Cases reported "Respiratory Insufficiency"

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1/137. Fatal pulmonary haemorrhage during anaesthesia for bronchial artery embolization in cystic fibrosis.

    Three children with cystic fibrosis (CF) had significant pulmonary haemorrhage during anaesthetic induction prior to bronchial artery embolization (BAE). Haemorrhage was associated with rapid clinical deterioration and subsequent early death. We believe that the stresses associated with intermittent positive pressure ventilation (IPPV) were the most likely precipitant to rebleeding and that the inability to clear blood through coughing was also an important factor leading to deterioration. Intermittent positive pressure ventilation should be avoided when possible in children with CF with recent significant pulmonary haemorrhage.
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2/137. Occipitocervicothoracic fixation for spinal instability in patients with neoplastic processes.

    OBJECT: Occipitocervicothoracic (OCT) fixation and fusion is an infrequently performed procedure to treat patients with severe spinal instability. Only three cases have been reported in the literature. The authors have retrospectively reviewed their experience with performing OCT fixation in patients with neoplastic processes, paying particular attention to method, pain relief, and neurological status. methods: From July 1994 through July 1998, 13 of 552 patients who underwent a total of 722 spinal operations at the M. D. Anderson Cancer Center have required OCT fixation for spinal instability caused by neoplastic processes (12 of 13 patients) or rheumatoid arthritis (one of 13 patients). Fixation was achieved by attaching two intraoperatively contoured titanium rods to the occiput via burr holes and Luque wires or cables; to the cervical spinous processes with wisconsin wires; and to the thoracic spine with a combination of transverse process and pedicle hooks. Crosslinks were used to attain additional stability. In all patients but one arthrodesis was performed using allograft. At a follow-up duration of 1 to 45 months (mean 14 months), six of the 12 patients with neoplasms remained alive, whereas the other six patients had died of malignant primary disease. There were no deaths related to the surgical procedure. Postoperatively, one patient experienced respiratory insufficiency, and two patients required revision of rotational or free myocutaneous flaps. All patients who presented with spine-based pain experienced a reduction in pain, as measured by a visual analog scale for pain. All patients who were neurologically intact preoperatively remained so; seven of seven patients with neurological impairment improved; and six of seven patients improved one Frankel grade. There were no occurrences of instrumentation failure or hardware-related complications. In one patient a revision of the instrumentation was required 13.5 months following the initial surgery for progression of malignant fibrous histiosarcoma. CONCLUSIONS: In selected patients, OCT fixation is an effective means of attaining stabilization that can provide pain relief and neurological preservation or improvement.
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3/137. Familial extensive idiopathic bilateral pleural fibrosis.

    The authors report three sisters with bilateral isolated apical pleural fibrosis of unknown origin, which did not respond to empirical antituberculosis therapy and oral corticosteroids. The disease evolved in an unrelenting fashion producing pleural fibrosis at the lung bases and leading to the death of two sisters and to lung transplantation in the other one. There was no history of other familial disease or consanguinity. The particular features of these cases and the differences from other reports of apparently cryptogenic pleural fibrosis are outlined.
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4/137. Treatment of primary pure angiosarcoma of ovary with multiple lung metastases: a case report.

    Angiosarcomas rarely involve the female genital tract. There have only been sporadic case reports of angiosarcomas of the cervix, uterus, vagina, parametrium, broad ligament and pelvis, and only 11 well-documented case reports of primary ovarian angiosarcoma in the English language literature to date. We present a case of primary pure ovarian angiosarcoma with lung metastasis that had partial response after chemotherapy with adriamycin and ifosfamide. But pulmonary hemorrhage and respiratory failure resulted in her death 7 months after initial diagnosis.
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5/137. Urethral atresia in a neonate with alveolar capillary dysplasia and pulmonary venous misalignment.

    Urethral atresia and alveolar capillary dysplasia (ACD) are rare congenital malformations. Urethral atresia is associated with severe pulmonary hypoplasia secondary to oligohydramnios. ACD is associated with pulmonary venous misalignment, results in severe pulmonary hypertension, and is uniformly fatal. We present a case of urethral atresia with successful, early placement of vesicoamniotic shunting, with resolution of the oligohydramnios, in which the neonate rapidly progressed to respiratory failure and death. Postmortem examination confirmed urethral atresia and diagnosed ACD. Given the surprisingly high mortality rate after vesicoamniotic shunting in patients with urethral atresia, we question whether there might be a possible link to ACD.
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6/137. Recurrent familial neonatal deaths: hereditary surfactant protein B deficiency.

    Hereditary surfactant protein B (SP-B) deficiency is an uncommon autosomal recessive lung disorder that causes hypoxemic respiratory failure in mature, morphologically normal infants. Recognition and diagnosis of this condition is of paramount importance, as it has significant implications for future pregnancies with a recurrence risk of 25%. In a family with three neonatal deaths over 20 years, SP-B deficiency was diagnosed following the death of the fourth affected infant. Previous deaths were mistakenly attributed to hyaline membrane disease (HMD), congenital mycoplasma hominis infection, and pulmonary hypertension, however, following the diagnosis in the proposita, SP-B deficiency was also confirmed in her deceased siblings by immunohistochemical staining of autopsy specimens. This case highlights the presentation, postnatal course, diagnosis, and therapeutic options of SP-B deficiency in addition to the mode of inheritance and the possibility of antenatal diagnosis. Genetic consultation is imperative in the investigations of recurrent neonatal deaths, especially in cases of remote events. The recent enormous advances in human genetics have shown that many conditions previously ascribed to environmental agents have a genetic basis.
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7/137. The diagnosis of brain depression in the presence of severe multisystem disease--a case study.

    Brain depression in a patient with severe multisystem disease can be a diagnostic challenge, particularly when the patient is maintained on artificial life-support systems. A case report is presented of a 13-year-old girl with severe pneumonia who was treated with prolonged cardiopulmonary bypass during which time she developed a clinical picture simulating brain death with marked depression of cerebral cortical activity on two successive EEGs. Following correction of some of her metabolic defects, the patient showed marked improvement of cortical function. Multisystem disease can be so severe as to produce a clinical picture of brain death. We wish to emphasize that brain hypofunction of depression is best evaluated by both clinical examination and the EEG, and that neither one alone is sufficient to conclude that cerebral death has occurred.
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8/137. Respiratory failure after liver transplantation.

    A rapidly growing haemangioendothelial sarcoma of the liver in a twenty-two year old woman was treated by liver transplantation. disseminated intravascular coagulation resulted in massive blood loss during surgery, and contributed to the death of the patient from respiratory failure on the fourth post-operative day, despite continuous post-operative intermittent positive-pressure ventilation. Other factors leading to her respiratory failure are discussed. There was no evidence of dysfunction in the transplanted liver.
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9/137. Nucleus of the tractus solitarius metastasis: relationship to respiratory arrest?

    BACKGROUND: A 52-year-old woman with metastases in brain and bone had clinical and radiological response to therapy but died about 10 weeks after diagnosis. General autopsy failed to identify a primary neoplasm or an anatomic cause of death. Investigation of sudden respiratory cessation was a consideration when undertaking an anatomic study of the brain. methods: review of patient records and careful examination of the brain following autopsy were carried out. RESULTS: The patient had terminal episodes of hypersomnia but episodes of sleep apnea were not observed. She received no respiratory support and no respiratory difficulties were recorded until she was pronounced dead at 7 a.m. autopsy revealed metastatic adenocarcinoma in a pattern suggestive of a primary pulmonary neoplasm, including multiple cerebral metastases, although no significant pulmonary lesions of any type were found. A 0.2 cm metastatic adenocarcinoma was found in the nucleus of the tractus solitarius (NTS). No other tumor was present in the brain stem. CONCLUSIONS: Unilateral destruction of the NTS in the medulla would have severely disturbed the most critical point of convergence of autonomic and voluntary respiratory control and of cardiocirculatory reflexes in the central autonomic network. It is postulated that this caused respiratory arrest during a state transition from sleeping to waking. Few metastases to the medulla are reported, most are relatively large, and several have caused respiratory symptoms before death. The very small metastasis in our patient could be the direct anatomic cause of death, and as such it is an unusual complication of metastatic disease of which clinicians should be aware. It is speculated that dysfunction of direct NTS connections to the pons or of connections passing close to the metastatic deposit resulted in terminal hypersomnia.
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10/137. Wound botulism in drug addicts in the United Kingdom.

    clostridium novyi has recently been identified as the causative organism responsible for the deaths of 35 heroin addicts who had injected themselves intramuscularly. We present two heroin addicts who developed C. botulinum infection following intramuscular or subcutaneous injection of heroin. Like C. novyi, this grows under anaerobic conditions and clinical presentation may be similar; however, descending motor or autonomic signs are invariably present in botulism. The prognosis is good if the diagnosis is made early and appropriate treatment commenced.
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