Spontaneous pregnancy in a patient with a relapse of lymphocytic hypophysitis successfully treated with azathioprine and glucocorticoids

Yang, Guoqing; Ba, Jianming; Dou, Jingtao; Mu, Yiming; Lu, Juming

doi:10.1007/s12020-011-9458-1

Cited by 7 publications

(7 citation statements)

References 5 publications

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“…The second pregnancy also confirmed normal pituitary gland function after LyH. Only a few reports have noted spontaneous pregnancy after a history of LyH ( 7 , 8 , 9 , 10 ). One patient had fully recovered from LyH and completed her pregnancy without any hormonal replacement, although she did not have hypopituitarism ( 7 ).…”

Section: Discussionsupporting

confidence: 62%

Spontaneous pregnancy after full recovery from hypopituitarism caused by lymphocytic hypophysitis

Funazaki

Yamada

Hara

et al. 2018

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryLymphocytic hypophysitis (LyH) has been known to be associated with pregnancy. We herein report the case of a 33-year-old woman who underwent vaginal delivery without massive bleeding at 40 weeks of gestation. Because of the presence of headache and terrible fatigue after childbirth, she visited our hospital. Severe hyponatremia (Na, 118 mEq/L) and visual field abnormality was noted upon examination. MRI revealed pituitary enlargement with a swollen pituitary stalk, albeit at low signal intensity. Basal pituitary hormone levels were all reduced and remained low after exogenous administration of hypothalamic-releasing hormones. She was diagnosed with LyH and was started on prednisolone 60 mg/day. A month later, her pituitary function had gradually improved together with a decrease in pituitary enlargement and recovery of her visual field. The dose of prednisolone was gradually reduced and finally withdrawn 27 months later. After prednisolone withdrawal, her pituitary function remained normal despite the absence of any hormonal replacement. A year later, she became pregnant without medication and delivered a second baby without LyH recurrence. Thereafter, her pituitary function has been normal for more than 5 years. Two valuable observations can be highlighted from the case. First, the patient completely recovered from LyH through prompt prednisolone therapy during its initial phase and had almost normal pituitary function. Second, after recovery from LyH, she was able to undergo spontaneous pregnancy and deliver a baby. We believe that reporting incidences of spontaneous pregnancy after complete normalization of pituitary function in patients with LyH is of great significance.Learning points:Females are more affected by LyH than males given its strong association with pregnancy.LyH possesses characteristic findings on pituitary MRI.Glucocorticoid therapy for LyH has been recommended as an effective treatment.A history of previous pregnancies does not increase the risk of developing AH in subsequent pregnancies.Early induction of high-dose prednisolone was therapeutically effective in treating LyH.

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Section: Discussionsupporting

confidence: 62%

Spontaneous pregnancy after full recovery from hypopituitarism caused by lymphocytic hypophysitis

Funazaki

Yamada

Hara

et al. 2018

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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“…Relapse rate in our series was almost 30 %, and there is no standard management approach for recurrent hypophysitis [48]. Treatment options include additional corticosteroids, other immunosuppressive agents such as azathioprine [49][50][51][52][53][54] or rituximab [55], surgery or radiosurgery [56,57].…”

Section: Discussionmentioning

confidence: 79%

Hypophysitis: a single-center case series

et al. 2014

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“…In patients with a severe form of primary LH, if the disease is recurrent after surgery or resistant to corticosteroid medications, other therapeutic modalities may be introduced such as immunosuppressive/lympholytic therapy (cyclosporine, methotrexate, azathioprine, or rituximab, a monoclonal antibody against CD20 that selectively induces apoptosis of B lymphocytes) (►Table 2). 13,[22][23][24][25][26][27][28][29][30] In our patient, in the view of the high dose of corticosteroids required to avoid clinical symptoms and the evidence of iatrogenic Cushing's syndrome, the immunosuppressive drug azathioprine was used as an alternative long-term treatment. In the first case describing the use of azathioprine in an aggressive form of LH, after 1 month of azathioprine treatment the suprasellar mass had completely disappeared.…”

Section: Discussionmentioning

confidence: 92%

“…In patients with refractory disease, various immunosuppressive therapies are used: cyclosporine, methotrexate, azathioprine, or rituximab. 14,[22][23][24][25][26][27][28][29][30] Only a few refractory patients have been treated with stereotactic radiosurgery (SRS). [31][32][33] SRS is a radiation therapy technique that delivers a precise high dose of irradiation (either single-fraction or multi-fraction SRS) to well-defined small to moderate targets.…”

mentioning

confidence: 99%

Lymphocytic Hypophysitis Successfully Treated with Stereotactic Radiosurgery: Case Report and Review of the Literature

Pekić

Bogosavljević

Peker

et al. 2017

J Neurol Surg A Cent Eur Neurosurg

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Lymphocytic hypophysitis (LH) is an autoimmune inflammatory infiltration of the pituitary gland, usually with a benign evolution. In rare circumstances the inflammatory process may extend beyond the pituitary and infiltrate the surrounding structures. We present a 42-year-old woman affected by an aggressive form of LH with extension to the cavernous sinus causing internal carotid artery occlusion and right sixth cranial nerve palsy. Prednisone therapy caused severe iatrogenic Cushing's syndrome, and the patient underwent transsphenoidal decompression. The histopathology report was consistent with LH. The patient was symptom free for a short period with reappearance of severe headache, diplopia, and hearing loss (middle ear inflammation) 3 months after surgery. Corticosteroids were reintroduced with the addition of azathioprine, but there was no regression of the pituitary mass. The patient was referred for stereotactic radiosurgery (SRS) using Gamma Knife (15 Gy to the margin). After 26 months, azathioprine was stopped, and the dose of prednisone was gradually tapered to 7.5 mg/day. Sellar magnetic resonance imaging showed regression of the pituitary mass. After follow-up for > 3 years after SRS, there was no clinical or radiologic evidence of the disease, but carotid arteries remained occluded. The patient developed secondary hypothyroidism and hypogonadism as consequences of SRS. An aggressive form of LH extending beyond the pituitary gland infiltrating surrounding structures is described. It was successfully treated with SRS after failure of transsphenoidal surgery and combined immunosuppressive therapy (prednisone, azathioprine). The review of the literature presents timely information concerning treatment with azathioprine and SRS of patients with an aggressive form of LH.

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Spontaneous pregnancy in a patient with a relapse of lymphocytic hypophysitis successfully treated with azathioprine and glucocorticoids

Cited by 7 publications

References 5 publications

Spontaneous pregnancy after full recovery from hypopituitarism caused by lymphocytic hypophysitis

Spontaneous pregnancy after full recovery from hypopituitarism caused by lymphocytic hypophysitis

Hypophysitis: a single-center case series

Lymphocytic Hypophysitis Successfully Treated with Stereotactic Radiosurgery: Case Report and Review of the Literature

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