fresh haemorrhage, focal parenchymal necrosis and gorged testicular vessels.
Intraoperative finding of a left necrotic testicle.
On the basis of the renal venous thrombosis, we diagnosed left-sided testicular necrosis due to haemorrhagic infarction. Urinary excretion was slightly reduced for 5 days, after which it normalized, as did serum creatinine, by the time the infant was discharged on day 11 of life. The platelet count had been intermittently abnormal (96 000/µl). While laboratory tests ruled out a clotting disorder, the infant was found to be homozygote for methylene-tetrahydrofolate reductase deficiency.1 Although ultrasound colour Doppler examinations showed normalisation of the renal size and no signs of reduced perfusion by day 11, a follow-up examination at 4 months of age showed reduced kidney volume (5 ml). Scintigraphic evaluation revealed complete non-function of the left kidney.
Discussion
Pathological processes occur frequently and well into adulthood in the vascular territory of the left testicular vein, which drains at a right angle into the left renal vein. Varicoceles, for instance, mainly occur on the left of the body.2 Under these anatomical circumstances, it is possible that thrombosis in the left renal vein is likely to cause critical compression of the left testicular vein, thus leading to disturbances in blood flow and, consequently, to haemorrhagic infarction in the left testicle. We assume that this pathomechanism is the most probable cause of acute scrotum in our patient's case, particularly because the intraoperative exploration revealed no evidence of torsion. The histological examination result showed fresh haemorrhage and focal parenchymal necrosis in the testicular tissue and gorged testicular vessels, indicating venostasis, consistent with a thrombosis in the left renal vein. The occurrence of haemorrhagic infarction in the left testicle due to thrombosis in the left renal vein is undoubtedly a rare complication. We found no description of a similar phenomenon in a literature search through PubMed. One case report describes a right-sided testicular vein thrombosis caused by cardiac catheterisation presenting as an acute scrotum. Another report describes a segmental testicular infarction but provides no explanation for this necrosis.3 4 The adrenal bleeding we described earlier occurs in 15% of neonates with renal vein thrombosis.5
In conclusion, we suggest to bear in mind that haemorrhagic infarction caused by impaired venous drainage may be the underlying diagnosis if a neonate presents with an acute enlargement of his left scrotum. If malperfusion of the testicle is diagnosed, the upper urinary tract and the retroperitoneum should be investigated by (Doppler) ultrasound. Since torsion of the spermatic cord in neonates is the most common reason for testicular venostasis, followed by necrosis and possible loss of the organ, surgical exploration should be performed to exclude this diagnosis.
Acknowledgments
The authors thank Priscilla Hermann, BA, for the linguistic assistance and Christian von Bodman, MD, for critically reviewing this manuscript.
Footnotes
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Competing interests None.
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Patient consent Obtained.
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Provenance and peer review Not commissioned; externally peer reviewed.