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Concept: Case

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BACKGROUND:Case reports indicate that the use of fluoroquinolones may lead to acute kidney injury. We studied the association between the use of oral fluoroquinolones and acute kidney injury, and we examined interaction with renin-angiotensin-system blockers. METHODS:We formed a nested cohort of men aged 40-85 enrolled in the United States IMS LifeLink Health Plan Claims Database between 2001 and 2011. We defined cases as men admitted to hospital for acute kidney injury, and controls were admitted to hospital with a different presenting diagnosis. Using risk-set sampling, we matched 10 controls to each case based on hospital admission, calendar time (within 6 wk), cohort entrance (within 6 wk) and age (within 5 yr). We used conditional logistic regression to assess the rate ratio (RR) for acute kidney injury with current, recent and past use of fluoroquinolones, adjusted by potential confounding variables. We repeated this analysis with amoxicillin and azithromycin as controls. We used a case-time-control design for our secondary analysis. RESULTS:We identified 1292 cases and 12 651 matched controls. Current fluoroquinolone use had a 2.18-fold (95% confidence interval [CI] 1.74-2.73) higher adjusted RR of acute kidney injury compared with no use. There was no association between acute kidney injury and recent (adjusted RR 0.87, 95% CI 0.66-1.16) or past (RR 0.86, 95% CI 0.66-1.12) use. The absolute in crease in acute kidney injury was 6.5 events per 10 000 person-years. We observed 1 additional case per 1529 patients given fluoroquinolones or per 3287 prescriptions dispensed. The dual use of fluoroquinolones and renin- angiotensin-system blockers had an RR of 4.46 (95% CI 2.84-6.99) for acute kidney injury. Our case-time-control analysis confirmed an increased risk of acute kidney injury with fluoroquinolone use (RR 2.16, 95% CI 1.52-3.18). The use of amoxicillin or azithro mycin was not associated with acute kidney injury. INTERPRETATION:We found a small, but significant, increased risk of acute kidney injury among men with the use of oral fluoroquinolones, as well as a significant interaction between the concomitant use of fluoroquinolones and renin- angiotensin-system blockers.

Concepts: Logistic regression, Acute kidney injury, Confounding, Trigraph, Gh, Case, Statistical terminology, Quinolone

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Abstract There has been only one reported case of neuromuscular scoliosis following chronic inflammatory demyelinating polyneuropathy (CIDP). However, no cases of scoliosis that were treated with surgery secondary to CIDP have been previously described. A 16-year-old boy with CIDP was consultant due to the progression of scoliosis with the coronal curve of 86° from T8 to T12. Posterior correction and fusion with segmental pedicle screws were performed under intraoperative spinal cord monitoring with transcranial electric motor-evoked potentials. Although the latency period was prolonged and amplitude was low, the potential remained stable. Coronal curve was corrected from 86° to 34° without neurological complications. We here describe scoliosis associated with CIDP, which was successfully treated with surgery under intraoperative spinal cord monitoring.

Concepts: Medicine, Surgery, Report, Myelin, Case, Potential, Neurological disorders, Chronic inflammatory demyelinating polyneuropathy

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Bochdalek hernia is the most common type of congenital diaphragmatic hernia. It appears frequently in infants but rarely in adults. We present the case of a 50-year-old female han patient with tremendous left-sided congenital posterolateral diaphragmatic hernia (Bochdalek hernia) who also has a pair of supernumerary breasts and pulmonary hypoplasia of the lower-left lobe. The patient had an experience of misdiagnosis and she was treated for bronchitis for one year until being admitted to our hospital. This case study emphasizes the rare presentation of Bochdalek hernia in adults and the necessity of high clinical attention to similar cases.

Concepts: Lung, Patient, Case study, Case, Adult, Congenital diaphragmatic hernia, Diaphragmatic hernia, Bochdalek hernia

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We sought to obtain novel insights into schizophrenia pathogenesis by exploiting the association between the disorder and chromosomal copy number (CNV) burden. We combined data from 5,745 cases and 10,675 controls with other published datasets containing genome-wide CNV data. In this much-enlarged sample of 11,355 cases and 16,416 controls, we show for the first time that case CNVs are enriched for genes involved in GABAergic neurotransmission. Consistent with non-genetic reports of GABAergic deficits in schizophrenia, our findings now show disrupted GABAergic signaling is of direct causal relevance, rather than a secondary effect or due to confounding. Additionally, we independently replicate and greatly extend previous findings of CNV enrichment among genes involved in glutamatergic signaling. Given the strong functional links between the major inhibitory GABAergic and excitatory glutamatergic systems, our findings converge on a broad, coherent set of pathogenic processes, providing firm foundations for studies aimed at dissecting disease mechanisms.

Concepts: Gene, Genetics, Bacteria, Copy number variation, Neuroscience, Case, Major

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Goldenhar syndrome is well known for its classical triad of epibulbar dermoids or lipodermoids, auricular appendages and pretragal fistulas. Here we report a case of Goldenhar syndrome with an unusual association of a fibroepithelial polyp attached to a limbal dermoid. A case of Goldenhar syndrome in a 5-month-old male infant presented with the features of a fibroepithelial polyp attached to a limbal dermoid, right-sided polydactylia with hypoplastic thumb, and accessory preauricular appendages on the left side. The association of a fibroepithelial polyp attached to a limbal dermoid with Goldenhar syndrome is a rare report in the literature. In these cases, regular follow-up with an ophthalmologist is important to monitor the visual development of the patient. Ideally, a multidisciplinary approach is required to manage the other associated anomalies.

Concepts: The Association, Case, Dermoid cyst, Teratoma, Sunshine pop, Hypoplasia

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Abstract This article reports the case of a 22-year-old woman with right renal angiomyolipoma (AML) and inferior vena cava thrombus. Laparoscopic right nephrectomy and thrombectomy were performed. To the authors' knowledge there have been only 46 reported cases of renal AML with endovascular extension and this is the first case to be completely removed by a laparoscopic approach. Laparoscopic management of this kind of tumour is feasible in spite of the vascular involvement. The centre’s experience and enlargement of the tumour are key points for this approach.

Concepts: Kidney, Report, Case, Renal vein, Inferior vena cava, Superior vena cava, Angiomyolipoma, Carly Simon

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INTRODUCTION: Benign paroxysmal positional vertigo (BPPV) is the most common vestibular disorder. However, BPPV in children has been studied less extensively than in the adult population. This is because the observation of benign paroxysmal positional nystagmus (BPPN) in children is technically very difficult and BPPV is rare in children. In this study, we present the only two cases of BPPV in children in which we successfully recorded and analyzed the BPPN. METHODS: One case was an 11-year-old boy and the other was a 3-year-old girl. We analyzed their BPPN three-dimensionally. RESULTS: Apogeotropic positional nystagmus was observed in the first case. We analyzed it to verify the presence of cupulolithiasis in the horizontal semicircular canal (HSCC). Geotropic positional nystagmus was observed in the second case, and the analyzed data indicated the presence of canalolithiasis in HSCC. Over the last decade, we have examined 3341 patients complaining of vertigo or dizziness. Among them, there were 63 children with the same complaint, so that the proportion of cases of BPPV in children was only 3% (2/63). DISCUSSION: Among patients complaining of vertigo or dizziness, children with BPPV are rare (3%). However, we have recorded their BPPN to confirm that BPPV does occur in children and that their characteristics of positional nystagmus are generally identical to those in adults. We emphasize that this is the first report of a child as young as 3 years old being diagnosed with BPPV.

Concepts: Scientific method, Observation, Semicircular canal, Ear, Case, Pathologic nystagmus, Complaint, Benign paroxysmal positional vertigo

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Aberrant regeneration of a third nerve palsy (oculomotor synkinesis) excludes an ischaemic cause and in the absence of relevant trauma strongly suggests a compressive aetiology. A scan is mandatory in such cases. We describe the case of a 52-year-old woman who presented with complete pupil-involving third nerve palsy from a posterior communicating artery aneurysm, who later developed widespread aberrant regeneration of pupil, eyelid and third nerve territory rectus muscles.

Concepts: Muscle, Posterior communicating artery, Cerebral aneurysm, Case, Oculomotor nerve, In the Absence of Truth, Oculomotor nerve palsy

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Pyoderma gangrenosum (PG) is an ulcerative skin disorder characterized by neutrophilic infiltrations. PG is generally classified into four types: (i) ulcerative; (ii) pustular; (iii) bullous; and (iv) vegetative. Among them, bullous PG is known as a rare type. Herein, we report a case of bullous PG together with a summary of the 12 PG cases treated in our department over the previous 15 years, and we review 38 well-documented bullous PG cases (65.8% female; aged 18-80 years [mean ± standard deviation, 51.6 ± 16.8]) in the published work, including the present case, from 1972-2011. Although the disease most frequently associated with PG is inflammatory bowel disease, bullous PG is most commonly associated with hematological disorders (25/38, 65.8%), which indicates the characteristic pathophysiology specific to bullous PG.

Concepts: Immune system, Inflammation, Ulcerative colitis, Crohn's disease, Gastroenterology, Inflammatory bowel disease, Case, Pyoderma gangrenosum

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Background: Concha bullosa is the pneumatisation of intranasal conchae (usually the middle turbinate, and rarely the inferior or superior turbinate); however, the term is generally used to describe aeration of the middle concha. Superior concha bullosa is a rare finding, and only a few cases of inferior concha bullosa have been reported in the medical literature. When symptomatic, concha bullosa may cause various problems including nasal congestion, headache, postnasal drip, anosmia and, sometimes, epiphora. Methodology: Computed tomography, following history-taking and physical examination, is a valuable tool in diagnosing turbinate pneumatisation. This article presents a very rare case with bilateral triple conchae pneumatisations. Results: The symptomatology, diagnosis and treatment options for cases of multiple concha bullosa are discussed. The surgical interventions performed in the presented case are briefly described. Conclusion: The presented patient had pneumatisation of all six turbinates. In such cases, we propose that this condition be termed ‘conchae bullosis’ rather than ‘conchae bullosa’, in a similar fashion to the use of nasal polyposis as the plural form of nasal polyp.

Concepts: Sinusitis, Medical imaging, English language, Case, Physical examination, Medical history, Concha bullosa, Grammatical number