Pulmonary benign metastasizing leiomyoma presented as acute respiratory distress

Abstract

Benign metastasizing leiomyoma (BML) is a very rare condition and is characterized by the presence of benign smooth muscle tumours in organs distant from the uterus, most commonly the lung. It generally affects women of reproductive age and prognostic is usually excellent. However, the course of the disease is unpredictable. We report here the case of a 76‐year‐old woman with a previous medical history of uterine benign leiomyomas in whom BML was acutely revealed by a respiratory distress due to voluminous pulmonary and pleural leiomyomas requiring surgical extraction. Clinical evolution was remarkable by resistance to medical treatment and development of rare bone localization. BML is a contradictory entity characterized by benign histological features but with metastatic potential. Pulmonologists as well as oncologists in charge of patients with multiple pulmonary nodules and a history of uterine leiomyoma should be aware of this potential diagnosis in order to implement appropriate diagnostic procedures for this benign tumour.

Benign metastasising leiomyoma presenting with respiratory distress

“…In previous case reports which include imaging, patients may have diffuse, small, miliary lesions with no significant change in lung function,5 6 or a low-moderate number of moderate sized lesions 7–13. There are cases where a single, large lesion can be blamed for the patients’ symptoms8 14 15 and surgical resection can prove curative. However, the combination of widespread, moderate sized lesions in all lobes is believed to be unique to our case and makes surgical cure impossible.…”

“…Tamoxifen,6 20 ulipristal,21 GnRH agonist with antiprogesterone22 and GnRH analogue monotherapy (our case) are all used. Aromatase inhibitors15 or no therapy10 11 16 may be used in postmenopausal women.…”

Pulmonary metastasectomy in uterine malignancy: outcomes and prognostic factors

“…Lesions are usually slow-growing, although symptoms such as cough and chest pain are not rarely reported (28,29). At least one case of death from respiratory failure due to massive hilar metastases and a case of potentially fatal haemoptysis have been described (30,31).…”

Benign Metastasizing Leiomyoma of the Uterus: Rare Manifestation of a Frequent Pathology

“…Uterine leiomyoma is the most common gynecological tumor [ 1 – 4 ]. BML is a rare variant [ 1 – 13 ] characterized by multiple leiomyomatous lesions in distant locations, most commonly the lungs [ 3 – 11 ]. Less frequently involved areas are lymph nodes, inferior vena cava, heart, brain, bones, abdomen, retroperitoneum, pelvis, breast, esophagus, liver, appendix, trachea, skin, muscle, and parametria [ 14 , 15 ].…”

“…The antagonistic terminology of BML reflects the coexistence of benign appearance with a biological behavior suggesting malignancy [ 2 , 3 , 8 , 16 ]. When multiple pulmonary nodules are incidentally detected in women with history of surgery for uterine leiomyoma, clinicians should be aware of this potential diagnosis [ 5 , 11 , 13 ].…”