Πέμπτη 17 Ιουνίου 2021

Complex Airway Management in Patients with Tracheal Cartilaginous Sleeves

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Objectives/Hypothesis

A tracheal cartilaginous sleeve (TCS) is a rare anomaly characterized by anterior fusion of tracheal cartilages. TCS is associated with syndromic craniosynostoses including Apert, Crouzon and Pfeiffer syndromes and FGFR2, FGFR3, and TWIST1 variants. This study presents a 30-year review of patients with syndromic craniosynostosis and TCS and describes diagnostic methods, genetic variants, surgical interventions, and long-term outcomes.

Study Design

Retrospective, single-institution review.

Methods

This review included patients with syndromic craniosynostosis and TCS treated at Seattle Children's Hospital from 1990 to 2020. Tracheostomy, genetic variants, and additional surgery were primary measures. Fisher's exact test compared need for tracheostomy in patients with proposed high-risk (FGFR2 p.W290 or FGFR2 p.C342) versus low-risk genetic variants.

Results

Thirty patients with TCS were identified. Average age at diagnosis was 12 months (range 2-weeks to 7.9-years; standard deviation 19.8 months). Syndromes included Pfeiffer (37%), Apert (37%), and Crouzon (26%). Severe obstructive sleep apnea was present in 76% of patients. Tracheostomy was performed in 17 patients (57%); five were successfully decannulated. Additional interventions included adenotonsillectomy (57%), nasal (20%), laryngeal (17%), and craniofacial skeletal surgery (87%). All patients with Pfeiffer syndrome and FGFR2 p.W290C variants and 83% of patients with FGFR2 p.C342 variants required tracheostomy, differing from other variants (P = .02, odds ratio 33, 95% confidence interval 1.56–697.96). One patient (3%) died.

Conclusion

TCS contributes to multilevel airway obstruction in patients with syndromic craniosynostosis. Genetic testing in patients with FGFR2-related syndromic craniosynostoses may identify those at risk of TCS and facilitate early intervention. A better understanding of this patient population may foster individualized airway management strategies and improve outcomes.

Level of Evidence

4 Laryngoscope, 2021

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Speech Performance among Healthy Malay Female Speakers during Dual-Task and Sentence Complexity

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Mdivi‐1 alleviates atopic dermatitis through the inhibition of NLRP3 inflammasome

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Abstract

Atopic dermatitis (AD) is chronic inflammatory cutaneous disorder with few treatment options. Dynamin related protein 1 (Drp1)-dependent mitochondrial fission contributes to the activation of NLRP3 inflammasome, and inhibiting Drp1 has been become an attractive therapeutic strategy for inflammatory diseases. This study aimed to investigate the effect of Drp1 inhibitor mdivi-1 on experimental AD. We firstly detected the effects of mdivi-1 on primary human keratinocytes in an inflammatory cocktail-induced AD-related inflammation in vitro. Results showed that mdivi-1 inhibited NLRP3 inflammasome activation and pyroptosis which were evidenced by decreased expression of NLRP3, ASC, cleavage of caspase-1, GSDMD-NT, mature interleukin (IL)-1β and IL-18 in keratinocytes under AD-like inflammation. Next, mouse model of AD-like skin lesions was induced by epicutaneous application of 2,4-dinitrochlorobenzene (DNCB) and mdivi-1 (25 mg/kg/day, days 5-33 during construction of AD model) was intraperitoneally injected into DNCB-induced mice. AD mice with mdivi-1 treatment exhibited ameliorated AD symptoms, lower serum IgE level, and reduced epidermal thickening, mast cells infiltration, and production of IL-4, IL-5 and IL-13 in the lesional tissues. Indeed, mdivi-1 significantly inhibited NLRP3 inflammasome activation and pyroptotic injury occurred in DNCB-treated skin tissues. Mechanically, mdivi-1 regulated the expression of mitochondrial dynamic proteins and suppressed the activation of NF-κB signal pathway which is an upstream of NLRP3 inflammasome both in vitro and in vivo. This study demonstrated that mdivi-1 could protect against experimental AD through inhibiting the activation of NLRP3 inflammasome and subsequent inflammatory cytokine release, and mdivi-1 might exert this function by inhibiting mitochondrial fission and subsequently blocking NF-κB pathway.

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Nasal Septal Angiofibroma

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Ear Nose Throat J. 2021 Jun 16:1455613211026517. doi: 10.1177/01455613211026517. Online ahead of print.

ABSTRACT

Juvenile nasopharyngeal angiofibroma is a benign vascular tumor seen predominantly in adolescent males in the second decade of life. Extranasopharyngeal angiofibroma includes vascular fibrous masses that occur outside the nasopharynx. The diagnosis of an angiofibroma is based on the clinical presentation and imaging, with biopsies being avoided to avoid excessive bleeding. Computed tomography scan is considered sufficient for the diagnosis of extranasopharyngeal angiofibroma as it clearly delineates and identifies the tumor.

PMID:34134540 | DOI:10.1177/01455613211026517

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Malignant Peripheral Nerve Sheath Tumor in the Nasal Cavity of a Neonate: A Case Report

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Ear Nose Throat J. 2021 Jun 16:1455613211014103. doi: 10.1177/01455613211014103. Online ahead of print.

ABSTRACT

Malignant peripheral nerve sheath tumor (MPNST) is a rare tumor that can develop on the lining of nerves and within the network of nerve fibers in different organs, and it is commonly found in the head and neck, limbs, and trunk. These tumors can occur in patients of any age. They most commonly occur in adults aged 20 to 50 years; however, fewer cases of this tumo r in children have been reported. To date, no neonatal case of MPNST in the nasal cavity has been reported. Here, we report the case of a 4-day-old female newborn who presented with a nasal mass that re-enlarged after surgery and was diagnosed as MPNST of the nasal cavity on the basis of pathological results. This is the first report of MPNST in the nasal cavity of a neonate. Differential diagnosis and treatment of nasal masses have been proposed in the related literature.

PMID:34134529 | DOI:10.1177/01455613211014103

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Sweat Gland Carcinoma of the Head and Neck: Case Report and Literature Review

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Ear Nose Throat J. 2021 Jun 16:1455613211016717. doi: 10.1177/01455613211016717. Online ahead of print.

ABSTRACT

INTRODUCTION: The main aim of this article is to discuss and summarize the research advancements and the treatment methods for sweat gland carcinoma (SGC) based on 2 cases of SGC in our hospital and the related literature.

CASE REPORT: This article presents 2 patients with SGC who were treated in the China Medical University, Liaoning Provincial Key Laborato ry of Oral Diseases from 2007 to 2019. We analyzed the clinical features, therapies, and prognosis of the patients and searched for related literatures.

DISCUSSION: Two patients underwent extended resection for local lesions with no adjuvant radiotherapy. Neither local recurrence nor distant metastasis was detected during follow-up. Reviewing previous literature, the treatment of SGC includes surgical resection, radiotherapy, and chemotherapy. We have not found an effective treatment. The prognosis of SGC occurred in head and neck is relatively good compared with another primary-site location, primary surgical excision with safe resection margins and neck dissection is recommended.

PMID:34134535 | DOI:10.1177/01455613211016717

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Functional Olfactory Neuroblastoma Inducing Symptomatic SIADH

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Ear Nose Throat J. 2021 Jun 16:1455613211022102. doi: 10.1177/01455613211022102. Online ahead of print.

ABSTRACT

Olfactory neuroblastoma (ONB) is a rare malignancy of the sinonasal cavity, originating from neuroepithelial olfactory cells. Olfactory neuroblastoma can be difficult to diagnose due to its anatomic position and variable symptomatic presentation, leading to diagnosis at a more advanced stage. Here, we present the case of a 35-year-old man with no previous medical history who had a bicycle accident secondary to syncope. He was found to be hyponatremic and suspected to have syndrome of inappropriate antidiuretic hormone secretion (SIADH). In the workup of SIADH, a brain magnetic resonance imaging revealed a mass in the left middle meatus. The lesion secreted inappropriate amounts of ADH, resulting in symptomatic paraneoplastic SIADH. This ultimately led to the early recognition and successful resection of this rare ONB. The patient has remained disease-free for over 5 years. This case is a fortunate example of a functional malignancy of the sinonasal tract that was discovered early and successfully treated as a result of symptomatic SIADH.

PMID:34134537 | DOI:10.1177/01455613211022102

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A Retrospective Review of 589 Percutaneous Tracheostomies in a Canadian Community Teaching Hospital

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Ear Nose Throat J. 2021 Jun 16:1455613211025744. doi: 10.1177/01455613211025744. Online ahead of print.

ABSTRACT

OBJECTIVE: The primary objective of this study was to review the complication rate of percutaneous tracheostomies performed by a single surgeon in a community teaching hospital.

METHODS: This retrospective study reviewed the patients who underwent percutaneous tracheostomy with bronchoscopic guidance in a community hospital setting between 2009 and 2017. Pat ients older than the age of 18 requiring percutaneous tracheostomy were chosen for this retrospective study. Patients who were medically unstable, had no palpable neck landmarks, and inadequate neck extension were excluded. Indications for percutaneous tracheostomy included patients who had failed to wean from mechanical ventilation, required pulmonary toileting, or in whom airway protection was required.

RESULTS: Of the 600 patients who received percutaneous tracheostomy, 589 patients were included in the study. Intraoperative complication (2.6%) and postoperative complication rates (11.4%) compared similarly to literature reported rates. The most common intraoperative complications were bleeding, technical difficulties, and accidental extubation. Bleeding, tube obstruction, and infection were the most common postoperative complications. Overall burden of comorbidity, defined by Charlson Comorbidity Index, and coagulopathy were also found to be associated with higher complica tion rates. The decannulation rate at discharge was 46.3%.

CONCLUSION: Percutaneous tracheostomy is a safe alternative to open tracheostomies in the community setting for appropriately selected patients.

PMID:34134536 | DOI:10.1177/01455613211025744

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Advances in the Diagnosis and Treatment of Primary Ciliary Dyskinesia

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This review discusses the diagnostic testing for primary ciliary dyskinesia and its potential novel molecular therapeutic targets, with a focus on the otolaryngological manifestations of the disease.
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Early Intervention for the Treatment of Acute Laryngeal Injury After Intubation—Excellent Outcomes With Low Risk?—Reply

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In Reply Aretha et al offer support for our recent observations detailing the importance of early intervention for the treatment of acute laryngeal injury after prolonged mechanical ventilation. In addition, the authors raise several good points that will shape future questions surrounding laryngeal functional recovery after critical illness. Their letter also highlights the inherent limitations involved in a retrospective cohort study. These limitations are salient, as evidenced by their mention and thorough discussion in our initial manuscript.
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Early Intervention for the Treatment of Acute Laryngeal Injury After Intubation

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To the Editor We read with great interest the recently published article by Lowery AS et al in JAMA Otolaryngology–Head & Neck Surgery, where it was shown that early intervention for patients with postintubation laryngeal injury was associated with a decreased duration of tracheostomy dependence, a higher rate of decannulation, and fewer surgical procedures compared with late intervention. Patients who underwent early intervention also avoided open reconstruction. We appreciate the important information presented in this study and we wish to comment on some associated issues.
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Nasal Mass in a Middle-aged Woman With Multiple Myeloma and Recurrent Fungal Sinusitis

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A woman in her 50s with multiple myeloma and persistent sinus mucormycosis is admitted with new left-sided nasal congestion, facial pain, and cheek numbness, and a nasal mass is found. What is your diagnosis?
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