Minoru Horisawa

c-Kit immunoreactive interstitial cells in the human gastrointestinal tract

c-Kit immunopositive cells are considered to be pacemakers and/or mediators of neurotransmission in the gastrointestinal tract. They also correspond to the interstitial cells of Cajal (ICs) in mice. The normal distribution of c-Kit positive cells and their relation to ICs in the human gastrointestinal tract remain unclear. In this study we examine the distribution of c-Kit positive cells and their ultrastructure in normal human tissue. We then classified them and examined their relationship to ICs. Thirty nine samples of gut from the esophagus to the sigmoid colon from humans (ranging in age from a 16 week old fetus to a 57 year old and without motility disorders), were processed for immunohistochemistry, electronmicroscopy and immuno-electronmicroscopy. c-Kit immunopositive cells were located in the external muscle from the lower esophagus to the sigmoid colon, wherever the external muscle was composed of smooth muscle cells, and they were classified morphologically into two groups. Cells in the first group were mainly spindle-shaped bipolar cells with few branches; these cells ran parallel to nearby smooth muscle. Ultrastructurally, they possessed many intermediate filaments and caveolae. The spindle-shaped cells were present in the esophagus, stomach and small intestine. The second group of cells were located only in the colon, and were multipolar or bipolar cells with numerous branches. Cells in the second group were also rich in caveolae and/or smooth endoplasmic reticulum, but intermediate filaments were not prominent. Although both groups of c-Kit immunopositive cells corresponded to ICs, some ICs in the human gut do not appear to express c-Kit immunoreactivity.

Frantz's tumor (solid and cystic tumor of the pancreas) with liver metastasis: Successful treatment and long-term follow-up

A rare case of Frantzs tumor with liver metastasis is reported. An 11-year-old girl had local resection of a Frantzs tumor in the head of the pancreas. Ten years later, she had a recurrence in the pancreatic head, and two liver metastases were also disclosed at reoperation. Pancreaticoduodenectomy and enucleation of metastatic lesions in the liver were performed. Six years after the second operation, she is well and free of disease. A review of 174 cases of Frantzs tumor disclosed that metastatic disease in the pediatric age group is extremely rare and yet the prognosis is favorable. On the other hand, the outcome in older patients, especially those over 40 years old, is very poor.

Distribution of c-Kit immunopositive cells in normal human colon and in Hirschsprung's disease

BACKGROUND Subpopulations of c-Kit immunopositive cells in the muscle coat of the gastrointestinal tract are considered pacemaker cells and have been investigated in human tissue relating to motility disorder. However, the morphology of c-kit immunopositive cells in intact human tissue is still unclear. METHODS The authors studied the distribution of c-Kit immunopositive cells in the normal human colon and their cellular configuration by confocal microscopy on whole-mount preparations. The authors then compared them with six cases of Hirschsprungs disease (HD; two of short segment aganglionosis, three of extensive aganglionosis, and one of total aganglionosis). RESULTS In the normal colon regional differences were found in the distribution of c-Kit immunopositive cells. The population in the muscle layers and at the submucosal border was larger in the anal part than in the oral part. Accumulation of positive cells at the myenteric plexus level was prominent only at the descending colon. In the descending colon of HD the authors could not demonstrate any differences in c-Kit immunopositive cells on aganglionic segments compared with the corresponding area of intact tissue. CONCLUSION More attention must be paid to these regional differences of distribution, and identical regions of affected and unaffected bowels must be compared when discussing the relation between the abnormality of c-Kit-positive cells and motility disorders including HD.

Changing profile of abdominal wall defects in Japan: Results of a national survey

BACKGROUND/PURPOSE The incidence of gastroschisis has increased over the past 3 decades in a number of countries. To elucidate the Japanese status of anterior abdominal wall defects, the Japanese Society of Pediatric Surgeons conducted a national survey in Japan. METHODS Information was obtained by sending out a questionnaire to 192 University Hospitals, Childrens hospitals, and general hospitals that each had more than 200 beds. The characteristics of the patients including the birth date, birth weight, gestations, rate of associated anomalies, rate of antenatal diagnosis and prognosis, maternal age, gravidity, history of smoking, and drug use were analyzed. RESULTS The authors obtained answers from 149 institutions, including 1,785 cases of omphalocele and 970 cases of gastroschisis, which were treated between 1975 to 1997. There was a significant increase in the incidence of gastroschisis, from 0.131 in 1975 to 1980, 0.269 in 1981 to 1985, 0.337 in 1986 to 1990, 0.461 in 1991 to 1995 to 0.467 per 10,000 births in 1996 to 1997. The incidence of omphalocele was 0.322 in 1975 to 1980, 0.567 in 1981 to 1985, 0.657 in 1986 to 1990, 0.741 in 1991 to 1995 to 0.626 per 10,000 births in 1996 to 1997, respectively. In the omphalocele group, 43.1% of the mothers were between 25 to 29 years of age, whereas in the gastroschisis group 42.6% of the mothers were 20 to 24 years of age. In the gastroschisis group, the number of primipara mothers was larger than that of multipara mothers. In the omphalocele group, approximately 10% of the mothers smoked during each period, whereas in the gastroschisis group, the percentage of smoking mothers increased chronologically from 12.9% in 1981 to 1985, 18.7% in 1986 to 1990, 23.5% in 1991 to 1995 and 29.3% in 1996 to 1997. A history of drug use by the mother was approximately 10% for both the omphalocele and gastroschisis groups. In the omphalocele group, 55.9% had associated anomalies against 21.8% in the gastroschisis group. Approximately 10% in the omphalocele group and less than 3% in the gastroschisis group showed chromosomal abnormalities. From 1986, a significant number of cases detected antenatally has been observed. CONCLUSIONS There have been substantial changes in the incidence of anterior abdominal wall defects, particularly regarding gastroschisis in Japan. The reasons for such changes are most likely multifactorial, further epidemiological monitoring is thus called for.

Anatomical reconstruction of the thyroglossal duct

The high postoperative recurrence rate of the thyroglossal duct cyst is well known. Since Sistrunks operation was used, the recurrence rate was remarkably reduced, but the anatomical description of the thyroglossal duct through the entire tract has not been clarified in detail. For a more accurate anatomical understanding of the thyroglossal duct, 10 specimens obtained from Sistrunks operation were studied using histological reconstruction, and a common running pattern of the thyroglossal duct was found. The cyst is usually located caudal to the hyoid bone mostly at the midline. The duct extends upward from the cyst ventral to the hyoid bone, with many or a few branches and secretory glands. These ducts or branches merge into a single duct at the level of the cranial portion of the hyoid bone. However, as it leaves the hyoid bone and approaches the foramen cecum, a single duct spreads out into many ductuli like the tip of a broom, which communicate with many secretory glands. There were three cases in which the duct was found behind the hyoid bone, but in no case did the thyroglossal duct run through the back of the hyoid bone. The duct behind the hyoid bone was recognized as a branch from the main duct in the dorsal direction. It ascended to the dorsal surface of the hyoid bone and terminated blind. These findings emphasized the importance of Sistrunks operation to prevent a recurrence.

Intrahepatic Bile Ducts in Biliary Atresia--A Possible Factor Determining the Prognosis

Intrahepatic bile ducts were investigated by operative cholangiography in 6 patients with the correctable type of biliary atresia, by percutaneous transhepatic cholangiography in 5 patients with noncorrectable type and by electron microscopy in biopsied hepatic tissues in 17 patients. Intrahepatic bile ducts were pathologic even in the early stage of the disease and they were involved all the way from bile ductules to large main hepatic ducts. The correlation between the prognosis and the electron microscopic presence of lumenal obstruction suggests that the pathologic change of intrahepatic bile ducts is one of the main factors determining prognosis.

What is the optimal depth for core-out toward the foramen cecum in a thyroglossal duct cyst operation

The high recurrence rate of thyroglossal duct cyst operations is well documented. Sistrunks operation is widely accepted as the best procedure to prevent recurrence. Nonetheless, the optimum depth of core-out is still not well documented. We previously reported a standard running pattern of the thyroglossal duct in an anatomical reconstruction study. In more detailed pathological studies, we have tried to determine the optimal depth for core-out toward foramen cecum and the optimal width of the hyoid bone to be resected. The following items were clarified. (1) Double the horizontal distance from midline to the most distant thyroglossal duct in front of the hyoid bone was 2.4 to 9.6 mm. (2) The length of the single duct above the hyoid bone which spreads into many ductuli as it approaches the foramen cecum was about 3 to 5 mm in 2- to 6-year old children. (3) The diameter of the thyroglossal duct at the level of the cranial top of the hyoid bone was 175 to 1,400 microns. Half of the examined cases were less than 500 microns, which may have rendered direct dissection impossible. Based on these studies, we propose: (1) that a minimum of 10 mm of the hyoid bone should be resected, and for the sake of safety, more than 15 mm is preferable; and (2) that the depth of the core-out should be less than 5 mm in young children to avoid the breakdown of the branched ductuli near the foramen cecum.

Thyroglossal duct remnant penetrating the hyoid bone—A case report

Examination of the thyroglossal duct (TGD) in a senile patient with a thyroglossal duct cyst (TGDC), as well as in children, is very valuable in understanding the pathology of TGDC. The precise anatomy of TGDC was studied in a specimen obtained from a 59-year-old man using three-dimensional reconstruction. The authors found the TGD penetrated the hyoid bone. This pathological evidence has not previously appeared in the literature. Penetration of the hyoid bone by the TGD is the result of a forward growth of the hyoid bone after development of a TGD, which had appeared ventral to the hyoid bone. In this gradual forward growth, the hyoid bone had first started to press against, before eventually engulfing, the TGD.

Intestinal Obstruction in the Terminal Ileum Caused by an Anomalous Congenital Vascular Band Between the Mesoappendix and the Mesentery: Report of a Case

We report a case of intestinal obstruction caused by a congenital abnormal vascular band in a 17-year-old boy. The patient was admitted with acute colicky abdominal pain, and an emergency laparotomy revealed that the ileum was strangulated by a fibrous band with vessels about 2 mm in diameter and 7 cm in length, extending from the antemesenterium of the terminal ileum to the mesoappendix. The affected intestine was resected with the band and the appendix. Histologically, the fibrous band was composed of loose connective tissue containing arteries, veins, and nerve fibers, suggesting that it was congenital and originated from a remnant of the ventral mesentery in the embryonic period. There have been few reports of intestinal obstruction being caused by a congenital vascular band, especially in patients beyond the pediatric age group.

先天性aganglionosisラット腸管におけるNervous System-Specific Enolase(NSE)およびS-100蛋白の分布に関する研究

Immunobiochemical and immunohistochemical studies were performed to find out changes in neural and glial components of the intestine in congenital aganglionosis rats. Nervous system-specific enolase (NSE) was used as a marker of the neural component. S-100 protein was used as a marker of the glial component. NSE and S-100 protein were quantitated by highly sensitive immunoassay. And distribution of NSE and S-100 protein of the intestine was studied by immunohistochemical methods in fetal rats of various gestational age. The results of our studies were followed; Significant decrease in NSE and S-100 protein was found in the aganglionic segment. Although there was no decrease in NSE, significant decrease in S-100 protein was found in the ganglionic segment just proximal to the aganglionic narrow segment. In the studies using rat embryos, NSE positive cells and S-100 protein positive cells were identified in the more caudal intestine with increasing gestational age. In addition, NSE positive cells appeared in the intestine few days earlier than S-100 protein positive cells did. In conclusion, our studies disclosed that there was abnormality of the glial component as well as the neural component in the intestine of Hirschsprungs disease.