Retrospective review of treatment outcomes and costs in children with sleep disordered breathing assessed with multi-channel studies.

Current UK guidance on OSA management recommends only selective use of sleep studies - when there is diagnostic uncertainty, in children with comorbidities or to evaluate perioperative risk in those with suspected severe OSA. Routine use of sleep studies to confirm a diagnosis of obstructive sleep apnoea (OSA) in children before adenotonsillectomy is not currently recommended. We report the findings of a novel paediatric sleep service based on routine use of multi-channel sleep studies (MCSS) before adenotonsillectomy and present the results of a service evaluation assessing the impact of our practise on treatment outcomes and cost. We conducted a retrospective study of 264 children with sleep disordered breathing seen in our centre between July 2018-June 2019, using medical records and a sleep study database to determine treatment outcomes and costs. Using responses from a questionnaire completed by otolaryngologists for a separate prospective study, we compare our costs with estimates of those associated with a standard UK model of care i.e. with selective use of sleep studies. We estimate that our routine use of MCSS reduced the number of adenotonsillectomies by 44 % but at higher monetary costs than those estimated for the standard model of care. We note however, that reconfiguring our service to arrange a sleep study before the initial appointment, rather than after, would result in the service being cost neutral compared with the standard model. We also estimate that use of home multi-channel studies in our service would bring a significant cost saving (∼£50,000 - £80,000 per annum) compared to standard care.

Prospective evaluation of the impact of multi-channel studies on treatment outcomes in children with sleep disordered breathing.

There are significant variations in practice regarding the use of sleep studies in children with symptoms of sleep disordered breathing (SDB) prior to adenotonsillectomy. Current UK guidance recommends the selective use of sleep studies to confirm a diagnosis of obstructive sleep apnoea (OSA) when there is diagnostic uncertainty, in children with comorbidities, or to assess perioperative risk when severe OSA is suspected. We have developed a novel paediatric sleep service over the past decade based on the routine use of multi-channel sleep studies (MCSS) before adenotonsillectomy. We present the results of a prospective evaluation assessing the impact of our service on treatment outcomes. We conducted a prospective service evaluation of 49 children with SDB seen between July 2021 and August 2022. We used medical records and a sleep study database to determine treatment outcomes. Otolaryngologists completed a questionnaire before each multi-channel sleep study to help evaluate the impact of sleep study findings on surgical decision making. Questionnaire responses before MCSS showed that clinicians thought 66 % of children were 'likely', 'very likely' or 'definitely' would require surgery but only 54 % of children underwent surgery following their sleep study. We estimate that the use of MCSS was associated with a 21 % reduction in children undergoing surgery in this small sample. We conclude that our use of MCSS facilitates conservative management, allowing a significant reduction in the number of children with SDB undergoing surgery, but further validation of MCSS against polysomnography is required.

Hemangiopericytoma of the neck.

Hemangiopericytoma (HPC) is an exceedingly rare tumor of uncertain malignant potential. Approximately 300 cases of HPC have been reported since Stout and Murray described HPCs as "vascular tumors arising from Zimmerman's pericytes" in 1942. After further characterization, the WHO reclassified HPC as a fibroblastic/myofibroblastic tumor. Long term follow up is mandatory because the histologic criteria for prediction of biologic behavior are imprecise. There are reports of recurrence and metastasis many years after radical resection. The head and neck incidence is less than 20%, mostly in adults. We report herein a case of HPC resected from the neck of a 74-year-old woman, who presented in our department with a painless right-sided neck mass. The mass was well circumscribed, mobile and soft during the palpation. The skin over the tumor was intact and normal. Clinical diagnosis at this time was lipoma. A neck computer tomography scan showed a large submucosal mass in the neck, which extended in the muscular sites. The tumor was completely removed by wide surgical resection. During surgery we found a highly vascularised tumor. The histopathologic examination revealed a cellular, highly vascularized tumor. The diagnosis was that of solitary fibrous tumor, cellular variant, with haemangiopericytoma-like features. The patient had normal postoperative course of healing and 24 months later she remains asymptomatic, without signs of recurrence or metastases.

Subcutaneus leiomyosarcoma of the neck: a case report.

INTRODUCTION: Leiomyosarcomas are rare tumors. The most common site for head and neck leiomyosarcomas is the oral cavity, followed by sinonasal tract and skin. Subcutaneous leiomyosarcomas are thought to arise from small to medium-sized blood vessels in the subcutaneous tissue. CASE PRESENTATION: A 67-year-old female patient underwent excision of a slow growing neck mass of the left posterior neck triangle after a thorough clinical and laboratory examination. The lesion was located in the subcutis and fine needle aspiration biopsy revealed malignant features. Histology revealed subcutaneous leiomyosarcoma and the patient is free from local recurrence and distant metastases 3 years after wide excision of the lesion. CONCLUSIONS: The primary modality of therapy of subcutaneous leiomyosarcoma is surgery, adjuvant radiotherapy or chemotherapy may be used for control of local recurrence, in case of positive surgical margins, high-grade or large tumors.

Hereditary angioedema: three cases report, members of the same family.

BACKGROUND: This current clinical case report highlights three cases of Hereditary angioedema (HAE) patients who are all members of the same family (father and his two daughters). The father has C1-INH deficiency, while his daughters have low C1-INH levels: the first possesses only 10% function and the second has low C1-INH level with 0% function. Of note, the second daughter was discovered to have HAE at the age of 2, thus making her the youngest known HAE case report in the English literature. METHODS: Assess the efficacy of administration of C1-INH before dental operation as regards the prevention of HAE episode, when total or partial C1-INH deficiency exist. RESULTS: Acute angioedema leading to laryngeal oedema is a possibly fatal complication for HAE patients undergoing dental procedures. Use of both short-term and long-term HAE prophylaxis prior to dental operations might be life saving for those patients. CONCLUSIONS: Prevention and early recognition of potential laryngeal oedema that can occur as a complication of dental procedures may be lifesaving for HAE patients.

Epidermoid cyst of the floor of the mouth: two case reports.

INTRODUCTION: Epidermoid cysts that appear in the midline floor of the mouth are, usually, a result of entrapped ectodermal tissue of the first and second branchial arches, which fuse during the third and fourth weeks in utero. The incidence in the floor of the mouth of the oral cavity is rare and development sites are the sublingual, submaxillary and submandibular spaces. It was present two cases of epidermoid cyst of the floor of the mouth and discussed the different surgical approaches for this lesion. CASES PRESENTATION: Two cases of midline epidermoid cysts of the floor of the mouth are presented, evaluating the different surgical approaches. The preoperative assessment was made using ultrasonography and computed tomography in both cases. Regarding surgical techniques used, a transcutaneous approach was adopted when the cysts were under the geniohyoid muscle and a midline incision of the oral mucosa along the lingual frenulum was used for sublingual cysts. During the postoperative course, there were no complications, except for mild edema in one case. Follow-up ranged between 5 months and 4 years; no recurrence or malignant changes were observed. CONCLUSIONS: Surgery of epidermoid cyst of the floor of the mouth is the treatment of choice. Access depends on the lesion's location in relation to the mylohyoid or geniohyoid muscles. If the cyst is located over the mylohyoid, surgery is carried out only through the oral cavity, whereas the extraoral incision was necessary only when the cysts were under the geniohyoid muscle.