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De Beaucoudrey L.,French Institute of Health and Medical Research | De Beaucoudrey L.,University of Paris Descartes | Samarina A.,French Institute of Health and Medical Research | Samarina A.,University of Paris Descartes | And 139 more authors.
Medicine | Year: 2010

Interleukin-12 receptor β1 (IL-12Rβ1) deficiency is the most common form of Mendelian susceptibility to mycobacterial disease (MSMD). We undertook an international survey of 141 patients from 102 kindreds in 30 countries. Among 102 probands, the first infection occurred at a mean age of 2.4 years. In 78 patients, this infection was caused by Bacille Calmette-Guérin (BCG; n = 65), environmental mycobacteria (EM; also known as atypical or nontuberculous mycobacteria) (n = 9) or Mycobacterium tuberculosis (n = 4). Twenty-two of the remaining 24 probands initially presented with nontyphoidal, extraintestinal salmonellosis. Twenty of the 29 genetically affected sibs displayed clinical signs (69%); however 8 remained asymptomatic (27%). Nine nongenotyped sibs with symptoms died. Recurrent BCG infection was diagnosed in 15 cases, recurrent EM in 3 cases, recurrent salmonellosis in 22 patients. Ninety of the 132 symptomatic patients had infections with a single microorganism. Multiple infections were diagnosed in 40 cases, with combined mycobacteriosis and salmonellosis in 36 individuals. BCG disease strongly protected against subsequent EM disease (p = 0.00008). Various other infectious diseases occurred, albeit each rarely, yet candidiasis was reported in 33 of the patients (23%). Ninety-nine patients (70%) survived, with a mean age at last follow-up visit of 12.7 years ± 9.8 years (range, 0.5-46.4 yr). IL-12Rβ1 deficiency is characterized by childhood-onset mycobacteriosis and salmonellosis, rare recurrences of mycobacterial disease, and more frequent recurrence of salmonellosis. The condition has higher clinical penetrance, broader susceptibility to infections, and less favorable outcome than previously thought. © 2010 Lippincott Williams & Wilkins. Source


Miyazaki N.,Organization Kumamoto Medical Center | Akita M.,Kumamoto Rosai Hospital | Ono Y.,Kumamoto Rosai Hospital | Ejima T.,Kumamoto Rosai Hospital | Kunitoku Y.,Kumamoto Rosai Hospital
Japanese Journal of Anesthesiology | Year: 2010

We experienced two cases of anaphylaxis during anesthesia using rocuronium in two months. In both cases, we carried out intradermal test and positive reaction occurred with rocuronium. In both cases, the second anesthesia without neuromuscular blockade was uneventful. Though it is difficult to diagnose anaphylaxis, we should suspect anaphylaxis when cardiovascular collapse, bronchospasm and/or dermal symptoms occur after induction of anesthesia. Source


Kotera A.,Organization Kumamoto Medical Center | Kouzuma S.,Organization Kumamoto Medical Center | Miyazaki N.,Organization Kumamoto Medical Center | Hashimoto M.,Organization Kumamoto Medical Center | And 2 more authors.
Japanese Journal of Anesthesiology | Year: 2011

Neroleptic malignant syndrome (NMS) is a serious side effect of antipsychotic medications. The risk factors for NMS are the patient's physiologic conditions such as dehydration, malnutrition, stress, and additional administration of sedative drugs including haloperidol. We report a case of 62-year-old schizophrenic man with bowel obstruction due to rectal cancer. Colostomy under general anesthesia was scheduled, and he had not taken oral medication. After intravenous injection of haloperidol for sedation, muscle rigidity, high fever, and an elevated serum level of creatine Phosphokinase were observed. He was diagnosed as NMS, and sodium dantrolene was administerd. After the improvement of NMS, colostomy was done. Dehydration and malnutrition of the patient were severe at the time of operation, and the possibility of NMS developing due to stress was thought to be very high. We administered sodium dantrolene to prevent NMS after the operation, and the management for prevention of NMS is discussed. Source

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