Labyrinthine Sequestrum: A Case Report and Review of the Literature.

OBJECTIVE: To report the presentation, diagnosis, management, and convalescence of labyrinthine sequestrum (LS) and summarize all previously published cases. PATIENT(S): Eleven-year-old female with LS. INTERVENTION(S): Multidisciplinary diagnostic evaluation and treatment. MAIN OUTCOME MEASURES: Imaging and laboratory findings, medical and surgical treatment. RESULTS: We describe a case of LS secondary to medically recalcitrant suppurative otitis media in an 11-year-old female and review all eight previously reported cases. The index patient presented after 6 months of otitis media, profound unilateral hearing loss, with symptoms suggesting meningitis. Temporal bone CT demonstrated marked bony destruction of the left otic capsule. Gadolinium-enhanced MRI showed an enhancing process with evidence of meningitis and subdural empyema. The patient was treated with surgical debridement and culture directed antibiotic therapy. Posttreatment imaging showed resolution of intracranial infection with fibrous bony healing of the otic capsule resembling fibrous dysplasia. CONCLUSION: LS is a rare form of labyrinthitis characterized by centrifugal destruction of the otic capsule. The current index case highlights the importance of combined medical and surgical treatment and describes for the first time in the literature the fibrous ossification of the otic capsule following disease resolution.

Cupriavidus pauculus bacteremia in a child on extracorporeal membrane oxygenation.

We report a case of bacteremia secondary to Cupriavidus pauculus in a 15-month-old boy on extracorporeal membrane oxygenation (ECMO). The source of the organism was water in the thermoregulator reservoir. The child responded well to cefepime and ciprofloxacin, a delayed oxygenator change out and replacement of the thermoregulator reservoir with a unit that was cleaned and decontaminated with sodium hypochlorite. Isolation of Cupriavidus pauculus from a patient on ECMO support should raise suspicion of the reservoir as a source.

Desulfovibrio legallii prosthetic shoulder joint infection and review of antimicrobial susceptibility and clinical characteristics of Desulfovibrio infections.

We describe a case of shoulder hemiarthroplasty infection with Desulfovibrio legallii. Antimicrobial susceptibilities of 36 Desulfovibrio isolates are presented. Metronidazole and carbapenems exhibited reliable activity, although piperacillin-tazobactam did not. Eleven previous cases of Desulfovibrio infection are reviewed; most arose from a gastrointestinal tract-related source.

Clostridium septicum brain abscesses in a premature neonate.

Brain abscesses in neonates are typically caused by Gram-negative organisms. There are no previously described cases caused by Clostridium septicum. We present a case of a premature male infant who developed recurrent episodes of suspected necrotizing enterocolitis followed by brain abscesses, cerebritis and ventriculitis caused by C. septicum.

Fatal post-operative Trichoderma longibrachiatum mediastinitis and peritonitis in a paediatric patient with complex congenital cardiac disease on peritoneal dialysis.

Trichoderma longibrachiatum is an emerging pathogen in immunocompromised patients. We report a case of Trichoderma post-operative mediastinitis and peritonitis in a child with complex congenital cardiac disease and functional asplenia. The patient was treated unsuccessfully, initially with caspofungin alone followed by a combination of voriconazole (systemic and topical), caspofungin and intraperitoneal amphotericin B.

Empiric antibiotic therapy for acute osteoarticular infections with suspected methicillin-resistant Staphylococcus aureus or Kingella.

The bacterial agents causing bone and joint infections have been changing. Currently, methicillin-resistant Staphylococcus aureus (MRSA) and Kingella kingae are emerging pathogens. For treatment of MRSA infections, clindamycin, vancomycin, and linezolid are commonly prescribed antibiotics. Kingella are sensitive to most penicillins and cephalosporins. Because MRSA osteoarticular infections tend to be severe, longer periods of antibiotic treatment with more frequent monitoring of inflammatory markers are sometimes required to obtain a complete cure with no residual complications. To assist management, we have included a clinical decision tree with antibiotic treatment protocols.

High-frequency oscillatory ventilation in an infant with cystic fibrosis and bronchiolitis.

Infants with cystic fibrosis (CF) may develop severe respiratory compromise related to viral lower respiratory tract infections due to impaired mucous clearance and plugging of small airways. Consequently air trapping may lead to lung hyperinflation, impaired gas exchange, and respiratory failure. We describe the case of an infant with newly diagnosed CF who developed severe hypercarbic respiratory failure in the setting of viral bronchiolitis successfully treated with high-frequency oscillatory ventilation (HFOV).

Exercise hyperemia and vasoconstrictor responses in humans with cystic fibrosis.

ATP released from circulating erythrocytes is a potential signal regulating muscle blood flow during exercise (exercise hyperemia), and intravascular ATP appears to blunt sympathetic vasoconstriction during exercise. Erythrocytes from patients with cystic fibrosis (CF) do not release ATP. The goal of the present study was to determine whether increases in forearm blood flow during exercise are blunted in CF patients and whether CF patients exhibit greater vasoconstrictor responsiveness during exercise. Nine control subjects and 10 CF patients who were free of other disease complications (approximately 96% O2 saturation) performed incremental rhythmic forearm exercise at 5, 10, and 15% of maximum handgrip strength for 21 min (7 min at each workload). We used a cold pressor test to evoke sympathetic vasoconstriction under resting conditions and at each exercise workload. As a control, subjects performed a second exercise bout without the cold pressor test. Continuous brachial artery blood velocity was monitored beat-to-beat, and vessel diameter was assessed by Doppler ultrasound. Artery diameter, as well as blood pressure, heart rate, and O2 saturation, was measured at steady-state exercise and at 1 min into the cold pressor stimulus. Blood pressure and heart rate responses to the forearm exercise and each cold pressor test were similar in both groups (P > 0.05). Contrary to our hypothesis, forearm blood flow (P = 0.91) and forearm vascular conductance (P = 0.82) were similar at rest and at each level of exercise between CF patients and controls. Additionally, there was no difference in the degree of sympathetic vasoconstriction between groups at rest and at each level of exercise (P = 0.22). Our results suggest that ATP released from the deformation of erythrocytes is not an obligatory signal for exercise hyperemia in human skeletal muscle.