Rationale: Multiple pulmonary leiomyomatous hamartoma (MPLH) is an extremely rare benign disease that mostly occurs in women of reproductive age.
Patient concerns: A 32-year-old female patient recently diagnosed with multiple bilateral pulmonary nodules. She has the symptoms of dry cough, chest tightness, dyspnea on exertion. Chest X-ray identified multiple bilateral pulmonary nodules in the lung, and the diameter of the largest nodule was about 3.1 cm.
Diagnoses: Pathology confirmed the diagnosis of MPLH based on morphology and immunohistochemical staining.
Interventions: The patient presented with multiple well-defined nodular shadows in chest computed tomography (CT), atypical image and symptoms were detected. Positron emission tomography/CT scan showed mild fluorine-18 fluorodeoxyglucose uptake in the lesions and no abnormal foci in any other parts of her body. She subsequently underwent a video-assisted thoracoscopic surgery with wedge resection of the biggest one of the nodules. Then the patient given symptomatic treatment, without hormone, no further treatment was prescribed.
Outcomes: The patient is in the good general condition and without obvious pulmonary symptoms after the follow-up of 1 year, chest CT scan showed no significant changes in the sizes and locations of her bilateral pulmonary nodules.
Lessons: Due to its rare presentation, the primary MPLH may be undiagnosed. Awareness of main morphologic and immunohistochemical features of MPLH is critical for the recognition of this uncommon disease.