Membranous glomerulopathy and acute interstitial nephritis following treatment with celecoxib.

Both membranous glomerulopathy and acute interstitial nephritis have been reported to occur following treatment with non-steroidal anti-inflammatory drugs. We report the first cases of membranous glomerulopathy and acute interstitial nephritis following treatment with celecoxib (Celebrex), a selective COX-2 inhibitor. The rapid and complete resolution of both conditions following discontinuation of Celebrex strongly implicates this agent in disease pathogenesis. These cases enlarge the spectrum of potential renal toxicities of the COX-2-specific non-steroidal anti-inflammatory drugs.

Findings of pelvic musculature and efficacy of laparoscopic muscle stimulator in laparoscopy-assisted anorectal pull-through for high imperforate anus.

BACKGROUND: Laparoscopic findings of levator muscle and the efficacy of laparoscopic muscle stimulator (LMS) in infants with high imperforate anus have not been reported. METHODS: Twelve patients underwent laparoscopically assisted anorectoplasty for high imperforate anus. Following laparoscopic dissection of the distal rectum and division of the fistula, levator muscles in the pelvic floor were stimulated with a 5-mm-diameter LMS. Dilatation was done by inserting a guidewire and balloon catheter through the center of the levator muscle sling and muscle complex. Rectal pull-through and anastomosis between the rectum and anus were successfully completed. RESULTS: LMS showed good contraction of levator muscles and enhanced accurate midline placement of pull-through rectum. LMS was particularly useful in observing weak muscles in infants with rectovesical fistula. CONCLUSIONS: Laparoscopy and LMS offer excellent visualization of the pelvic musculature and precise tract of rectal pull-through. Fecal continence will be assessed by long-term follow-up.

[A surgical case of giant mediastinal neurilemmoma].

A 45-year-old male was admitted to Nagoya University Hospital with dyspnea. He was examined by chest X-ray, CT, MRI, and bronchofiberscope. The chest X-ray showed a large abnormal shadow in the right lung field. A large tumor mass pressing the right lung occupied a half of thoracic cavity on a chest CT and MRI. Bronchofiberscopic findings showed a stenosis of the right intermediate bronchus. The clinical diagnosis was posterior mediastinal tumor. He underwent a posterolateral thoracotomy and the tumor was removed smoothly. The size of resected specimen was 15 x 13 x 11 cm. Histopathological examination of the specimen revealed a mediastinal neurilemmoma. He has been well for 4 months postoperatively.

Maturation of mass-screened localized adrenal neuroblastoma.

BACKGROUND/PURPOSE: In infants, neuroblastoma has been known to spontaneously differentiate into a benign ganglioneuroma. Although several investigators have compared mass-screened with unscreened, disseminated with localized, and adrenal with retroperitoneal neuroblastoma, there are very few cross-comparisons of the above parameters. Herein, the authors report the maturation of mass-screened, localized adrenal neuroblastoma. METHODS: Fifty-one mass-screened adrenal neuroblastomas were divided into 2 groups. In infants less than 1 year of age (Group A), 45 neuroblastomas were resected, whereas 6 neuroblastomas were resected after observation in 1- to 4-year-old children (group B). Histopathology of the tumors in the 2 groups was compared. Data were analyzed by X(2) test, and P <.05 was considered significant. RESULTS: According to the International Neuroblastoma Pathological Classification, 41 of 45 tumors of group A were "differentiating neuroblastoma" and 4 of 6 tumors of group B were "maturing ganglioneuroma." Maturation toward ganglioneuroblastoma was observed in 16 neuroblastomas of group A (36%) and 6 neuroblastomas of group B (100%). In group A, 58% had low mitosis karyorrhexis index (MKI); all patients in group B had low MKI. CONCLUSIONS: If left untreated, maturation of mass-screened, localized adrenal neuroblastomas is a common phenomenon. These children do not need to undergo early operation.

Surgical treatment for abdominal neuroblastoma in the laparoscopic era.

BACKGROUND: The role of laparoscopy in children with cancer has not been fully defined. The aims of this study were to develop an optimal surgical procedure for the treatment of abdominal neuroblastoma in the laparoscopic era and to evaluate the advantages and disadvantages of laparoscopy in the pediatric population. METHODS: Since July 1997, 37 children were diagnosed with abdominal neuroblastoma at our center, and 44 surgical procedures were performed on them. Patients with advanced neuroblastoma underwent laparoscopic biopsy, open biopsy, and delayed primary or second-look excision, whereas early neuroblastoma cases had either laparoscopic or open excision. We compared the length of the operation, intraoperative blood loss, length of hospital stay, complications, and time to start postoperative feeding and chemotherapy for the laparoscopic and open surgery groups. RESULTS: Length of stay and time to postoperative feeding and chemotherapy were significantly lower in the laparoscopic group than the open surgery group. However, there were no significant differences between the two groups in length of operation and intraoperative blood loss. CONCLUSION: Laparoscopic biopsy and excision of abdominal neuroblastoma are effective and efficient surgical procedures in children.

[An epithelial cyst at the frontal convexity].

We report a rare case of an epithelial cyst at the right frontal convexity. A 60-year-old woman came to our hospital suffering from dysarthria and gait disturbance. Computed tomography (CT) of the brain on admission showed a round low-density area, 6 cm in diameter, at the right frontal convexity. Midline shift and displacement of the lateral ventricle are seen. There is no contrast enhancement effect. Excision of the cyst wall and fenestration into the subarachnoid space was carried out and the patient was discharged with no neurological deficits. After 5 years, regrowth of the cyst was seen. Cyst-peritoneal shunt was performed and she was discharged uneventfully. Light microscopy showed the cyst wall consisted of a monolayer of flat epithelium and loose connective tissue. An electronmicrograph of the cyst wall showed a non-ciliated epithelial layer with microvilli and basement membrane. The epithelial cells connecting with each other through desmosome have numerous endoplasmic reticulum and mitochondria. Interdigitation of the cytoplasmic membrane was not seen. The term "epithelial cyst" seems ambiguous. We discuss the pathogenesis and the nomenclature for intracranial benign cystic lesions based on the previous literature.

Intracerebral haematomas with agenesis of the internal carotid artery and tetralogy of Fallot.

We report a rare case with tetralogy of Fallot (TOF) and agenesis of the internal carotid artery (ICA) who presented serious intracerebral haematomas. In the literature, this is the first documented case having these complications simultaneously. Extreme hypoxic insults followed by recovery were detected by O2 saturation monitor before two bleeds. Chronic brain hypoxia could make the vasculature weak, which was shown in the histological examination. A 2-year-old girl was transferred to us with a general convulsion due to intracerebral haematoma. She had been showing general cyanosis from birth due to TOF. Repeated intracerebral haemorrhages ended her life. Histological study showed dilated vascular channels in the subarachnoid space and necrotizing vasculature obstructed by fibrinous thrombi adjacent to the haematoma. Fibrosis of the vessel wall with infiltration of macrophages suggested subacute or chronic lesions rather than acute necrosis due to the multiple haemorrhages. The intracerebral haematomas and agenesis of the ICA were observed as unilateral hemispheric vascular complications of TOF. Chronic brain hypoxia could play an important role in weakening the vessel wall and erythrocytosis caused obstructing thrombi. We speculate these factors generated the intracerebral haematomas.

Acute subdural hematoma in patients who underwent aneurysm clipping--four case reports.

Four patients presented with intracranial hemorrhage mainly consisting of acute subdural hematoma (ASDH), who had all undergone aneurysm clipping 2-20 years earlier. Whether the clips had slipped or new trauma had caused the bleeding was difficult to determine, since the initial computed tomography showed that the subarachnoid hemorrhage or the intracerebral hematoma developed near the clips. Angiography in three patients showed that the clips had not slipped off. Three of four ASDHs appeared in the same side as the craniotomy used for the previous aneurysm surgery. Anti-platelet agents and ventriculoperitoneal shunting had been previously used in two patients with no causal signs of trauma. The outcomes were poor in three patients and one patient died. Weakening of the extra- or intracranial structure after aneurysm surgery might have been involved together with the postoperative anti-platelet agent and shunt treatment in the etiology of the present ASDH.

Free hand aspiration for large periventricular hemorrhage. Case report.

BACKGROUND: At present, there are several therapeutic options, including craniotomy and stereotactic aspiration, for large intracerebral hemorrhage perforating into the lateral ventricle. In the cases with Glasgow Coma Scale (GCS) scores under 6 with anisocoria, external ventricular drainage would be the first choice [2-4]. We have also performed anterior ventricular horn puncture in a standard manner. The target was the foramen of Monro, at a depth of 5.5 cm from the inner table of the skull. The point of insertion was located just anterior to the coronal suture, approximately 10 cm above the nasion, and 3 cm from the midline [1]. However, we noticed that the insertion of a catheter into the periventricular hematoma adjacent to the lateral ventricle was made easier by tilting the catheter 30 degrees laterally as in the first case (Figure 1). METHODS: In our method, inclining the catheter by 30 degrees laterally, we used a silicone tube 3.5 mm in internal diameter (Silascon ventricle drainage tube, Kaneka Medix Corp., Osaka, Japan) and then replaced it with another Silascon tube with a 2.5 mm-internal diameter. From January 1999 through December 2000, eleven patients who all had GCS scores under 6 and anisocoria preoperatively were treated by this method. The series included two patients who were undergoing hemodialysis because of renal failure, two with bleeding tendency because of liver dysfunction, and one with heart failure. RESULTS: The insertion itself caused no complications. Cerebrospinal fluid was drained smoothly after removal of hematoma because the hematoma cavity connecting with the lateral ventricle was opened. Two typical cases using this technique are shown (Figures 1 and 2). All patients recovered favorable consciousness postoperatively compared with the preoperative state but hemiparesis remained. Postsurgical follow up at 3 months revealed the outcomes evaluated by Glasgow Outcome Scale (GOS) were moderate disability in 5 patients and severe disability in 6 patients . CONCLUSION: This direct aspiration and drainage of a large intracerebral hematoma that ruptures into the lateral ventricle is superior to simple ventricular drainage in regard to the removal of the hematoma clot. This technique would be the choice especially in patients with serious complications such as cardiac disease and renal failure.

Elicitation of N400m in sentence comprehension due to lexical prosody incongruity.

The role of lexical prosody in the semantic integration of spoken sentences consisting of a quiz stem and an answer word was investigated analyzing the event-related magnetic response, N400m. Three conditions regarding the relations between the quiz and the answer word were prepared: pitch-accent violation, phonemic violation and no violation. Both the pitch-accent and phonemic violations elicited significant N400m without any significant differences in the peak latency and magnitude of the equivalent current dipoles, suggesting that the role of pitch-accent in semantic integration is equivalent to that of phonemes. However, the rate of violation detection and the successful N400m source estimation were lower for the pitch-accent violation than for the phonemic violation, suggesting differential neural processes for the phonemic and prosodic cues.

Challenges of laparoscopic resection of abdominal neuroblastoma with lymphadenectomy. A preliminary report.

BACKGROUND: The laparoscopic procedure involving total resection of abdominal neuroblastoma combined with lymphadenectomy has not been reviewed in English literature. The aim of this study was to evaluate the significance and accuracy of laparoscopic resection of abdominal neuroblastoma. METHODS: Since July 1997, five patients with abdominal neuroblastoma underwent laparoscopic resection combined with lymphadenectomy or sampling of the lymph nodes. The length of operation, intraoperative blood loss, resectability, and complications were retrospectively reviewed and evaluated. RESULTS: Four cases were managed laparoscopically, but one case was converted to open procedure because of poor visualization around large vessels. The mean operation time was 135 min and the intraoperative blood loss 52 ml. CONCLUSIONS: Good visualization of the primary tumor and large vessels is, arguably, the most important factor for successful completion of this procedure laparoscopically. Precise indicators for laparoscopic resection of abdominal neuroblastoma provide a better prognosis and a good quality of life for children with neuroblastoma.

Ultrasound activates the auditory cortex of profoundly deaf subjects.

Using three-dimensional PET, the cortical areas activated by bone-conducted ultrasound were measured from five profoundly deaf subjects and compared with the cortical areas of normal-hearing subjects activated by stimuli through bone-conducted ultrasonic, air-conducted, bone-conducted, and vibro-tactile hearing aids. All of the hearing aids, including the ultrasonic hearing aid, consistently activated the medial portion of the primary auditory cortex of the normal volunteers. The same cortical area was also significantly activated in the profoundly deaf subjects although the percentage increase in regional cerebral blood flow (rCBF) was smaller than in normal subjects. These results suggest that extra-cochlear routes convey information to the primary auditory cortex and can therefore produce detectable sound sensation even in the profoundly deaf subjects, who reported a sensation themselves.

Organized chronic subdural hematoma requiring craniotomy--five case reports.

Two child and three elderly patients underwent craniotomy for organized and/or partially calcified chronic subdural hematomas (CSHs). The characteristic feature of magnetic resonance imaging was a heterogeneous web-like structure in the hematoma cavity. Both children had undergone one side subduroperitoneal shunt for bilateral CSHs when infants. As a result, the opposite hematoma cavities persisted and developed into calcified CSHs after a couple of years. All three elderly patients with senile brain atrophy showed various systemic complications such as cerebral infarction, diabetes mellitus, leg ulceration, cirrhosis, and bleeding tendency. Craniotomy for removal of the hematoma and calcification achieved good results in all patients. Subdural space created by shunt, craniotomy, or brain atrophy and persisting for a certain period, and additional various brain damage such as microcirculatory disorder, meningitis, encephalitis, or premature delivery may be important in generating calcified or organized CSH.

Deep-seated segmental neurofibromatosis without café au lait spots.

Segmental neurofibromatosis is a rare disease characterized by neurofibromas with or without café au lait spots localized to one segment of the body. The majority of reported cases have had cutaneous neurofibromas, and patients with deep involvement have rarely been described. We report on two patients with deep-seated segmental plexiform neurofibromatosis and review the literature. All reviewed cases including the present two had no café au lait spots, axillary freckling, Lisch nodules, family history or malignant progression of disease. Differential diagnoses from neuro-fibromatosis 1 (von Recklinghausen disease) and malignant peripheral nerve sheath tumor are important for genetic counseling and avoiding overtreatment.

Repeated dedifferentiation of low-grade intraosseous osteosarcoma.

Low-grade intraosseous osteosarcoma is an uncommon bone tumor that is characterized by minimum cytological atypism and a much better prognosis than conventional osteosarcoma. This report describes a patient who had a low-grade osteosarcoma that mimicked fibrous dysplasia (FD). The tumor had an area of high-grade sarcoma at the initial diagnosis. Ten years after incomplete resection of FD-like tumor, local recurrence with areas of high-grade tumor developed. This case illustrates the potential of dedifferentiation in low-grade intraosseous osteosarcoma.

Accurate localization of an insulinoma by preoperative selective intra-arterial calcium injection and intraoperative glucose monitoring.

The role of pre- and intraoperative procedures for the localization of insulinomas has been extensively debated. We report a case of successful treatment using preoperative selective intra-arterial calcium injection and intraoperative glucose monitoring. A 12-year-old boy with hypoglycemic attacks had a large insulinoma in the head of the pancreas on computed tomography. Preoperative selective angiography combined with arterial stimulation-venous sampling (ASVS) by intra-arterial injection of calcium revealed no other insulinomas in the body and tail of the pancreas. Elevation of serum glucose on intraoperative monitoring confirmed complete enucleation of the insulinoma. Preoperative ASVS can accurately localize an insulinoma, and may help to increase the success rate of surgery and avoid blind pancreatectomy.

Laparoscopic surgery in neonates and infants weighing less than 5 kg.

BACKGROUND: Laparoscopic surgery in small infants is still an uncommon procedure in Japan. The present study was conducted to evaluate the advantages and disadvantages of laparoscopic surgery in neonates and infants weighing less than 5 kg. METHODS: Between July 1997 and November 1999, 54 infants underwent laparoscopic surgery. They were evaluated for length of operation, intra- and postoperative complications, changes in intra-operative body temperature, time to postoperative feeding, length of hospital stay and changes in serum levels of C-reactive protein (CRP), creatinine phosphokinase (CPK) and interleukin (IL)-6 on days 0, 1 and 4. These parameters in the laparoscopic pyloromyotomy (LP) and laparoscopic fundoplication groups were compared with those in the open pyloromyotomy (OP) and open fundoplication groups, respectively, which were performed during the same period. RESULTS: Three laparoscopy cases were converted to open procedures. One case of fundoplication had panperitonitis due to failed gastrostomy and required long-term parenteral nutrition. Time to postoperative feeding and length of hospital stay in the LP group were significantly shorter than in the OP group. In LP group, intra-operative body temperature did not markedly decrease during CO, pneumoperitoneum. Although serum levels of CRP, CPK and IL-6 were elevated in all groups on postoperative day 1, there were no significant differences between the groups. CONCLUSIONS: Better quality of life after laparoscopy is a significant advantage over conventional surgical procedures. This advantage not only outweighs the incidence of intra- and postoperative complications in small infants, but further emphasizes the need to improve laparoscopic techniques to avoid complications.

Intra-abdominal metastases in musculoskeletal sarcomas.

This study examined the incidence, histological type, clinical symptoms, and prognosis in patients with intra-abdominal metastases of musculoskeletal sarcomas. The medical records of 505 patients with musculoskeletal sarcomas were reviewed for examples of intra-abdominal metastases. The incidence of intra-abdominal metastases (excluding lung) was: 4% in the liver (20 patients), 1.2% in gastrointestine (6 patients), 0.8% in pancreas (4 patients), and 0.8% on the peritoneal surface (4 patients). Patients with a previous hisory of lung metastases and those with high-grade liposarcoma tended to show metastasis in the intra-abdominal organs. Most patients with liver metastasis had no symptoms. Patients with gastrointestinal metastasis had abdominal pain, anemia, and melena. Patients with pancreatic metastasis had diabetes and jaundice. Six patients underwent surgical treatment, and two of them survived for more than 2 years. Metastases within the abdomen must be considered as a possible site for dissemination of musculoskeletal sarcomas, especially in patients with advanced disease and those with liposarcoma.