Scrotal hematoma due to neonatal adrenal hemorrhage

Abstract

Neonatal adrenal hemorrhage affects 0.2% of newborns and mostly due to birth-trauma, large birthweight, hypoxia, and asphyxia, but it can occur spontaneously. Clinical features are varied, depending on the amount of blood lost. The frequent clinical manifestations are anemia, persistent jaundice, and abdominal distention associated with an abdominal mass. However, some infants are asymptomatic and the diagnosis is made only incidentally1. Only 19 cases of neonatal adrenal hemorrhage presenting with scrotal hematoma have been described in studies so far, and unnecessary surgery was carried out in most of these cases due to suspicion of testicular torsion2. In this study, we present a patient with hemorrhage of the right adrenal gland, who presented with a scrotal mass, misdiagnosed as torsion of the testes, and developed disseminated intravascular coagulation (DIC), postoperatively. In this paper, the importance of preoperative diagnosis and the risk of unnecessary surgical exploration of the scrotum under general anesthesia in a newborn, are discussed. A 4200 g male was born after 40 weeks’ gestation to a 41-year-old woman after an uneventful pregnancy. A difficult vaginal delivery was carried out without instrumental assistance. Apgar scores were 4 and 7 at 1 and 5 min, respectively. Only oxygen was given after birth. Pertinent findings in the initial physical examination were absence of the Moro and grasp reflexes on the right side because of right brachial plexus injury. A bluish discoloration, swelling and distinct 2 cm mass were noted in the right hemiscrotum, 20 h after birth. The mass did not transmit light. Testicular torsion was suspected and surgical exploration was done immediately. A large inguinoscrotal hematoma within the tunica vaginalis surrounding the testicles was found. Six hours after surgery, the hematocrit was 29%, hemoglobin was 10.2 g/dL and platelet count was 49 × 109/L, while preoperation values were 38%, 13.6 g/dL, and 171 × 109/L, respectively. The results of postoperative blood coagulation studies were abnormal with prolonged bleeding time, prothrombin time, and activated partial thromboplastin time. The fibrinogen level decreased to 0.88 g/dL, with increased fibrin degrading products and d-dimer levels. Postoperatively, the patient's renal and liver function tests and blood gasses were within normal limits, except for very high levels of serum aminotransferase activities. Abdominal ultrasonography revealed a large right adrenal hemorrhage (Fig. 1). Transabdominal transverse sonogram demonstrating right adrenal hematoma (arrows). The newborn was treated for DIC. Fresh frozen plasma and thrombocyte infusions were given for replacement therapy. Intravenous antibiotic treatment was given until the negative results for blood and urine cultures were reported. Serologic examination for TORCH (Toxoplasma gondii, rubella, cytomegalovirus, herpes symplex) and viral hepatitis were unremarkable. The patient's clinical condition and abnormal laboratory findings began to improve after replacement therapy for DIC, and at the end of the first week the results of serum aminotransferase activities and blood coagulation tests decreased to normal limits. The patient was discharged on the 12th day postnatal. Ultrasonographic examinations revealed resolution of the adrenal hematoma after 3 weeks. He was closely monitored with active and passive corrective exercises for right brachial plexus injury. At present, the patient is 2 years old and is physically normal, with normal size and consistency of testes, except for the sequel of brachial plexus injury despite two subsequent operations. The clinical manifestations associated with adrenal hemorrhage are anemia, scrotal swelling and bluish discoloration, as seen in this case. Scrotal swelling in the newborn can result from different causes, including torsion of the testes, epididymis and testicular appendages, hydrocele, scrotal edema, inguinal hernia, hematocele, scrotal or testicular tumor, orchitis, meconium peritonitis, and hematoma3,4. Intra- or retroperitoneal bleeding from an adrenal hemorrhage is the most common source of blood in the scrotum. The passage of blood down the inguinal canal and into the scrotum is the main cause of scrotal hematoma3. Nineteen cases of neonatal adrenal hemorrhage presenting as scrotal hematoma have been described in studies so far, and unnecessary surgical exploration of the scrotum was carried out in eight of these cases for suspected torsion of the testes. Surgical exploration showed a normal testis in all of these eight cases, and postoperative abdominal ultrasonography revealed ipsilateral adrenal hemorrhage. In most of the remaining cases, previous experiences prompted physicians to suspect adrenal hemorrhage as the cause of the scrotal swelling, and therefore surgical exploration was not done. The difficulty in differentiating torsion of the testes, epididymis, and testicular appendages from other causes of scrotal swelling remains unabated despite great strides in imaging techniques. It is frequently recommended to consider an acute scrotal incident with swelling and bluish discoloration as torsion of the testes until proved otherwise and to intervene promptly to improve testicular salvage rates in neonates. The unreliability of medical history and physical examination to distinguish torsion from other scrotal disorders has been cited frequently in other studies5. Recently, Doppler ultrasonography was found technically successful in 91% of children in differential diagnosis of acute scrotal swelling, yielding a sensitivity of 89% and specifity of 100%6. Radioisotope scans have also been used to determine whether there is blood flow to the testes in acute scrotum. Although precise preoperative diagnosis is the goal before surgical treatment, excessive preoperative evaluation should be avoided if delay places the testicle in any additional risk7. However, abdominal ultrasonography can easily confirm ipsilateral adrenal hemorrhage in a newborn with scrotal swelling and prevent misdiagnosis such as testicular torsion. A number of pathologic life-threatening processes, such as sepsis, hypoxia, acidosis, tissue necrosis, shock, and endothelial damage, may incite episodes of DIC, especially for newborns who are at greater risk than older children. DIC was not reported in any of the 19 newborns described in other studies with adrenal hemorrhage and scrotal hematoma. In this study, DIC probably occurred due to hypoxia and endothelial damage, since there was no clinical or laboratory data confirming sepsis, shock, or acidosis. During surgical exploration, sevoflurane was used as the anesthetic agent in our study. Although an early investigation in human volunteers showed no change in serum aminotransferase activities with sevoflurane anesthesia, and sevoflurane has been used safely in many children, altered hepatic blood flow and oxygenation may have an impact on hepatocellular damage, as well as hepatocellular function and hepatic drug clearance8−10. In addition, the results of animal studies suggest that if hepatic arterial flow is compromised, there may be a smaller margin of safety against hypoxia with sevoflurane compared to halothane or isoflurane11. Although the clinical implications at present are unclear, all inhaled anesthetics are capable of decreasing the albumin, transferrin, and fibrinogen synthesis during the 4 h liver perfusion. Sevoflurane decreases the synthesis of those proteins by 60−70%8. Those factors may also contribute to development of DIC. In conclusion, adrenal hemorrhage may present as scrotal swelling, mimicking a condition of acute scrotal pathology resulting in unnecessary surgical exploration. The differential diagnosis between this condition and other causes of scrotal swelling in newborns is primarily based on an accurate clinical history and physical and laboratory examination. However, it appears mandatory to perform an ultrasonographic examination of both scrotum and abdomen in all neonates presenting with acute scrotal swelling because it yields rapid and accurate results and it does not significantly delay surgery even if it is necessary. Although the anesthetic risk for the neonate undergoing testicular operation is accepted to be negligible in the current anesthesiologic practice, early intervention and diagnosis of an adrenal hemorrhage avoids unnecessary surgical exploration of the scrotum and possible postoperative surgical complications of those newborns who generally have perinatal hypoxia.