Tohoku J. Exp. Med., 2016 March, 238(3)

Reversible Hypopituitarism Associated with Intravascular Large B-Cell Lymphoma: Case Report of Successful Immunochemotherapy

YUSUKE SAWADA,¹ SUMIYASU ISHII,¹ YASUHIKO KOGA,¹ TAKU TOMIZAWA,¹ AYAKO MATSUI,¹ TAKUYA TOMARU,¹ ATSUSHI OZAWA,¹ NOBUYUKI SHIBUSAWA,¹ TETSUROU SATOH,¹ HIROAKI SHIMIZU,² JUNKO HIRATO³ and MASANOBU YAMADA¹

¹Department of Medicine and Molecular Science, Gunma University Graduate School of Medicine, Maebashi, Gunma, Japan
²Department of Medicine and Clinical Science, Gunma University Graduate School of Medicine, Maebashi, Gunma, Japan
³Department of Pathology, Gunma University Hospital, Maebashi, Gunma, Japan

Intravascular large B-cell lymphoma (IVLBCL) is a rare subtype of diffuse large B-cell lymphoma. There have been only a limited number of reports regarding pituitary dysfunction associated with IVLBCL. We present a 71-year-old woman with hypopituitarism without any hypothalamic/pituitary abnormalities as assessed by magnetic resonance imaging. She presented with edema, abducens palsy, and elevated levels of lactate dehydrogenase and soluble interleukin-2 receptor. Provocative testing showed that the peaks of luteinizing hormone, follicle-stimulating hormone, thyroid-stimulating hormone and adrenocorticotropic hormone were evoked to normal levels by simultaneous administration of luteinizing hormone-releasing hormone, thyrotropin-releasing hormone and corticotropin-releasing hormone, but the responses of these four pituitary hormones showed a delayed pattern. She was diagnosed with IVLBCL with cerebrospinal invasion by pathological findings of the bone marrow, skin, and cerebrospinal fluid. She achieved hematological remission after immunochemotherapy. Pituitary function was also restored without hormonal replacement, and the improvement of the pituitary function was confirmed by dynamic testing. We reviewed the literature with respect to hypopituitarism associated with IVLBCL. There were less than 20 case reports and most of the patients died. Endocrinological course was described in only two cases, and both of them required hormonal supplementation. To our knowledge, this is the first case of hypopituitarism induced by IVLBCL that was successfully managed by immunochemotherapy alone. This case suggests that early diagnosis and treatment of IVLBCL might improve anterior pituitary function and enable patients to avoid hormone replacement therapy.

keywords —— abducens palsy; hypopituitarism; immunochemotherapy; intravascular large B-cell lymphoma; pituitary dynamic test

© 2016 Tohoku University Medical Press

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Tohoku J. Exp. Med., 2016, 238, 197-203

Correspondence: Sumiyasu Ishii, M.D., Ph.D., Department of Medicine and Molecular Science, Gunma University Graduate School of Medicine, 3-39-15 Showa-machi, Maebashi, Gunma 371-8511, Japan.

e-mail: sishii@gunma-u.ac.jp