Incidental lymphangioleiomyomatosis in pelvic lymph nodes associated with Malignant neoplasm of the ovary – two case reports

To the Editor,
Lymphangioleiomyomatosis (LAM), a rare, destructive and progressive neoplastic disease, generally arises in the lung and occurs predominantly in women of childbearing age or premenopausal age [1]. Primary extrapulmonary LAM is extremely rare, with only a handful of cases being reported, leading to limited information being available regarding the pathologic characteristics. Here we report two cases of LAM of the pelvic lymph node, accidentally discovered after surgical staging of ovarian malignancy. This report shows that occult LAM can be detected in surgical staging of pelvic tumors. The diagnosis should be based on clinicopathological features and immunohistochemical examination, to avoid a missed diagnosis or misdiagnosis.
Written informed consent was obtained from the patient described in this letter, and the investigation was conducted in accordance with the Declaration of Helsinki (1975). The ethics committees of the Affiliated Hospital of Southwest Medical University approved this study.
The first case was of a 55-year-old postmenopausal woman admitted to the hospital with abnormal uterine bleeding for more than 2 months. Doppler ultrasound suggested mixed cystic-solid echogenicity in the posterior uterus with a size of 10  9.5  7.5 cm, leading to a differential diagnosis of malignant tumor with pelvic lymph node metastasis. The second case was of a 54-year-old woman, who presented with a mass in her right ovary. The patient underwent an oophorectomy ten years ago, for clear cell carcinoma of the left ovary. After reviewing the abdominal computed tomography, a space-occupying lesion was seen in the right ovary, but no further treatment was given at the time and the tumor gradually enlarged. Total hysterectomy, bilateral adnexectomy and lymph node dissection were performed in both patients. Their past and family histories were unremarkable.
Final pathological examination revealed that the right ovarian lesion of the first patient was consistent with high-grade serous carcinoma, and the right ovarian lesion of the second patient was consistent with clear cell carcinoma. No lymph node metastases were found in either of the patients, although multifocal spindle cell proliferation was an unexpected finding. A total of 91 lymph nodes were dissected in the first patient, out of which 18 showed spindle cell proliferation, including left pelvic lymph node (3/32), right pelvic lymph node (2/13), paraaortic lymph node...


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