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Volume 49, Issue 5
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2013 Volume 49 Issue 5 Pages 001-010
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSDownload PDF (338K)
Originals
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Tsuyoshi Kuwahara, Miyuki Kohno, Sadayoshi Takahashi, Yoshitomo Yasui, ...2013 Volume 49 Issue 5 Pages 969-974
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSGonadblastoma is a rare tumor that may occur in the gonad of Y-chromosome-related disorders of sex development (DSD). Although gonadoblastoma is a benign tumor, dysgerminoma could occur from gonadoblastoma. Therefore, preventive gonadal resection is recommended. A relevant period of gonadal resection was discussed based on our study on 5 cases with Y-chromosome-related DSD and other reported Japanese cases since 2000.
Among our 5 cases, gonadoblastoma was observed in 2 cases that were 8 and 4 years old at the time of surgery; no neoplastic lesion was observed in the other 3 cases (less than 2 years old). No cases had dysgerminoma. In the reported Japanese cases, gonadoblastoma was observed in 17 cases, and dysgerminoma in 10 cases (9 cases: overlapping). In our cases and reported cases, the youngest case with gonadoblastoma was 2 years old, and the other with dysgerminoma was 15 years old.
It is possible for children with Y-chromosome-related DSD to suffer gnadoblastoma at 2 years of age or older, and additionally dysgerminoma at 15 years old or older. Therefore, preventive gonadal resection should be considered for cases more than 2 years old.View full abstractDownload PDF (670K) -
Taizo Furukawa, Osamu Kimura, Koji Higuchi, Shigehisa Fumino, Shigeyos ...2013 Volume 49 Issue 5 Pages 975-980
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSPurpose: Among patients with congenital diaphragmatic hernia (CDH), those with late-presenting CDH (LP-CDH) have been reported to have a better prognosis. However, it has also been reported that the diagnosis is not always easy, and delayed diagnosis may sometimes be life-threatening. This study reports on LP-CDH cases experienced in an institution for 23 years.
Methods: There were 10 LP-CDH cases (11.6%) between January 1987 and December 2010. They were retrospectively reviewed and investigated on the relationship with their ages at the LP-CDH onset, presenting symptoms, the period taken for diagnosis, contents of herniated organs, presence or absence of hernia sac and their outcome.
Results: The median age at diagnosis of LP-CDH was 1 year and 4 months (76 days to 15 years). Presenting symptoms were respiratory failure in 3, digestive symptom such as abdominal pain with vomiting in 6 cases. One case had no symptoms, and was found on routine chest X-ray examination at healthy check-up. The mean period from onset to diagnosis was 4.2 days. Hernia occurred in the gastrointestinal tracts in all cases, and none had liver herniation. Nine of 10 cases were rescued without any complications, although 1 case developed cardiopulmonary arrest on the way to our hospital and died without surgical intervention.
Conclusions: Unlike neonatal cases, LP-CDH cases often occur with digestive problems and diagnosis is often delayed. It should be noted that emergent chest X-ray and insertion of nasogastric tube to decompress stomach contents should be performed as soon as possible for their rescue when LP-CDH is suspected.View full abstractDownload PDF (351K) -
Tasuku Kodama, Yasuhiko Iiboshi, Toshikazu Ito, Hitoshi Mizuno, Noriyu ...2013 Volume 49 Issue 5 Pages 981-985
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSPurpose: Urachal sinus is presented with infections of the umbilicus mainly in adolescents; symptomatic urachal sinus is frequently excised. However, it is unknown whether asymptomatic urachal sinuses after infection require removal. The patients with urachal sinus were reviewed to investigate the necessity of removal.
Methods: A 5-year retrospective study revealed 15 patients with urachal sinuses. An ultrasound scan revealed urachal remnants, and cystography were mainly used to detect the patency of sinus to the urinary bladder.
Results: A total of 15 patients with urachal sinus consisted of 14 males and 1 female aged 5–31 (mean: 19.3) years. Lower abdominal pain, redness and swelling of the umbilicus were observed in all patients, and discharge of pus was noted in 12. The length of sinuses were 14–75 (mean: 30.7) mm, and all patients had inflammation at the umbilicus. Patients were treated with antibiotics perorally (n = 8) and intravenously (n = 7). Umbilical granulation was improved with betamethasone valerate gentamicin sulfate in 3 patients. Abscess under the muscle layer was drained with a Nelaton catheter in 4 patients. Incision of abscess was required in 7 patients. Removal of urachal sinus was performed in 4 patients. No carcinoma and no recurrence of infection were observed for a mean 28-month follow-up period. Histological studies in 4 patients showed squamous cells and fibrous tissue, and adenocyte was not recognized.
Conclusion: All patients improved after drainage, and recurrence was not observed. Although urachal carcinoma usually occurs as adenocarcinoma just near the urinary bladder, adenocyte was not histologically revealed in our 4 patients. Therefore, propriety of prophylactic removal of urachal sinus to prevent infection and carcinoma should be reconsidered.View full abstractDownload PDF (311K)
Case Reports
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Eriko Saito, Ryugo Hiramoto, Keita Terui2013 Volume 49 Issue 5 Pages 986-990
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSA-12-year-old girl who complained of right lower abdominal pain with suspicion of acute appendicitis was referred to us. Lower abdominal tenderness with muscular defense was observed without any fever. Ultrasonography indicated a 50-mm diameter unhomogenous mass surrounded with fluid on the right side of the bladder. CT scan showed an enhanced tumor in the pelvis. Because ovarian torsion could not be ruled out, laparoscopic surgery was performed. Laparoscopic examination revealed that the right ovary had split into two portions with massive hematoma around the ovary. These findings were diagnosed as ovarian hemorrhage. The split of the ovary was then sutured. The postoperative course was uneventful, and she was discharged on postoperative day 5.
Although ovarian hemorrhage is common in adult women, it is greatly rare in pediatric patients. Without knowledge of the present clinical state, it may be difficult to diagnose appropriately. Laparoscopic examination was useful for both the diagnosis and the treatment.View full abstractDownload PDF (442K) -
Hiromi Koyama, Hirotsugu Terakura, Kazuhiko Yoshimoto, Seiichi Kawabat ...2013 Volume 49 Issue 5 Pages 991-995
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSHepatic Hemangioendothelioma (HHE) has a wide clinical spectrum, from a state of spontaneous regression to life-threatening condition. In addition, many cases require surgical treatment. We report on a case of HHE found by chance during a laparoscopic percutaneous extraperitoneal closure (LPEC) operation, showing spontaneous regression without any therapy.
A one-month-old girl underwent LPEC operation for a left external inguinal hernia with sliding ovary in our institution. Under laparoscopic observation, multiple hepatic nodules were discovered unexpectedly. Abdominal ultrasonography, enhanced CT and MRI after the operation concluded a diagnosis of HHE. Since the nodules had spread to the liver, surgical treatment was considered to be difficult. There were no symptoms in this case and HHE sometimes regresses spontaneously, we therefore observed its natural course followed by imaging examinations. The foci showed reduction in size and indistinctness 6 months later. Two years and one month later, the tumors disappeared completely on MRI.View full abstractDownload PDF (376K) -
Akira Nishi, Hideki Yamamoto, Fumiaki Toki, Norio Suzuki, Takuji Kyoya ...2013 Volume 49 Issue 5 Pages 996-1002
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSWe report a female neonate with a congenital fibrosarcoma (CFS) in the chest wall, which was resected immediately after birth. A 27-year-old primigravida woman was referred to our hospital at 24-week gestation because of a large tumor detected on the chest wall by antenatal ultrasound diagnosis. It was 70 mm in diameter, and protruded from the right side of the chest. The tumor had multi-lobular cysts, and a parenchyma fed by a feeding branch from a fetal intercostal vessel. Fetal magnetic resonance imaging also revealed the parenchyma contained a small mass of fatty tissues and calcification, suggesting teratoma. There was no signs of hydrops fetalis, fetal anemia, or fetal heart failure on fetal ultrasound diagnosis. At 31-week gestation, neonate weighing 2,199 g was born by elective cesarean section because the tumor was over 10 cm in diameter. During a period of few hours, the tumor grew progressively with severe anemia associated with thrombocytopenia and disseminated intravascular coagulation (DIC), and was urgently resected 7 hours after birth. Although spillage of the content occurred intraoperatively, the tumor was grossly removed. The tumor resected was 373 g in weight, and was well encapsulated. Histopathologic studies showed the operated site indicated no margin, suggesting complete resection of the tumor, although massive bleeding into the multiple cysts was observed. CFS was then diagnosed pathologically. Her postoperative course was uneventful. She is now 12 months old and shows no signs of recurrence that required adjuvant chemotherapy.View full abstractDownload PDF (960K) -
Mitsumasa Okamoto, Akiko Yokoi, Shogo Zuo, Mari Sonoda, Hiroshi Arai, ...2013 Volume 49 Issue 5 Pages 1003-1007
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSWe encountered a baby boy suffered from giant omphalocele with the liver and intestine contained within the sac. Soon after birth a silo using an Alexis® Wound Protector/Retractor S was sutured to the skin edges with excision of the amniotic sac. During the subsequent 11-day course, attempts at sac reduction resulted in minimal improvement. After removal of the silo, fascial closure was achieved using the GORE-TEX® Soft Tissue Patch. Skin closure was then completed. At 18 days old, due to skin necrosis around the suture line and infection under the patch, debridement of necrotic tissue and drainage under the patch using a J-VACTM drainage system were performed. To prevent infection, the patch was initially dressed with Aquacel® Ag. To avoid the occurrence of abdominal compartmental syndrome, we planned to undertake gradual closure of the fascia. The patch was removed partially and plicated at 56 days old. At 91 days old, the residual patch was removed and the abdominal wall was completely closed without stress, although patch infection had to be treated a week preoperatively. We suggest that control of patch infection without skin closure is a feasible approach, and that it is safe for the patient to have close fascia by gradual patch plication due to the lack of influence on hemodynamic and respiratory status.View full abstractDownload PDF (589K) -
Naonori Kawakubo, Shidu Miura, Yukiko Takahashi, Yoshio Zaizen2013 Volume 49 Issue 5 Pages 1008-1011
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSMallory-Weiss syndrome is characterized by lacerations of the gastro-esophageal junction caused by high intraabdominal pressure. We report a 6-year-old boy who had Mallory-Weiss tear accompanied by acute gastroenteritis with frequent vomiting. He was admitted to our hospital because of hematemesis after frequent vomiting. Upper gastrointestinal endoscopy was performed on diagnosis of upper gastrointestinal bleeding. The endoscopy revealed no lesions in the stomach and duodenum, although two lacerations in the lower esophageal mucosa close to the gastroesophageal junction were noted. After the endoscopy, he developed a fever with large-quantity diarrhea, acute gastroenteritis was therefore suspected as a cause of the vomiting. He received conservative treatment, and was discharged 4 days after admission.View full abstractDownload PDF (368K) -
Yoshitomo Yasui, Miyuki Kohno, Tsuyoshi Kuwahara, Sadayoshi Takahashi, ...2013 Volume 49 Issue 5 Pages 1012-1017
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSWe encountered 3 cases involving only male children of anorectal malformation (ARM) with recto-urethral fistula. In all cases, the level of the rectal pouch was below the I-line. We performed anterior perineal anoplasty and obtained satisfactory results in all cases. Case 1 involved a long fistulous tract running parallel to the urethra from the rectal pouch to the spongy urethra; the tract was thought to be an ano-penile-urethral fistula. Case 2 showed a rectourethral fistula and urethro-cutaneous fistula that opened to the median raphe of the scrotum and was thought to be an ano-urethro-cutaneous fistula. They were rare, unclassified cases of ARM. Case 3 involved a recto-urethral fistula. The patient underwent colostomy at birth, and then underwent anterior perineal anoplasty at 4 ～6 months of age. In this procedure, we dissected the rectal pouch and the fistula using an endoscope and a guide wire from the colostomy as an index. This procedure allowed dissection of the rectum and fistula with minimal damage to the pelvic plexus and anal sphincter. This procedure may be feasible for such cases of ARM.View full abstractDownload PDF (571K) -
Ryota Souzaki, Satoshi Ieiri, Takuya Kondo, Makoto Hashizume, Tomoaki ...2013 Volume 49 Issue 5 Pages 1018-1021
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSSeveral types of bezoars, including diospyrobezoars and trichobezoars, have been reported. Among them, trichobezoar is most common in children, and most children with a trichobezoar undergo laparotomy for removal. We herein report on the use of laparoscopic intra-gastric surgery employing an X-Gate® for a child with a gastric trichobezoar. A 13-year-old female was admitted to our department due to an abdominal mass and stomachache. A CT scan and endoscopy revealed two trichobezoars in the stomach and jejunum; they were 18 and 8 cm in diameter, respectively. Because endoscopic removal of these trichobezoars was impossible, we attempted laparoscopic intra-gastric surgery using an X-Gate® from an umbilical wound made in the stomach; the gastric trichobezoar was successfully removed. After closing the gastric wall, we cut the jejunum wall with the single port surgical technique using the X Gate®, and collected the jejunal trichobezoar in a bag. It was removed via the umbilical wound attached to the X-Gate®. The total weight of the bezoars was 554 g. The removal of the gastric trichobezoars took only one hour and 47 minutes. Our technique shortens the operation, and is superior to the conventional operation in terms of the cosmetic results. This approach could be a very useful technique for the removal of gastric trichobezoars.View full abstractDownload PDF (547K) -
Yutaka Hirayama, Yasushi Iinuma, Hisataka Iida, Daisuke Masui, Shinich ...2013 Volume 49 Issue 5 Pages 1022-1026
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSMesenteric panniculitis is a chronic non-specific inflammatory disease that arises in the mesenteric adipose tissue with an unknown origin. We herein report a rare case of mesenteric panniculitis in an 11-year-old female. She initially presented abdominal pain with a fever and diarrhea. She looked haggard and showed muscular defense. An abdominal CT scan showed ascites and an abscess in the pelvic cavity. Because we diagnosed the acute abdomen due to adnexitis, we performed an emergency operation. Laparotomy revealed redness and edema of the peritoneum, and multiple small white nodules of mesentery from the sigmoid colon to the pelvic cavity. Although we suspected the peritoneal dissemination of a malignant tumor based on the first impression, we could not find any other changes in the abdomen. Therefore, a biopsy and drainage were performed. After the operation, the pathological diagnosis revealed a small nodule as mesenteric panniculitis. This disease is difficult to diagnose before laparotomy, and is generally diagnosed during operation. Because a conservative therapy is effective for this disease, in the case of a patient who has abdominal symptoms and findings like peritonitis on an abdominal CT scan, this disease should therefore be kept in mind to make a differential diagnosis.View full abstractDownload PDF (482K) -
Kan Terawaki, Makoto Komura, Yutaka Kanamori, Masahiko Sugiyama, Kan S ...2013 Volume 49 Issue 5 Pages 1027-1031
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSThe presence of an anterior urethral valve (AUV) is one of the causes of obstructive uropathies in children. Here we report three cases of children with AUV or anterior urethral diverticula (AUD). Case 1: Fetal ultrasonography at 31 weeks of gestation revealed a lower urinary tract obstruction. After delivery, voiding cystourethrogram (VCUG) demonstrated an AUD and left vesicoureteral reflux (VUR). Temporary cutatenous vesicostomy was performed and urethral reconstruction was done when the patient was one year old.
Case 2: In a 9-month-old boy, dribbling of urine with a narrow urinary stream was noted from early infancy, and was diagnosed to have AUD with valve by VCUG. Transurethral resection of the valve was performed when he was one year old.
Case 3: A 3-year-old boy with trisomy 21 complained of difficulty in urination. He was diagnosed with AUD and VCUG, and cutaneous vesicostomy was performed.
A total of 147 cases have been reported as ssuffering from AUV or AUD as of 2011 in Japan. Renal and bladder function can be preserved by early diagnosis and treatment. Therefore, we reviewed the symptoms at the time of diagnosis in reported Japanese cases.View full abstractDownload PDF (499K) -
Yasuhiko Iiboshi, Tasuku Kodama, Toshikazu Ito, Hitoshi Mizuno, Noriyu ...2013 Volume 49 Issue 5 Pages 1032-1036
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSA case of thyroglossal duct cyst with papillary carcinoma in a child is reported. This is a rare finding in childhood, and a therapy has not been established as yet. A 13-year-old boy presented with an elastic lump attached to the hyoid bone in the submental region along the midline. An ultrasound scan showed a 30- × 17-mm cyst which included a high-echoic lesion between the hyoid bone and mentum. The patient underwent the Sistrunk procedure for excision of the thyroglossal duct cyst. Pathological examination of the surgical specimen revealed that a papillary carcinoma arising within the thyroglossal duct cyst invade the muscle attached to the hyoid bone. A postoperative ultrasound scan showed no lesion of the thyroid and lymph node, and the postoperative course was uneventful. We followed up the observation because invasion of the papillary carcinoma was restricted in the muscle attached to the hyoid bone. The carcinoma was subsequently resected with the thyroglossal duct cyst. After 28 months of follow-up, there was no evidence of recurrence. If ultrasound scan of the neck showed a hyperechoic lesion in the thyroglossal duct cyst, we would suspect a cancer that needed careful diagnosis and therapy.View full abstractDownload PDF (373K)
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2013 Volume 49 Issue 5 Pages 9999
Published: August 20, 2013
Released on J-STAGE: August 20, 2013
JOURNAL FREE ACCESSDownload PDF (140K)
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