25 March 2019 : Case report
Challenging differential diagnosis, Unusual or unexpected effect of treatment, Rare diseaseSinem Koc-Günel1ABCDEF*, Nesrin Tekeli1A, Christina Smaczny1D, Thomas Vogl2DE, Gernot Rohde1CDE
Am J Case Rep 2019; 20:390-393
BACKGROUND: Lymphangioleiomyomatosis (LAM) is a rare progressive cystic and nodular disease of the lung characterized by smooth muscle cell proliferation. LAM predominantly affects young premenopausal women. This report is of a case of LAM presenting in a 47-year-old woman with a past history of breast cancer and discusses the possibility of an association between the two conditions.
CASE REPORT: A 47-year-old woman presented as an emergency with an exacerbation of a four-month history of shortness of breath and dry cough. Her symptoms began following the start of anti-hormonal treatment with letrozole and goserelin acetate for a moderately differentiated (grade 2) invasive ductal carcinoma of the breast (pT2, pN0, M0) which was positive for expression of estrogen receptor (ER+), progesterone receptor (PR+), and human epidermal growth factor receptor 2 (HER2+). Until the previous four months, she had breast-conserving treatment with radiotherapy and tamoxifen therapy. Following hospital admission, she was found to be in type I respiratory failure. Chest X-ray, lung computed tomography (CT), and positron-emission tomography (PET) showed diffuse cystic and nodular lung lesions, consistent with a diagnosis of LAM, and antihormonal therapy was discontinued. She developed pericarditis that was treated with the anti-inflammatory agent, colchicine. Treatment with letrozole and sirolimus improved her respiratory symptoms.
CONCLUSIONS: A rare case of LAM is presented in a woman with a recent history of breast cancer. Because both tumors were hormone-dependent, this may support common underlying gene associations and signaling pathways between the two types of tumor.
Keywords: Aromatase Inhibitors, Carcinoma, Ductal, Breast, Colchicum, Estrogen Receptor Modulators, Goserelin, lymphangioleiomyomatosis, Breast Neoplasms, Lung Neoplasms, Middle Aged, Tomography, X-Ray Computed
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