Cases reported "Cyanosis"

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11/38. Fourteen cases of imposed upper airway obstruction.

    Imposed upper airway obstruction was diagnosed as the cause of recurrent and severe cyanotic episodes in 14 patients. Episodes started between 0.8 and 33 months of age (median 1.4) and occurred over a period of 0.8 to 20 months (median 3.5). diagnosis was made by covert video surveillance, instituted after either (a) the observation that episodes began only in the presence of one person, or (b) characteristic findings on physiological recordings, lasting between 12 hours and three weeks, performed in hospital or at home. Surveillance was undertaken for between 15 minutes and 12 days (median 24 hours) and resulted in safety for the patient and psychiatric assessment of the parent: mother (n = 12), father (n = 1), and grandmother (n = 1). These revealed histories of sexual, physical, or emotional abuse (n = 11), self harm (n = 9), factitious illness (n = 7), eating disorder (n = 10), and previous involvement with a psychiatrist (n = 7). Management of the abusing parents is complex, but recognition of their psychosocial characteristics may allow earlier diagnosis. Imposed upper airway obstruction should be considered and excluded by physiological recordings in any infant or young child with recurrent cyanotic episodes. If physiological recordings fail to substantiate a natural cause for episodes, covert video surveillance may be essential to protect the child from further injury or death.
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12/38. Hypereosinophilia presenting as eosinophilic vasculitis and multiple peripheral artery occlusions without organ involvement.

    We report here a case with hypereosinophilia and peripheral artery occlusion. A 32-yr-old Korean woman presented to us with lower extremity swelling and pain. angiography revealed that multiple lower extremity arteries were occlusive. The biopsy specimen showed perivascular and periadnexal dense eosinophilic infiltration in dermis and subcutaneous adipose tissue. Laboratory investigations revealed a persistent hypereosinophilia. She was prescribed prednisolone 60 mg daily. Her skin lesion and pain were improved and the eosinophil count was dramatically decreased. After discharge, eosinophil count gradually increased again. cyanosis and pain of her fingers recurred. She had been treated with cyclophosphamide pulse therapy. Her eosinophilia was decreased, but the cyanosis and tingling sense were progressive. The extremity arterial stenoses were slightly progressed. skin biopsy showed perivascular eosinophilic infiltration in the dermis and cd40 ligand (CD40L) positive eosinophilic infiltration. The serum TNF-alpah was markedly increased. These results suggest that CD40L (a member of TNF-alpah superfamily) could play a role in the inflammatory processes when eosinophil infiltration and activation are observed. We prescribed prednisolone, cyclophosphamide, clopidogrel, cilostazol, beraprost and nifedipine, and she was discharged.
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ranking = 0.13028677737475
keywords = pain
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13/38. Congenital tracheo-oesophageal fistula in a young adult.

    Congenital tracheo-oesophageal fistula (TEF) is rare in adults. patients who present with repeated attacks of chest infection since birth or cough, choking and cyanosis during feeding should be investigated for TEF. It should be possible to detect all cases of tracheo-oesophageal fistulae using bronchoscopy, oesophagoscopy and CT either singly or in combination. These investigations also help in deciding on the route of exploration and the type of surgery. Disconnection of the abnormal fistulous tract brings dramatic relief of symptom and prevents further pulmonary damage.
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ranking = 0.61202631890518
keywords = chest
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14/38. Non-programmed clamping of superior vena cava. The anesthesiologist's Achilles' heel.

    This case presentation focuses on the hemodynamic alterations due to acute clamping of superior vena cava (SVC) during a right pneumonectomy for lung cancer and on the alternatives for drug administration. In a 71-yr-old female patient without clinical manifestations of SVC syndrome, this large vein was clamped for 22 minutes for patch placement after sudden and unpredictable hemorrhage. The patient became acutely cyanotic and edematous in the face and upper extremities, arterial blood pressure dropped and the venous pressure in the right internal jugular vein was elevated. Drugs for managing the patient were given endobronchially and via an established right atrium line. Postoperatively, no neurologic deficit was noted. This case demonstrates the difficulties for managing patients without superior vena cava syndrome in which acute, non-programmed intra-operative SVC clamping is performed, as this is followed by systemic and brain hemodynamic deteriorations that may lead to bad outcome.
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15/38. Dysphagia as a risk factor for sudden unexplained death in infancy.

    The TRIAD of encephalopathy, subdural haemorrhages, and retinal haemorrhages is commonly considered diagnostic of shaken baby syndrome, but the original paper describes a statistically linked QUADRAD of features, the fourth of which is a previous history of feeding difficulties (dysphagia). Recent reviews of giving pacifiers (dummies) to infants during sleeping periods have found a significant reduction in the incidence of sudden infant death Syndrome. Stimulation of swallowing is a possible connection with dysphagia, which is examined here, illustrated by a well documented case. Although amniotic fluid passes freely through the larynx of fetal mammals during fetal breathing, application of pure water to the laryngeal epithelium in infants causes choking and laryngeal closure. "water sensors" in the surface respond to lack of chloride ions and adapt very slowly or not at all. Others have found in puppies that following application of pure water only 32% resume breathing in less than 30-40s. The rest needed at least one saline flush, and some required artificial ventilation in addition. These receptors also respond to high potassium concentrations and acid or alkaline solutions. Normally, airway closure during swallowing or vomiting prevents entry of feed or oesophageal reflux, but in some forms of dysphagia leakage can occur, causing paroxysmal coughing, reflex laryngeal closure, and so prolonged apnoea. Recently, it has been realised that the TRIAD injuries can also result from high intracranial vascular pressures transmitted from intra-thoracic pressure surges during paroxysmal coughing, choking, etc. Triggering of such pressure surges by dysphagic accidents provides a physiological link to injuries commonly considered diagnostic of shaken baby syndrome, completing the statistically identified QUADRAD of features. Further dysphagic research might reveal predictive factors, and preventative measures such as feeds of optimal pH.
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ranking = 121.88272323007
keywords = breathing
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16/38. Congenital pulmonary arteriovenous fistula: a rare cause of cyanosis in childhood.

    Two children (both females) aged 15 months and 4 years are described as very rare cases of central cyanosis in childhood being caused by a congenital pulmonary arteriovenous fistula. The initial diagnosis was made based on cyanosis and chest radiographs, with normal physical, ECG, and radiological findings of the heart. They had no family history of the Rendu-Weber-Osler syndrome. The patients underwent cardiac catheterization and pulmonary angiography, where the diagnosis was confirmed. After the surgery, both were symptom-free, and had no evidence of the disease.
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keywords = chest
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17/38. Hypoxia and cyanosis in alpha-1-antitrypsin deficiency. Cirrhosis as an unusual etiologic factor.

    A patient with type PiZZ alpha-1-antitrypsin deficiency was found to have severe hypoxia despite normal pulmonary function testing and a normal chest radiograph. A nuclear medicine ventilation-perfusion study revealed a right-to-left shunt. Computed tomography showed minimal bleb formation, no diffuse changes, and hepatic changes of cirrhosis with portal hypertension. No nodular pulmonary masses or enlarged peripheral pulmonary vessels were found. The diagnosis of diffuse intrapulmonic arteriovenous shunts ("pulmonary spiders of cirrhosis") was suggested and then confirmed with a dynamic radionuclide flow study.
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ranking = 0.61202631890518
keywords = chest
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18/38. Lactobezoar in a full-term breast-fed infant.

    A full-term, healthy, breast-fed infant presenting with apnea and cyanosis was found to have a gastric bezoar during a contrast upper gastrointestinal study. The condition resolved spontaneously, with no recurrence of symptoms after resumption of feeding. This represents an unusual case, in that the infant had none of the previously described predisposing factors to bezoar formation.
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19/38. Sudden and unexpected death between 1 and 5 years.

    Of a population of 9856 children followed up from birth, 9251 of whom underwent 24 hour tape recordings of electrocardiograms and abdominal wall breathing movements during early infancy, five died suddenly and unexpectedly at home at ages ranging from 16 months to 4 years. Postmortem examination, including full histological and microbiological investigations. failed to identify abnormalities ordinarily associated with death in all five cases. Two of the children were known to have had frequent cyanotic episodes and died during these events. In the three remaining cases there was no previous history of cyanotic or apnoeic episodes. The death of one of these three children was seen by his parents and the clinical features suggested that apnoea rather than a cardiac arrhythmia was the primary mechanism for his death. As in infancy, sudden and unexpected death for which no adequate cause is found at necropsy seems to constitute a major component of mortality between 1 and 5 years.
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ranking = 60.941361615034
keywords = breathing
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20/38. Respiratory complications of metatropic dwarfism.

    Two infants with clinical and radiologic features of metatropic dwarfism presented in the neonatal period with episodes of cyanosis. Diagnostic studies to determine the etiology of these spells, including electrocardiogram, electroencephalogram, arterial blood gases, and metabolic and sepsis studies, were unremarkable. Chest roentgenograms revealed the characteristic long, narrow thoracic cage with no evidence of parenchymal disease. Cervical spine stability evaluation, pulmonary function studies, and chest impedance monitoring with qualitative air flow thermistor studies and transcutaneous oxygen monitoring were carried out. Both patients demonstrated a significant increase in resistance of the respiratory system following passive maneuvering of the head from a neutral position, suggestive of hypopharyngeal air flow obstruction. Obstructive sleep apnea resulting in cyanosis was documented in both patients. All other studies failed to yield a cause for the episodes of cyanosis. Our investigation failed to alter the clinical course which resulted in respiratory arrest and death by 7 months of age. A table is presented for the differentiation of skeletal dysplasias presenting in the perinatal period.
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ranking = 0.61202631890518
keywords = chest
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