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1/193. Acute confusion and unreal experiences in intensive care patients in relation to the ICU syndrome. Part II.

    The intensive care unit syndrome (ICU syndrome) is defined as an altered emotional state occurring in a highly stressful environment, which may manifest itself in various forms such as delirium, confusion, crazy dreams or unreal experiences. The purpose of this part of a study of patients' experiences is to describe and illuminate patients' experiences of acute confusion, disorientation, wakefulness, dreams and nightmares during and after their stay in the ICU. The data were obtained from 19 ventilated patients, who were interviewed twice and had stayed at least 36 hours in the ICU, the first interview being about one week after discharge from the ICU, and the second 4-8 weeks later. The hermeneutic approach used when interpreting and analysing the text from the interviews revealed that patients' experiences of unreal experiences were often associated with intense fear. Intense or continuous unbearable fear seems to result in frightening unreal experiences, which further increase the level of fear. Care actions or caring relationships with relatives or nurses can reduce this fear, which can help to prevent the occurrence and/or duration and intensity of the unreal experiences. trust and confidence in nurses or significant others and feelings of self-control or trust in self-control seemed to reduce the risk of unreal experiences so that adverse stimuli might only trigger a mild confusion.
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2/193. Successful unrelated umbilical cord blood transplantation in a child with Omenn's syndrome.

    Omenn's syndrome is a variant of combined immunodeficiency disease (CID). Like other CID forms, it causes death unless the patient receives a bone marrow transplant (BMT). Previous reports have shown that BMTs from unrelated donors in Omenn's syndrome have very poor results, with a high rate of infections during transplantation and graft rejection, when compared with transplants from related donors or patients with other CID. This study discusses the case of a 19-month-old child with Omenn's syndrome, who received an unrelated cord blood stem cell transplant (CBT). Donor and recipient had 1 HLA-Ag mismatched on HLA-B. Symptomatology improved early after CBT. The child achieved leukocytes and platelet engraftment and was discharged on day 34. His follow-up has been uneventful and at this time, 27 months after CBT, immune functions have been recovered.
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3/193. The use of patient-controlled boluses of local anaesthetic via a psoas sheath catheter in the management of malignant pain.

    patients who develop malignant infiltration of the psoas muscle and the lumbar plexus often experience a severe complex pain syndrome characterised by deep somatic pain, neuropathic pain and psoas spasm. Conventional analgesic regimes may not relieve these symptoms adequately. We describe the use of patient-controlled boluses of local anaesthetic via a psoas sheath catheter in this scenario. The recent availability of portable infusion pumps with the capability to deliver large volume boluses with long lockout times made this intervention possible and allowed the patient to be discharged home with effective relief of pain.
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4/193. Anesthesia for Freeman-Sheldon syndrome using a laryngeal mask airway.

    PURPOSE: To present a case of Freeman-Sheldon syndrome (FSS) with a previously unreported technique of anesthetic management, consisting of a malignant hyperthermia free anesthetic and laryngeal mask airway. CLINICAL FEATURES: Freeman-Sheldon syndrome (also known as whistling face syndrome, Windmill-Vane-hand syndrome, cranio-carpo-tarsal dysplasia and distal arthrogryposis type 2) is a rare congenital disorder defined by facial and skeletal abnormalities. The three basic abnormalities are microstomia with pouting lips, camptodactyly with ulnar deviation of the fingers and talipes equinovarus. patients with FSS frequently present for surgical correction of musculoskeletal or facial abnormalities. There are several anesthetic challenges including difficult airway, intravenous cannulation and regional technique. They may be at increased risk for malignant hyperthermia and postoperative pulmonary complications. We present a case of a two-year-old child with FSS undergoing elective unilateral inguinal hernia repair. A non-triggering anesthetic technique was used, consisting of 2 mg x kg(-1) propofol followed by a continuous infusion, nitrous oxide 50%/oxygen, and 3 microg x kg(-1) fentanyl. Intraoperative and postoperative analgesia was provided by an ilioinguinal nerve block with 10 ml bupivacaine 0.25% with epinephrine 1:200,000. The airway was maintained with a #2 laryngeal mask airway. The anesthetic was uneventful and there were no signs or symptoms of malignant hyperthermia. The patient was discharged home later the same day in good health. CONCLUSION: The use of a laryngeal mask airway and non-triggering anesthetic technique should be considered as options for anesthetic management in patients with FSS for short procedures that do not require neuromuscular blockade.
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5/193. Unreported RSK2 missense mutation in two male sibs with an unusually mild form of coffin-lowry syndrome.

    An unreported missense mutation of the ribosomal S6 kinase 2 (RSK2) gene has been identified in two male sibs with a mild form of coffin-lowry syndrome (CLS) inherited from their healthy mother. They exhibit transient severe hypotonia, macrocephaly, delay in closure of the fontanelles, normal gait, and mild mental retardation, associated in the first sib with transient autistic behaviour. Some dysmorphic features of CLS (in particular forearm fullness and tapering fingers) and many atypical findings (some of which were reminiscent of FG syndrome) were observed as well. The moderate phenotypic expression of this mutation extends the CLS phenotype to include less severe mental retardation and minor, hitherto unreported signs. The missense mutation identified may be less deleterious than those previously described. As this mutation occurs in a protein domain with no predicted function, it could be responsible for a conformational change affecting the protein catalytic function, since a non-polar amino acid is replaced by a charged residue.
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6/193. Thoracic epidural analgesia via the caudal approach using nerve stimulation in an infant with CATCH22.

    PURPOSE: To illustrate insertion of an epidural catheter via caudal route in a small infant under electrical stimulation guidance. CLINICAL FEATURES: A six month old boy, weighting 4.25 kg, with a diagnosis of CATCH22 (Cardiac abnormality/abnormal faces, T cell deficit due to thymic hypoplasia, cleft palate, hypocalcemia due to hypoparathyroidism resulting from 22q11 deletion) was scheduled for fundoplication and gastrostomy tube (G-tube) insertion. A combined light general anesthesia and continuous epidural anesthesia technique was selected. Following induction of general anesthesia and tracheal intubation with 1.5 mg midazolam, 10 microg fentanyl and 10 mg succinylcholine, a 16G intravenous catheter was inserted into the caudal space. A 19G epidural catheter (Arrow Flextip Plus) epidural catheter was then inserted up cranially. A low electrical current (1-10mA) was then applied through the catheter. The level of motor movement was advanced from the lower limb muscles to the upper abdominal muscles as the catheter was threaded cranially. After 19 cm of epidural catheter had been inserted, intercostal muscle movement (T9-10 level) was observed at 4.2mA. The tip of the catheter was later confirmed to be at the T9-10 interspace by radiographical imaging. The patient awakened without distress and the trachea was extubated the same evening. The infant was discharged to the ward next morning with good pain relief from a continuous epidural infusion of bupivacane 0.1% with 1 microg x ml(-1) at 1.6 ml(-1). CONCLUSION: Epidural stimulation may help placement of the epidural catheter at the appropriate dermatome for effective anesthesia and analgesia.
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7/193. Central retinal vein occlusion due to hyperviscosity syndrome.

    A 57-year-old man with no previous medical history entered the emergency department with 2 days of painless vision loss in the left eye. The patient was diagnosed with central retinal vein occlusion (CRVO) and admitted for treatment. Further work-up revealed that the cause of his CRVO was a hyperviscosity syndrome secondary to multiple myeloma. The patient received two rounds of plasmapheresis with slight recovery of vision and was discharged 28 days later.
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8/193. Solitary rectal ulcer syndrome in children.

    The solitary rectal ulcer syndrome (SRUS) is an unusual disorder in childhood. Although well recognized in adult literature, the pediatric experience with this condition is limited, so SRUS often goes unrecognized or misdiagnosed. There are very few pediatric case reports in the English literature. This report describes four patients who presented with rectal bleeding, constipation, mucous discharge, and lower abdominal pain, with a diagnosis of SRUS. The diagnosis was made by rectoscopy, defecogram, anorectal manometry and histopathological evaluation. In two patients, defecogram showed a rectocele with both, the sphincter failed to relax to voluntary squeeze pressure on anorectal manometric examination. The histopathological finding in all patients was fibrous obliteration of the lamina propria with disorientation of muscle fibers. All of the patients responded well to conservative therapy, which included defecation training, laxatives, sulfasalazine, and application of rectal sucralfate enema, and remained asymptomatic on the follow-up. Although rare in the pediatric population, SRUS should be relatively easy to recognize in the child with rectal bleeding, after elimination of other causes. If suspected, the diagnosis of SRUS may be made at endoscopy and confirmed by rectal biopsy.
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9/193. Right-sided pleural effusion in spontaneous esophageal perforation.

    Spontaneous esophageal perforation (Boerhaave's syndrome) is a rare clinical entity in which overindulgence in a large meal precedes vomiting and chest pain. early diagnosis and aggressive management are keys to minimizing the morbidity and mortality. We report an unusual presentation of this already uncommon occurrence in a 33-year-old female. She presented to the Emergency Department with severe chest pain following vomiting with hematemesis after a large meal. The initial chest radiograph showed up nothing in particular. dyspnea developed two days later, and a right-sided pleural effusion was seen on chest x-ray. Panendoscopy was highly suggestive of Boerhaave's syndrome. She underwent emergency operation. After three months of hospital care, she was discharged in relatively good condition. This case of right-sided pleural effusion extends the reported description of Boerhaave's syndrome.
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10/193. The split notochord syndrome with dorsal enteric fistula, meningomyelocele and imperforate anus.

    A male infant was referred to our department because of lumbosacral meningomyelocele, dorsal enteric fistula and imperforate anus. The mother had received a parenteral drug containing estradiol benzoate and progesterone for inducing abortion in the first trimester. She also used an anal pomade containing triamcinolone and lidocaine-HCl during the pregnancy for hemorrhoids. Sigmoid end colostomy was performed after meningomyelocele repair. On abdominal exploration a wandering spleen was detected but no other anomalies. Two months later, an abdominoperineal pullthrough was performed, and the patient was discharged well after three weeks. Our case is the sixth that had split notochord syndrome associated with dorsal enteric fistula and imperforate anus. Additionally, penoscrotal transposition and wandering spleen were present in this case. To our knowledge, these associated anomalies have been extremely rare.
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