Methotrexate-associated lymphoproliferative disorder with hypopituitarism and central diabetes insipidus

Abstract

Summary Patients treated with immunosuppressive drugs, especially methotrexate (MTX), rarely develop lymphoproliferative disorders (LPDs), known as MTX-related LPD (MTX–LPD). The primary site of MTX–LPD is often extranodal. This is the first reported case of MTX–LPD in the pituitary. A 65-year-old woman was admitted to our hospital with symptoms of oculomotor nerve palsy and multiple subcutaneous nodules. She had been treated with MTX for 11 years for rheumatoid arthritis. Computed tomography showed multiple masses in the orbit, sinuses, lung fields, anterior mediastinum, kidney, and subcutaneous tissue. Brain magnetic resonance imaging revealed a sellar mass. She was diagnosed with hypopituitarism and central diabetes insipidus based on endocrine examination. Although pituitary biopsy could not be performed, we concluded that the pituitary lesion was from MTX–LPD, similar to the lesions in the sinuses, anterior mediastinum, and subcutaneous tissue, which showed polymorphic LPD on biopsy. MTX was discontinued, and methylprednisolone was administered to improve the neurologic symptoms. After several weeks, there was marked improvement of all lesions, including the pituitary lesion, but the pituitary function did not improve. When pituitary lesions are caused by MTX–LPD, the possibility of anterior hypopituitarism and central diabetes insipidus needs to be considered. Further studies are needed to investigate the effectiveness of early diagnosis and treatment of MTX–LPD in restoring pituitary dysfunction. Learning points Pituitary lesions from MTX–LPD may cause hypopituitarism and central diabetes insipidus. Pituitary metastasis of malignant lymphoma and primary pituitary lymphoma, which have the same tissue types with MTX–LPD, have poor prognosis, but the lesions of MTX–LPD can regress only after MTX discontinuation. In cases of pituitary lesions alone, a diagnosis of MTX–LPD may be difficult, unless pituitary biopsy is performed. This possibility should be considered in patients treated with immunosuppressive drugs. Pituitary hypofunction and diabetes insipidus may persist, even after regression of the lesions on imaging due to MTX discontinuation.

Methotrexate-Associated Lymphoproliferative Disorders Mimicking Granulomatosis With Polyangiitis: A Radiological Diagnostic Challenge

“…5 Methotrexate-associated lymphoproliferative disorder frequently involves the extranodal organs throughout the body. 2,6 Among the extranodal occurrences of MTX-LPD, pulmonary involvement is most frequent. 7,8 In contrast, there are only a few reports of MTX-LPD in the nasal cavity or paranasal sinuses.…”

“…7,8 In contrast, there are only a few reports of MTX-LPD in the nasal cavity or paranasal sinuses. 6,7,[9][10][11] Moreover, there are no previous reports of MTX-LPD mimicking granulomatosis with polyangiitis (GPA) in imaging examinations. Herein, we report a rare case of MTX-LPD mimicking GPA in the nasal cavity and lungs.…”

Importance of Imaging Procedures in the Evaluation of Methotrexate-Associated Lymphoproliferative Disorder of the Thyroid Gland: A Case Study

“…Patients with rheumatoid arthritis (RA) who are treated with methotrexate (MTX) occasionally develop lymphoproliferative disorders (LPDs) [1, 2]. MTX withdrawal often induces prompt regression of LPD, rendering tissue biopsy unnecessary.…”

Tratamento clínico de endometriose profunda com uso de droga antiproliferativa (metotrexato) acoplado a nanoemulsão lipídica (LDE)

“…Este mesmo estudo não encontrou alterações significativas nos valores de TSH das pacientes que receberam a medicação, com diminuição discreta nos parâmetros de T4 livre e T3 livre (Kailajärvi et al, 2000). De apresentação bastante rara, as desordens linfoproliferativas associadas ao metotrexato poderiam cursar com hipopituitarismo (Aoshima et al, 2019).…”