A 17-year-old man with the Kleine-Levin syndrome died unexpectedly of cardiopulmonary arrest during a period of autonomic instability that followed an episode of megaphagia. At autopsy, the only pertinent finding was mild depigmentation of the locus ceruleus and substantia nigra. Premortem CSF levels of 5-hydroxytryptamine (5-HT) and 5-hydroxyindoleacetic acid (5-HIAA) levels were elevated. These findings indicate that many symptoms of the Kleine-Levin syndrome are a result of a neurotransmitter imbalance in the serotonergic pathway of the brainstem.
Simple melanosis of the bladder is an uncommon condition with fewer than 15 reported cases characterized by multifocal, diffuse melanin pigmentation of the urothelial mucosa. It is frequently associated with urinary incontinence or urgency. Histologically, melanin granules are present within urothelial cells or lamina propria macrophages with or without bland-appearing mucosal melanocytes. Although considered a benign entity, the rarity of the lesion warrants regular follow-up cystoscopic evaluation with biopsies to screen for the development of malignancy, especially malignant melanoma. This study presents a "typical" case with light, cytochemical, immunohistochemical, and ultrastructural characterization.
S!CIHIE:~IIC COMPLICATIONS related to dopamine therapy have been reported in adults administered relatively high doses (greater than 10 mcg/ kgjmir'c).1 Usually these patients had pre-existing vascular disease, such as diabetes mellitus, Raynaud's disease, frostbite, or arteriosclerosis. Peripheral gangrene has been reported with tow to medium dose dopamine infusion in patients whose clinical course was complicated by shock, fever, blood loss, disseminated coagulation or pre-existing vascular disease. 2-5 However, the specific effects of these clinical findings on the peripheral adrenergic neural effector mechanism have not been stressed.The present report describes an unusual case off peripheral gangrene developing in a child that received a low to medium dose (between 4 and 9 mcg/ kg/min) intravenous dopamine infusion. This patient's clinical course was complicated by congestive heart failure, respiratory distress, oliguria, and mild disseminated intravascular coagulation. The clinical features of the case are described and the physiologic relationship of these clinical findings to function of the peripheral alpha adrenergic mechanism are discussed. Case ReportThe patient was a 23-month-old Caucasian male with a clinical and cardiac catheterization diagnosis of pentalogy of Fallot. A ~vaterston-Cc~oley shunt was performed at age 12 months. At age 23 months he underwent definitive repair of the pentalogy and takedown of the previous ivaterston shunt.The initial 24 hours postoperatively were marked by congestive heart failure, hypotension, hypoxia, and oliguria. Abnormal right ventricular compliance was reflected by high filling pressures in the right atrium (15-20 mm Hg). Left atrial pressures were lour (2-8 mm Hg), reflecting pulmonary pooling and poor right sided cardiac output. Although a low Pa02 of 33 mm Hg was recorded, the average Poz was 48 mm Hg. The disparity of blood How between the right and left lungs was visible roentgenographically. This was due to the marked disparity between the size of the pulmonary arteries in the shunted and unshunted lungs. This roentgenographic difference disappeared over the intervening days, and the P02 values improved. The mean aortic blood pressure reached a low of 36 mm Hg for less than one hour and then resumed its mean pressure of 50 to 65 mm Hg with the institution of dopamine. Mildly elevated liver enzymes were consistent with congestive heart failure. Hypofibrinogenemia, 123 n~g/dl (normal 15Q-400 mg/dl) and abnormal elevation of fibrin split products 74 mcg/ml (normal less than 10 mg/ dl) were also present. Total fluid volume in the first FIG. 1. Gangrene of fingers, sharply demarcated with proximal interphalangeal joints to the tips.
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