Shigeyoshi Aoi

Intralobar pulmonary sequestration diagnosed by MR angiography

A definitive diagnosis of pulmonary sequestration necessitates evidence of a systemic arterial supply to the sequestered lung segment by aortography. We report a case of intralobar pulmonary sequestration in an 11-month-old girl in whom the diagnosis was made by contrast-enhanced magnetic resonance (MR) angiography. Contrast-enhanced MR angiography is the most reliable noninvasive method for the definitive diagnosis of pulmonary sequestration in children.

A bleeding tendency as the first symptom of a choledochal cyst

Abstract We report an 8-month-old male presenting with gingival hemorrhages and nasal bleeding as the first symptom of a choledochal cyst (CC). On physical examination, there was a large cystic mass in the right upper abdominal quadrant. Laboratory studies on admission revealed moderate liver dysfunction and a bleeding tendency due to vitamin K deficiency. After administration of 5 mg vitamin K the bleeding tendency disappeared. At laparotomy, a large CC 5 cm in diameter was found and the liver showed moderate cholestasis. The sudden onset of a bleeding tendency in infants with congenital liver or biliary-tract disease may suggest not only biliary atresia, but also CC.

Surgical intervention strategies for pediatric congenital cystic lesions of the lungs: A 20-year single-institution experience

BACKGROUND The aim of this study was to assess surgical intervention strategies for congenital cystic lesions of the lungs (CCL), focusing on the safety of lung resection. MATERIALS AND METHODS The clinical features of 27 children (CCAM, n=16; bronchial atresia, n=4; bronchogenic cyst, n=3; pulmonary sequestration, n=3; lobar emphysema, n=1) who were treated at our institution between 1995 and 2014 were analyzed. RESULTS Of the 27 patients, 14 were asymptomatic, and 13 were symptomatic. The youngest symptomatic patient presented with pneumonia at 9months of age. The mean age at surgery was 4months in the asymptomatic group and 4.1years in the symptomatic group. The mean operating time was 167minutes in the asymptomatic group and 275minutes in the symptomatic group (P<0.001). The mean amount of intraoperative bleeding was 15g in the asymptomatic group and 83.4g in the symptomatic group (P<0.05). All of the prenatally diagnosed patients underwent surgery within six months of birth. Three patients had remnant cystic lesions, all of which involved cystic lesions located over the lobulation anomalies of the lung. CONCLUSIONS To minimize surgical invasiveness, surgery for CCL should be performed during the asymptomatic period or within six months after birth.

Prenatal administration of neuropeptide bombesin promotes lung development in a rat model of nitrofen-induced congenital diaphragmatic hernia

BACKGROUND/PURPOSE Fetal medical treatment to improve lung hypoplasia in congenital diaphragmatic hernia (CDH) has yet to be established. The neuropeptide bombesin (BBS) might play an important role in lung development. The present study aims to determine whether prenatally administered BBS could be useful to promote fetal lung development in a rat model of nitrofen-induced CDH. METHODS Pregnant rats were administered with nitrofen (100mg) on gestation day 9.5 (E9.5). BBS (50mg/kg/day) was then daily infused intraperitoneally from E14, and fetal lungs were harvested on E21. The expression of PCNA was assessed by both immunohistochemical staining and RT-PCR to determine the amount of cell proliferation. Lung maturity was assessed as the expression of TTF-1, a marker of alveolar epithelial cell type II. RESULTS The lung-body-weight ratio was significantly increased in CDH/BBS(+) compared with CDH/BBS(-) (p<0.05). The number of cells stained positive for PCNA and TTF-1 was significantly decreased in CDH/BBS(+) compared with CDH/BBS(-) (p<0.01). The TTF-1 mRNA expression levels were significantly decreased in CDH/BBS(+) compared with CDH/BBS(-) (p<0.05). CONCLUSIONS Prenatally administered BBS promotes lung development in a rat model of nitrofen-induced CDH. Neuropeptide BBS could help to rescue lung hypoplasia in fetal CDH.

Tumor-homing effect of human mesenchymal stem cells in a TH-MYCN mouse model of neuroblastoma

BACKGROUND Human mesenchymal stem cells (hMSCs) are multipotent stem-like cells that are reported to have tumor-suppression effects and migration ability toward damaged tissues or tumors. The aim of this study was to analyze the tumor-homing ability of hMSCs and antitumor potency in a transgenic TH-MYCN mouse model of neuroblastoma (NB). METHODS hMSCs (3×106) labeled with DiR, a lipophilic near-infrared dye, were intraperitoneally (i.p.) or intravenously (i.v.) administered to the TH-MYCN mice. hMSC in vivo kinetics were assayed using the IVIS® imaging system for 24h after injection. Immunohistochemistry using human CD90 antibody was also performed to confirm the location of hMSCs in various organs and tumors. Furthermore, the survival curve of TH-MYCN mice treated with hMSCs was compared to a control group administered PBS. RESULTS i.p. hMSCs were recognized in the tumors of TH-MYCN mice by IVIS. hMSCs were also located inside the tumor tissue. Conversely, most of the i.v. hMSCs were captured by the lungs, and migration into the tumors was not noted. There was no significant difference in the survival between the hMSC and control groups. CONCLUSION The present study suggested that hMSCs may be potential tumor-specific therapeutic delivery vehicles in NB according to their homing potential to tumors.

Thoracoscopic resection for mediastinal thymolipoma in a child: Thoracoscopic resection of thymolipoma

Thymolipoma is a rare disease among benign tumors. We herein report the case of a child who underwent thoracoscopic resection of a large thymolipoma. A 3‐year‐old boy was diagnosed with an anterior mediastinal solid tumor. Thoracic imaging revealed a fat density mass that was 8.0 × 5.0 × 3.5 cm in size. Given the MRI findings of the tumor, we suspected that the lesion was a teratoma. We decided to perform thoracoscopic exploration and, if possible, resection of the solid tumor sequentially. We successfully resected the tumor thoracoscopically. A histological examination revealed thymolipoma. For large mediastinal tumors in the intrathoracic space in children, the thoracoscopic approach is recommended when the tumor is preoperatively considered to be benign and resectable.

Successful laparoscopic extirpation of a large omental lipoblastoma in a child

Omental lipoblastoma is extremely rare among benign tumors. We herein report the case of a child who underwent laparoscopic extirpation of a large omental lipoblastoma. A 4‐year‐old girl was diagnosed with an intra‐abdominal solid tumor. Abdominal imaging revealed a fat density mass that was well encapsulated and measured 18 × 15 × 7.5 cm in size. Considering the MRI findings and movability of the tumor, we strongly suspected that the lesion was an omental lipoblastoma. We initially decided to perform laparoscopic exploration and, if possible, extirpation of the solid tumor sequentially. A total of five trocars were used, and the tumor was found to originate from the omentum. We successfully performed complete resection of the tumor laparoscopically. A histological examination revealed lipoblastoma. For large abdominal tumors in children, the laparoscopic approach is recommended as the first procedure when the tumor is preoperatively considered to be benign and resectable.