Journal Article

Sequential development of diffuse panbronchiolitis and myasthenia gravis after thymectomy for thymic neoplasm: a case report

Chou, Chun-Ying, Kuo, Ping-Hung, Hsieh, Min-Shu

2024

Overview

Background Myasthenia gravis (MG) is the most common paraneoplastic disorder associated with thymic neoplasms. MG can develop after thymectomy, and this condition is referred to post-thymectomy myasthenia gravis (PTMG). Diffuse panbronchiolitis (DPB), is a rare form of bronchiolitis and is largely restricted to East Asia, has been reported in association with thymic neoplasms. Only three cases of combined MG and DPB have been reported in the literature. Case presentation A 45-year-old Taiwanese woman presented to our hospital with productive cough, rhinorrhea, anosmia, ear fullness, shortness of breath, and weight loss. She had a history of thymoma, and she underwent thymectomy with adjuvant radiotherapy 7 years ago. Chest computed tomography scan revealed diffuse bronchitis and bronchiolitis. DPB was confirmed after video-assisted thoracoscopic surgery lung biopsy, and repeated sputum cultures grew Pseudomonas aeruginosa . She has been on long-term oral azithromycin therapy thereafter. Intravenous antipseudomonal antibiotics, inhaled amikacin, as well as oral levofloxacin were administered. Three months after DPB diagnosis, she developed ptosis, muscle weakness, and hypercapnia requiring the use of noninvasive positive pressure ventilation. MG was diagnosed based on the acetylcholine receptor antibody and repetitive stimulation test results. Her muscle weakness gradually improved after pyridostigmine and corticosteroid therapies. Oral corticosteroids could be tapered off ten months after the diagnosis of MG. She is currently maintained on azithromycin, pyridostigmine, and inhaled amikacin therapies, with intravenous antibiotics administered occasionally during hospitalizations for respiratory infections. Conclusions To our knowledge, this might be the first case report of sequential development of DPB followed by PTMG. The coexistence of these two disorders poses a therapeutic challenge for balancing infection control for DPB and immunosuppressant therapies for MG.

• Publisher: BioMed Central,BioMed Central Ltd,Springer Nature B.V,BMC
• Subject: Acetylcholine receptors / Amikacin / Anosmia / Anti-Bacterial Agents - therapeutic use / Antibiotics / Azithromycin / Biopsy / Bronchiolitis / Bronchiolitis - etiology / Bronchitis / Bronchopneumonia / Care and treatment / Case Report / Case reports / Coexistence / Complications and side effects / Computed tomography / Corticosteroids / Cough / Critical Care Medicine / Development and progression / Diagnosis / Diffuse panbronchiolitis / Drug resistance / Dyspnea / Etiology / Female / Haemophilus Infections - diagnosis / Haemophilus Infections - etiology / Humans / Hypercapnia / Immunoglobulins / Infections / Intensive / Internal Medicine / Intravenous administration / Levofloxacin / Lung transplants / Lymphocytes / Mechanical ventilation / Medical imaging / Medical prognosis / Medicine / Medicine & Public Health / Methods / Middle Aged / Myasthenia gravis / Myasthenia Gravis - etiology / Neuromuscular junctions / Olfaction disorders / Patients / Pneumology/Respiratory System / Pyridostigmine / Radiation therapy / Respiration / Respiratory tract infection / Sinusitis / Taiwan / Thymectomy / Thymectomy - adverse effects / Thymic neoplasm / Thymoma / Thymoma - surgery / Thymus / Thymus Neoplasms - complications / Thymus Neoplasms - surgery / Tomography, X-Ray Computed / Tumors