Transoral traumatic perforation of the pyriform sinus by a marker pen: report of an infant case.

BACKGROUND: Perforation of the pyriform sinus, included in hypopharyngeal injury, is a rare condition typically caused by iatrogenic factors. We present a case of an infant who developed deep cervical and mediastinal abscesses due to a traumatic pyriform sinus perforation caused by accidentally falling with a marker pen in the mouth. CASE PRESENTATION: An 11-month-old healthy male infant fell on a trampoline with a marker pen in his mouth. The patient developed swelling in the neck 3 h after the incident and was taken to a regional general hospital. Although a laryngoscopy showed no perforation in the oral cavity or posterior pharynx, a computed tomography (CT) scan revealed significant emphysema extending from the cervix to the mediastinum. The patient was transferred to our tertiary hospital and admitted to the intensive care unit, where he was mechanically ventilated, and antibiotic therapy was initiated. On day 3 of admission, a CT scan revealed deep abscesses in the cervical and upper posterior mediastinum with pneumomediastinum. Although his respiratory status stabilized and he was temporarily weaned, the fever recurred. Pharyngoesopagography revealed significant leakage of contrast from the left pyriform sinus to the mediastinum. Consequently, surgical drainage of the abscess was performed on day 10. Two low-pressure continuous suction drains were placed, one in the posterior mediastinum and the other close to the pyriform sinus. Pharyngoesophagography on postoperative day (POD) 7 demonstrated decreased contrast leakage into the posterior mediastinum. The patient was initiated on enteral nutrition through a nasogastric tube. The patient was discharged on POD 31 after the suction drains were replaced with open Penrose drains, and enteral nutrition via nasogastric tube was continued at home. The Penrose drains were removed on POD 54, and salivary leakage ceased on POD 111. CONCLUSIONS: Although injuries to the oral cavity and posterior pharynx are more easily recognized, the existence of injury in the pyriform sinus can be challenging to evaluate. However, prompt and appropriate management, including intubation, antibiotic therapy, surgical drainage, and nutritional support, is critical in preventing life-threatening complications.

Giant pulmonary bulla causing respiratory compromise in a very low-birthweight infant.

Acquired cystic lung disease in premature infants is a serious respiratory complication, and pulmonary interstitial emphysema (PIE) has been widely reported. We report a rare case of giant pulmonary bulla in an infant treated with bullectomy where chest computed tomography was useful in directing treatment.

Volvulus of the stomach and wandering spleen after repair of congenital diaphragmatic hernia: unexpected manifestations in a neonate.

BACKGROUND: Congenital diaphragmatic hernia (CDH) is sometimes associated with complications involving herniation of intrathoracic organs, which further increase mortality rate. We encountered a case of postoperative gastric and splenic volvulus shortly after left CDH repair in a female neonate who was treated with gastropexy. CASE PRESENTATION: At 39 weeks gestation, a female patient with left Bochdalek CDH was delivered (birth weight: 3748 g, Apgar score: 3/4). The patient was provided ventilator support with nitric oxide. After pulmonary hypertension improved, CDH repair was performed via the abdominal approach on day 7. The stomach, small intestine, large intestine, and spleen were herniated through a diaphragmatic defect of 4 × 2 cm. Although the diaphragm was directly closed, it was tight and the reconstructed diaphragm "dome" was shallow, restricting space for the spleen and stomach. Nonetheless, the spleen was positioned in the left upper abdomen and the stomach was positioned medially. The postoperative course was complicated by organo-axial gastric volvulus, and laparotomy was performed on day 14. In addition to the gastric volvulus, we confirmed a wandering splenic volvulus. The spleen was easily detorted and returned to the left upper abdomen. However, the patient experienced relapse of gastric volvulus without splenic volvulus. Gastropexy was performed electively on day 47. Postoperatively, the patient could be fed orally, and the patient's development was satisfactory 6 years after surgery. CONCLUSIONS: The cause of these rare complications appeared to be tight direct diaphragmatic closure, which reduced space for the spleen and stomach beneath the left diaphragm.

Postoperative hyperthermia-induced multiple organ failure in a child with Down syndrome: a case report.

BACKGROUND: Psychological stress has been reported to cause hyperthermia. Persistent excessive hyperthermia can, in turn, cause hypercytokinemia and organ damage. We report a case of postoperative severe hyperthermia leading to a systemic inflammatory response and multiple organ failure in a child with Down syndrome. CASE PRESENTATION: A 10-month-old native Japanese boy with Down syndrome and Hirschsprung's disease is described. Newborn screening showed congenital hypothyroidism and a ventricular septal defect, but these conditions were stable upon administration of levothyroxine and furosemide. His development was equivalent to that of a child with Down syndrome. He developed a noninfectious high fever twice after preoperative preparations at age 8 months and again at 9 months. He was readmitted to hospital at age 10 months to undergo the Soave procedure to correct Hirschsprung's disease. However, he contracted a fever immediately after the surgical procedure. Hyperthermia (42 °C) was refractory to acetaminophen treatment and deteriorated to multiple organ failure due to hypercytokinemia, with increased serum levels of interleukin-6 (44.6 pg/mL) and interleukin-10 (1010 pg/mL). He died on postoperative day 2 with hypoxemia, respiratory/metabolic acidosis, increased serum levels of transaminases, reduced coagulation, and pancytopenia. Various infectious and noninfectious causes of hyperthermia could not be identified clearly by culture or blood tests. CONCLUSIONS: We speculated that the proximate cause of the fever was psychological stress, because he suffered repeated episodes of hyperthermia after the invasive procedure. Hyperthermia, together with the immune-system disorders associated with Down syndrome, may have induced hypercytokinemia and multiple organ failure. This rare case of noninfectious postoperative hyperthermia leading to multiple organ failure may help to shed further light on the currently unclear pathogenic mechanism of hyperthermia and associated multiple organ failure during the perioperative period in children.

Multiseptate gallbladder coexisting with pancreaticobiliary maljunction treated by laparoscopic cholecystectomy: report of a pediatric case.

BACKGROUND: A multiseptate gallbladder is a very rare congenital malformation in which the lumen is divided into variously sized multiseptal compartments. The pathogenesis and natural history of this disease remain uncertain. We herein describe a pediatric case of a multiseptate gallbladder with pancreaticobiliary maljunction (PBM), which was treated by laparoscopic cholecystectomy. CASE PRESENTATION: A 5-year-old girl was referred to our hospital, because a multiseptate gallbladder had been incidentally detected on abdominal ultrasonography when she presented for transient abdominal pain. Ultrasonography showed hyperechoic septa throughout the lumen of the gallbladder, giving it a honeycomb appearance. The atrophied gallbladder had weak or no contractility. Magnetic resonance cholangiopancreatography performed to detect other coexisting biliary disorders revealed PBM without dilatation of the common bile duct. Although physical examination and laboratory tests revealed no abnormalities, we performed laparoscopic cholecystectomy to prevent cholecystitis and reduce the risk of cancer secondary to the PBM. CONCLUSIONS: In recent pediatric case reports, the indication and timing of cholecystectomy has tended to be determined by the patient's symptoms and the presence of biliary complications. In the present case, however, the combination of a multiseptate gallbladder and PBM may become problematic in the future. Surgical treatment without delay was appropriate even in this pediatric patient.

Congenital communicating bronchopulmonary foregut malformation including ectopic pancreatic tissue in an infant.

BACKGROUND: Several reports have documented that the pulmonary sequestration is in communication with the gastrointestinal tract and the concept of bronchopulmonary foregut malformation (BPFM) has become more widespread. However, there are few reports of the sequestration associated with the pancreas derived from the foregut. We describe the history and pathophysiology of BPFM including pancreatic tissue in a male infant with respiratory distress. CASE PRESENTATION: A male patient was born at 38 weeks of gestation and weighed 2752 g at birth. He developed pneumonia and was hospitalized at 3 months of age. Chest radiographs and CT scans led to the diagnosis of a lung abscess in the left lower intralobar pulmonary sequestration with aberrant arteries from the abdominal cavity. At 4 months of age, when the abscess had resolved, left lower lobectomy and the resection of the intralobar sequestration were performed. The pulmonary sequestration was conjoined with the esophagus. A fistula was found between the lower esophageal wall and the pulmonary sequestration. An additional small segment of the esophageal wall was excised. Histologically, the mediastinal surface of the sequestration tissue contained pancreatic tissue. Furthermore, esophageal and gastric tissue, cartilage tissue, and ciliated epithelium were confirmed. A definitive diagnosis of BPFM was made. CONCLUSIONS: We postulated the rare case of a communicating BPFM with intrapulmonary sequestration on one end and the esophagus on the other forming a mass lesion, which included ectopic pancreatic tissue in a male infant.

Caliceal diverticulum with ureteropelvic junction obstruction in a dysplastic kidney: a pediatric case report.

A caliceal diverticulum is a rare entity in children. Its etiology is closely associated with that of ureteropelvic junction malformations and renal dysplasia. We herein present a case of these complex disorders in an infant. A renal cyst and hydronephrosis were found in the left kidney during the fetal period. The postnatal diagnosis was hydronephrosis due to ureteropelvic junction obstruction and a caliceal diverticulum in the left dysplastic kidney. Although left renal function was severely decreased, the patient had no symptoms. Therefore, we did not perform surgical treatment. At the time of this writing, the patient was 3 years 8 months old and had developed no symptoms.

Non-occlusive intestinal ischemia in the ascending colon and rectum: a pediatric case occurring during encephalitis treatment.

BACKGROUND: Non-occlusive mesenteric ischemia (NOMI) is a rare and severe pathological condition that can cause intestinal necrosis without mechanical obstruction of the mesenteric artery. NOMI often develops during the treatment of severe disease in elderly patients and mostly occurs in the intestine supplied by the superior mesenteric artery (SMA). We experienced a 12-year-old patient with NOMI that was segmentally localized in the ascending colon and rectum during encephalitis treatment. CASE PRESENTATION: A 12-year-old boy was hospitalized with limbic encephalitis. On day 41 after admission, he abruptly developed hypotension following diarrhea and fever, and presented abdominal distension. A computed tomography scan revealed pneumatosis intestinalis localized in the ascending colon and rectum coexisting with portal venous gas. The presence of peritoneal signs required an emergency laparotomy. Intraoperatively, skip ischemic lesions were found in the ascending colon and the rectum without bowel perforation. SMA and superior rectal arterial pulsation were present, and the patient was diagnosed with NOMI. The remaining colon, from the transverse to the sigmoid colon, appeared intact. We performed a distal ileostomy without bowel resection. Postoperative colonoscopies were carried out and revealed rectal and ascending colon stenosis with ulceration but demonstrated the patency of the two lesions. We confirmed the improvement of the transient bowel strictures; therefore, the ileal stoma was closed 14 months after the previous laparotomy. CONCLUSION: NOMI can be present in childhood during encephalitis treatment and can be segmentally localized in the ascending colon and the rectum. Although NOMI is most often seen in elderly patients, we should also consider the possibility of NOMI when pediatric patients with severe illness manifest abdominal symptoms.