Journal of the Japanese Society of Pediatric Surgeons

Purpose: A retractile testis is a normally descended testis that retracts into the inguinal canal as a result of cremaster muscle contraction. There has long been a controversy as to its indication of surgery. The purpose of this study is to analyze our experience of patients with retractile testis. Materials and Methods: We retrospectively reviewed the medical charts of 110 patients with a retractile testis between 1998 and 2009. Group A (14 cases): patients who received orchidopexy after annual observation. Group B (21 cases): patients who required surgery because of associated conditions such as inguinal hernia, hydrocele, and contralateral undescended testis. Group C (75 cases): patients who were followed up without surgery. Results: There were no significant differences in demographic data or follow-up period between group A and C. Group B had a significantly shorter follow-up period than group A or C. Fourteen of 89 patients (15.7%) required surgery after observation. In group A, 6 patients developed ascending testis and 2 were diagnosed as cryptorchidism. Conclusion: These findings suggest that the majority of patients with retractile testes have a spontaneously favorable outcome. However, 8 cases required orchidopexy because of ascending testis or cryptorchidism. It is important that patients with retractile testis be monitored annually until the testes have clearly descended.

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Purpose: Central venous access ports (CVAPs) are implanted to treat children with intestinal dysfunction and malignant disease. This paper aims to clarify problems with CVAP in pediatric patients. Methods: The clinical records of 98 CVAP implanted in children were retrospectively investigated. Age of patients was 4.9±4.2 years old, and 74 patients had malignant diseases. Numerical values are presented as mean ± standard deviation. Results: Body weight was 16.8±10.2 kg. In 98 catheters, 87 were inserted with cut-down procedure, 5 with exchange of a catheter from the same vein, 5 with subclavian venous puncture, and 1 with a purse-string suture. Operative time was 50±18 minutes. Of the total CVAPs, 23 were introduced with difficulty, and 2 could not be advanced to the central vein, because of venou variation. Period that CVAP remained in place was 594±432 days. Operative complications were found in 6 patients (postoperative bleeding, phlebitis, wound laceration, port infection, leakage); late complications in 18 patients (occlusion, catheter infection, leakage, phlebitis, skin necrosis). Reasons of CVAP removal were completion of treatment in 41, death in 19, exchange of a catheter in 5, occlusion in 9, infection in 3, leakage in 2, skin necrosis in 2, a broken port in 1, and unknown in 1. In 66 patients with malignant disease, 20 patients had been treated with anticancer drugs, and 26 patients had fallen in grade 4 bone marrow suppression within 10 days before operation. However, there were no differences between patients with benign disease and those with malignant disease in complication rate or period in place. Conclusions: Although the 23.5% of CVAPs led to difficulties in catheterization, 98.0% were successfully introduced into the central venous vein. Even in infants or patients with severe bone marrow suppression, CVAPs could be safely implanted, and long-term outcome was excellent. We should pay attention to prevention of occlusion and infection.

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A patient with Alport-leiomyomatosis syndrome in which leiomyomatosis was associated with Alport syndrome was treated. This patient was an 8-year-old boy. He visited the hospital with a chief complaint of vomiting and blood in the urine. A marked wall thickening from the lower thoracic esophagus to the abdominal esophagus was observed in CT and MRI, and stenosis and disorder of esophageal lumen due to tumor formation were observed in esophagography. Moreover, the middle intrathoracic esophageal wall became thickened diffusely. Leiomyomatosis was proved in tumor biopsy, and COL4A5 defect was observed in glomerular basement membrane in renal biopsy. Lower esophagectomy and cardiectomy were conducted because of the disorder of the esophagus. Although a diffuse leiomyomatosis still remains in the esophagus two years after the operation, no tendency for enlargement of the tumor is seen. The prognosis of this syndrome is poor due to renal damage from Alport syndrome. Alport-leiomyomatosis syndrome is an X-linked dominantly inherited disease, and only six cases including our own experience have been reported in this country. Since his mother and sister (already dead) also had lesions in their esophagi and kidneys with a high possibility of the same disease, a prudent heredity consultation is needed.

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Pediatric pneumothorax is rare disease but almost all cases are cured by a resection of bulla or treatment for a primary disease. We report a rare case in which the disease began at the bilateral pneumothorax and finally led to living donor lung transplantation, due to rapidly worsening of bilateral bulla and parenchymal fibrosis. Pathological diagnosis of the lung was Non-Specific Interstitial Pneumonia. The treatment for interstitial pneumonia is mainly medical. On the other hand, the one for pneumothorax is generally surgical. It is important to find out whether any primary disease exists when a treatment of pneumothorax is initiated, because surgical treatment alone will not heal a pneumothorax which has a primary medical cause. We also suggest careful decision making regarding indication of pleurodesis in a case of pneumothorax with interstitial pneumonia. It is rare but some cases of interstitial pneumonia lead to lung transplantation and the unconsidered pleurodesis to pneumothorax may be the cause of prolonged operative duration or increased intraoperative hemorrhage at lung transplantation due to intrathoracic adhesion.

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