Severe laryngeal sarcoidosis in a child managed by intralesional steroid, debulking, and methotrexate

IF 0.3 Q4 OTORHINOLARYNGOLOGY Acta Oto-Laryngologica Case Reports Pub Date : 2022-12-10 DOI:10.1080/23772484.2022.2153052
Jesper Stensig Aa, P. Toftedal, Joyce H Schultz, S. Fast
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引用次数: 1

Abstract

Abstract Laryngeal sarcoidosis is a rare condition, particularly in the pediatric population. Acute airway intervention may be required in severe cases, but guidelines to support the therapeutic strategy are absent. Moreover, evidence regarding systemic therapy is scarce. This encouraged us to report the result of airway management and systemic methotrexate at a one-year follow-up in a child with severe laryngeal sarcoidosis. A 14-year-old Caucasian female presented with dyspnea, dysphagia, and hoarseness. Fiberoptic laryngoscopy revealed profound swelling of the epiglottis and the arytenoid regions. Laryngeal sarcoidosis was suspected, and direct laryngoscopy, including biopsies, cold steel debulking, and intralesional corticosteroid injections, was performed to secure the airway. Histology was compatible with sarcoidosis, and long-term therapy with methotrexate was prescribed. At one-year follow-up, the patient was asymptomatic, and fiberoptic laryngoscopy demonstrated a near complete remission.
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儿童严重喉部结节病经局灶内类固醇、消肿和甲氨蝶呤治疗
喉结节病是一种罕见的疾病,特别是在儿童人群中。严重病例可能需要急性气道干预,但缺乏支持治疗策略的指南。此外,关于全身治疗的证据很少。这鼓励我们报告一名患有严重喉结节病的儿童在一年随访中气道管理和全身甲氨蝶呤的结果。一位14岁的白人女性表现为呼吸困难、吞咽困难和声音嘶哑。纤维喉镜检查显示会厌和杓状区严重肿胀。怀疑喉部结节病,进行直接喉镜检查,包括活检、冷钢减压和病灶内皮质类固醇注射,以确保气道安全。组织学符合结节病,并给予甲氨蝶呤长期治疗。在一年的随访中,患者无症状,纤维喉镜检查显示几乎完全缓解。
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审稿时长
29 weeks
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