Subglottic ectopic thymus mimicking airway haemangioma

Subglottic haemangioma presents as progressive obstruction in the neonatal and infantile airway, with a soft lesion seen during endoscopy. Diagnosis is based on macroscopic findings, biopsy is not usually performed and propranolol is first-line treatment. In contrast, ectopic thymus is a rare differential diagnosis for subglottic mass made by histopathological examination after excision or autopsy. In this article, we present a case of an infant with a subglottic lesion with endoscopic features consistent with haemangioma. After initial clinical response to propranolol, the patient represented with progressive stridor no longer responding to therapy. Open excision of the lesion was performed, and histopathology revealed ectopic thymus tissue. In this case, ectopic thymus tissue mimicked the presentation of subglottic haemangioma, and confirmation bias persisted due to an apparent initial clinical response to treatment with propranolol. In cases of subglottic mass refractory to medical treatment, excision of the lesion should be considered.

Treatment failure with propranolol for subglottic haemangioma

The use of propranolol for the treatment of subglottic haemangioma has become hugely popular due to its effectiveness and safety profile. We report a case of 7-month-old boy who presented with stridor and histopathology suggestive of subglottic haemangioma following microlaryngoscopy and bronchoscopy (MLB). However, he did not respond to propranolol treatment. This could be due to an older age of propranolol commencement. In general, early commencement of propranolol is necessary when diagnosis of symptomatic infantile haemangioma is made to achieve maximal improvement in symptoms and prevent further proliferation. There should be a high index of suspicion for subglottic haemangioma in children presenting with chronic biphasic stridor, with early MLB and diagnosis. This will allow early treatment, giving the best chance to avoid our situation.

A case report of subglottic haemangioma

<p class="abstract">We report a case of 18 year old female who presented with hoarseness of voice and hemoptysis for 6 months eventually diagnosed as subglottic haemangioma through video laryngoscopy, CT imaging and histopathological examination of excision biopsy. The tumor excision was done by endolaryngeal surgery with tracheostomy under general anesthesia. The diagnosis of subglottic haemangioma, its management and prognosis were discussed.</p><p class="abstract"> </p>

Great Ormond Street Hospital treatment guidelines for use of propranolol in infantile isolated subglottic haemangioma

Objective:Treatment options for large subglottic haemangioma include steroids, laser ablation, open excision, tracheostomy and, more recently, propranolol. This article aims to present the Great Ormond Street Hospital guidelines for using propranolol to treat infantile isolated subglottic haemangioma by ENT surgeons.Methods:The vascular malformations multidisciplinary team at Great Ormond Street Hospital has developed guidelines for treating infantile haemangioma with propranolol.Results:The Great Ormond Street Hospital guidelines for propranolol treatment for infantile subglottic haemangioma include investigation, treatment and follow up. Propranolol is started at 1 mg/kg/day divided into three doses, increasing to 2 mg/kg/day one week later. On starting propranolol and when increasing the dose, the pulse rate and blood pressure must be checked every 30 minutes for the first 2 hours. Lesion response to treatment is assessed via serial endoscopy.Conclusion:Recent reports of dramatic responses to oral propranolol in children with haemangioma and acute airway obstruction have led to increased use. We advocate caution, and have developed guidelines (including pre-treatment investigation and monitoring) to improve treatment safety. Propranolol may in time prove to be the best medical treatment for subglottic haemangioma, but at present is considered to be still under evaluation.

Failure of propranolol in the treatment of childhood haemangiomas of the head and neck

Background:Infantile haemangiomas enter a rapid proliferative phase within months of birth, before slowly involuting. Those with the potential for disfigurement or morbidity require intervention. Propranolol has emerged as an effective new treatment modality, with the potential to become the first-line treatment of choice.Methods:Four children with haemangiomas of the head and neck were treated with propranolol at a tertiary referral centre. The size of the haemangioma and the symptoms resulting from airway compromise were monitored.Results:Three of the four children showed a dramatic response to treatment with propranolol. However, one child responded initially but was readmitted with stridor secondary to new haemangioma proliferation.Conclusions:We report a cautionary case in which a subglottic haemangioma developed contemporaneously with propranolol treatment, requiring surgical intervention. This finding highlights the need for regular follow up of treatment response, and the need for monitoring for treatment side effects.