Thoracoscopic resection of esophageal duplication causing tracheobronchomalacia in a 5-year-old patient.
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ABSTRACT:
SUBMITTER: Alushani D
PROVIDER: S-EPMC8304868 | biostudies-literature |
REPOSITORIES: biostudies-literature
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Project description:BACKGROUND:It is presumed that tracheobronchomalacia in adults is caused by airway pressure-induced injury due to chronic cough related to pulmonary emphysema or chronic bronchitis. Commonly, a posterolateral approach using stabilizing materials is the surgical technique of choice for treating tracheobronchomalacia. We report a case in which thoracoscopic plication of the membranous portion was performed instead of airway stent placement for tracheobronchomalacia in an elderly individual. CASE PRESENTATION:An 87-year-old man who had been treated for bronchial asthma, pulmonary emphysema, and tracheobronchomalacia was admitted to our hospital with acute exacerbation of dyspnea. The patient underwent tracheal intubation, which was followed by tracheostomy 16?days later. Insertion of the tip of the adjustable-length tracheostomy tube to the end of the stenotic lesion enabled him to breathe spontaneously. However, conservative management failed due to recurrent pneumonia caused by the tracheobronchomalacia. Crescent-type tracheobronchomalacia (Johnson's classification grade III) was diagnosed, and the main narrowed area of the trachea was assumed to be approximately 3-10?cm from the tracheal bifurcation. A thoracoscopic approach was selected because a posterolateral approach was considered too invasive considering the patient's age and general condition. We placed eight stitches on the tracheal membranous portion and four stitches on the membranous portion of the right main bronchus, using the horizontal mattress suture technique. The use of foreign materials was avoided because meropenem-resistant Pseudomonas aeruginosa was cultured in a tracheal specimen. Immediately after the operation, the expiratory airway stenosis improved, and subsequently, spontaneous ventilation was possible using a normal type of tracheostomy tube instead of an adjustable-length tracheostomy tube. CONCLUSIONS:Tracheobronchomalacia is not a rare condition in patients with chronic obstructive pulmonary disease. The thoracoscopic approach is less invasive than the posterolateral approach and is suitable in cases that are otherwise refractory to medical treatment. We believe that thoracoscopy may be a useful treatment option in cases where conservative treatment is not appropriate.
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Project description: Not available
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Project description:We present a clinical report of an 11-year-old male patient with an interstitial duplication of 19p13.3 (829?kb in size) at genomic coordinates 3,804,495-4,033,722?bp (hg19) identified by chromosomal microarray analysis and review the literature from nine published reports adding knowledge regarding this chromosomal anomaly and clinical outcomes. The size of the duplication ranged from 0.83 to 8.9 Mb in the nine individuals. The young boy in our report was dysmorphic with microcephaly, abnormal craniofacial features, intellectual disability, aggression, and a heart murmur. All patients were found to have a psychomotor developmental delay and/or intellectual disability with the majority having microcephaly, intrauterine growth retardation, and hypotonia. Common craniofacial findings included a tall, prominent forehead, an elongated face, epicanthal folds, hypertelorism, prominent low-set ears, philtrum anomaly, and a small mouth. Other less common features included abnormal digits, sparse hair, and cardiac defects. Clinical features, chromosome duplication sizes, locations, and the number of genes will be summarized in a tabular form.
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Project description:We report a 12-year-old boy referred to the Clinical Genetics service in view of facial dysmorphism, learning difficulties and autistic spectrum disorder. 60K arrayCGH revealed an 8.2-Mb duplication on chromosome 13q31.3q32.3, which was paternally inherited. This specific duplication on chromosome 13 has not been previously reported in the medical literature, and there are no familial or de novo patients with the same duplication breakpoints. This region contains 24 OMIM genes, including the glypicans GPC5 and GPC6, and the ZIC2 gene. We discuss the relevance of this chromosome imbalance and discuss the impact of this duplication on our patient's phenotype. Given that the duplication on 13q was paternally inherited, and although initially thought to be of uncertain significance, on exploring the family history further, it became apparent that the father had learning difficulties as a child and previous surgery for congenital diaphragmatic hernia. Here we explore the phenotype in association with this novel duplication on chromosome 13q and add to the existing literature on array findings within this region.
| S-EPMC4188164 | biostudies-literature
Project description:BackgroundIntradural, extramedullary capillary hemangiomas of the cauda equina are exceedingly rare malformations arising from the endothelial cells of the nervous system vasculature. Roughly 20 cases have been reported in the literature, with the youngest and only pediatric case being in a 17-year-old patient. We report the youngest case of intradural extramedullary capillary hemangioma of the cauda equina in a 14-year-old patient.Case descriptionA 14-year-old female presented with two-month history of low back pain with bilateral leg pain and numbness. Magnetic resonance imaging (MRI) revealed an L2 well-defined homogenous contrast-enhancing intradural, extramedullary mass causing severe spinal canal stenosis. Patient underwent laminoplasty for resection of an intradural tumor. Intraoperative hemostasis was readily achieved via early identification and coagulation of the predominate feeding vessel. Postoperatively, the patient awoke with no deficits and resolved leg pain. A 3-month postoperative MRI revealed no tumor recurrence and fully healed lamina.ConclusionsGiven the benign nature, the operative goal is complete excision of the tumor without damage to surrounding neural structures. Postoperatively the goal is relief of pain and improvement in neurologic function. To our knowledge we report the first case in which laminoplasty is utilized for the treatment of this pathology in a pediatric patient. Evidence for laminoplasty in this patient population is sparse and future studies are still needed. In any case, reconstruction of the surgical site in a manner that returns the patient's normal anatomy should be strongly considered especially in younger patients.
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Project description:A Fabry patient with poikilothermia is described. Laboratory investigations, neuro-imaging and autonomic function tests did not disclose a cause. Assessment of intra-epidermal nerve fibre density and quantitative sensory testing revealed small fibre neuropathy with a highly impaired cold sensation. We speculate that the poikilothermia is either caused by a vascular lesion in the hypothalamus not visible on MRI or by small fibre neuropathy leading to disturbed body temperature perception and therefore impaired thermoregulation.
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Project description:BackgroundSince the conception of enhanced recovery after surgery protocols, tubeless strategies have become popular. Herein, we introduce a previously unreported alternative air-extraction strategy for patients who have undergone thoracoscopic wedge resection and explore its feasibility and safety.MethodsBetween January 2015 and June 2017, 264 consecutive patients underwent thoracoscopic wedge resection with different drainage strategies. Patients were divided according to the postoperative drainage strategies used: routine chest tube drainage (RT group), complete omission of chest tube drainage (OT group), and prophylactic air-extraction catheter insertion procedure (PC group). Using the propensity score matching method, clinical parameters and objective operative qualities were compared among the three groups.ResultsOptimal 1:1 matching was used to form pairs of RT (n =36) and PC (n =36) groups and balance baseline characteristics among the three groups. The incidence rates of pneumothorax were 5.6% (2/36), 9.8% (5/51), and 19.4% (7/36) in the RT, OT, and PC groups, respectively (P = 0.07). Chest tube reinsertion incidence for postoperative pneumothorax was 19.4% (1/7) in the PC group and 60% (3/5) in the OT group. Other postoperative complications were comparable among these groups.ConclusionsThe prophylactic air-extraction strategy may be an alternative procedure for selected patients. Remedial air extraction may reduce the occurrence of chest tube reinsertion for pneumothorax patients, but further investigation is required.
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Project description:We report a highly unusual case of a 22-year-old woman with single-ventricle anatomy and a history of Fontan palliation who developed takotsubo cardiomyopathy. To our knowledge, takotsubo cardiomyopathy has not previously been described in a patient with single-ventricle anatomy, or more generally in any patient with congenital heart disease. The patient presented at our clinic for urgent examination after a 2-day history of chest pain that had begun upon the death of her dog. Invasive evaluation was refused by the patient; instead, she underwent electrocardiogram-gated coronary computed tomographic angiography, to rule out obstructive coronary disease. A physician who suspects takotsubo cardiomyopathy in a patient should look for new-onset chest pain and dyspnea coupled with these features: immediate onset of segmental akinesia in the midventricular or apical aspects of the heart, elevation of brain natriuretic peptide level 12 to 24 hours after onset of akinesia, elevation of troponin level 24 to 48 hours after onset, and disappearance of the segmental akinesia a few days after onset.
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Project description: Not available
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Project description:The choice of prosthetic heart valve type is largely dependent upon patient's age at implantation and on what, in his eyes, seems more pertinent: avoidance of complications associated with anticoagulation of mechanical valves or structural valve deterioration of bioprosthetic valves. Long lasting and new promising concepts such as transcatheter aortic valve implantation are promoting the use of bioprosthesis even in younger patients. However, it is up to the individual patient to decide.
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