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Successful treatment of medically and surgically refractory lymphocytic hypophysitis with fractionated stereotactic radiotherapy: a single-center experience and systematic literature review

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Abstract

Purpose

To explore the potential role of focused radiotherapy in managing the lymphocytic hypophysitis (LH) refractory to medical therapy and surgery.

Method

A systematic literature review was conducted following PRISMA guidelines to identify the studies on radiation treatment for hypophysitis, along with the experience in our institution.

Results

The study included eight patients, three from our institution and five from existing literature. The age at presentation ranged from 37 to 75 years old, with a median age of 58. The presenting symptoms involved headache in seven patients and diplopia in two patients. Pre-radiation visual field defects were noticed in four patients. All patients exhibited variable degrees of hypopituitarism before radiation, with oral corticosteroids being the initial medical treatment. Immunosuppressive therapy was attempted in two patients prior to radiation. Seven patients had a history of transsphenoidal surgery with a histologically confirmed LH. Three patients underwent stereotactic radiosurgery (SRS), while the remaining received FSRT, with a mean irradiation volume of 2.2 cm3. A single-session total dose of 12 -15 Gy was administered in the SRS group. In the FSRT group, doses ranged from 24 to 30 Gy with a median dose of 25 Gy, delivered in 2 Gy fractions. Four patients achieved a resolution of visual field defects, while another two patients demonstrated improvement in their associated focal neurologic deficits. No change in pre-existing endocrine status was shown after radiation, except in one patient. Clinical response was achieved in seven patients after a single course of radiation, while one patient required the second course. Six patients remained stable on low-dose glucocorticoid during at least a 12-month follow-up period, and one discontinued it entirely without experiencing relapse. Three patients demonstrated a complete radiologic response, while the remaining showed a partial radiologic response.

Conclusions

Focused radiation, including FSRT, can play a role in symptomatic relief, effective mass shrinkage, and minimizing radiation exposure to critical surrounding structures in patients with refractory LH. However, further research efforts are necessary to better clarify its effects and optimal dose planning.

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Funding

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Authors and Affiliations

Authors

Contributions

Data collection: MK, GF, and VM. Literature review and data interpretation: MK and GF. Writing: MK. Figure preparation: MK, GF, PK, DB, VM, KD Critical review and final approval: all authors

Corresponding author

Correspondence to Daniel Prevedello.

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Conflicts of interest

Dr. Daniel Prevedello is a consultant for Stryker Corp., Medtronic Corp., BK Medical and Integra; he has received an honorarium from Mizuho and royalties from KLS- Martin and ACE Medical. All other authors do not have conflict of interest to report.

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This paper abstract has been selected to be presented in the Congress of Neurological Surgeon (CNS) in September 2023. The authors declare that this manuscript is a unique submission and has not been published in whole or in part elsewhere in any form.

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Khaleghi, M., Finger, G., Wu, K.C. et al. Successful treatment of medically and surgically refractory lymphocytic hypophysitis with fractionated stereotactic radiotherapy: a single-center experience and systematic literature review. Pituitary 27, 213–229 (2024). https://doi.org/10.1007/s11102-023-01367-8

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