As previously mentioned, the cystic presentation of benign metastizing leiomyomas is rare, prompting rare cystic entities to be considered in the differential diagnosis such as LAM, Langerhans cell histiocytosis, and cystic nodular amyloidosis. The immunophenotype for benign metastasizing leiomyoma consists of spindle cells which are positive for desmin, smooth muscle actin, ER, PR, CD10, and bcl-2. The spindle cells are negative for S100 protein, CD117, CD34, CD99, HMB45, Melan-A, and α-inhibin. This immunoprofile matched our case. In the few reported pulmonary cystic examples of metastasizing leiomyoma, the immunophenotype was similar with immunoreactivity for desmin, smooth muscle actin, ER, PR, CD10, and bcl-2. The Ki-6711 nuclear proliferation index is typically low.