Patient-reported outcomes in refractory hormone-producing pituitary adenomas: an unmet need

Abstract

Purpose: To describe quality and outcomes of patient-reported outcome (PRO) measures (PROMs) used in patients with refractory hormone-producing pituitary adenomas, and to provide an overview of PROs in these challenging pituitary adenomas.

Methods: Three databases were searched for studies reporting on refractory pituitary adenomas. For the purpose of this review, refractory adenomas were defined as tumors resistant to primary therapy. General risk of bias was assessed using a component approach and the quality of PROM reporting was assessed using the International Society for Quality of Life Research (ISOQOL) criteria.

Results: 20 studies reported on PROMs in refractory pituitary adenomas, using 14 different PROMs, of which 4 were disease specific (median general risk of bias score: 33.5% (range 6-50%) and ISOQOL score: 46% (range 29-62%)). SF-36/RAND-36 and AcroQoL were most frequently used. Health-related quality of life in refractory patients (measured by AcroQoL, SF-36/Rand-36, Tuebingen CD-25, and EQ-5D-5L) varied greatly across studies, and was not always impaired compared to patients in remission.

Conclusion: There is a scarcity of data on PROs in the subset of pituitary adenomas that is more difficult to treat, e.g., refractory and these patients are difficult to isolate from the total cohort. The patients' perspective on quality of life, therefore, remains largely unknown in refractory patients. Thus, PROs in refractory pituitary adenomas require adequate analysis using properly reported disease specific PROMs in large cohorts to enable appropriate interpretation for use in clinical practice.

Keywords: Functioning adenomas; Health-related quality of life; Patient-reported outcome measure; Pituitary adenomas; Quality of reporting; Refractory adenomas.

Fig. 1

Patient reported outcome measures for refractory patients per study. AcroQoL Acromegaly Quality of Life Questionnaire, ACCQ Acromegaly Comorbidities & Complaints Questionnaire, BDI Beck Depression Inventory, CushingQoL Cushing Quality of Life Questionnaire, EQ-5D-5L 5-level EuroQoL-5, HADS Hospital Anxiety and Depression Scale, MBSRQ Multidimensional Body-Self Relations Questionnaire, PIT QOL Pituitary Quality of Life, PROM patient reported outcome measure, SCL-90-R Symptom Checklist-90-Revised, SF-36 Short Form-36, RAND-36 Research and Development-36, TPQ Cloninger’s Tridimensional Personality Questionnaire, Tuebingen CD-25 Tuebingen Cushing’s Disease Quality of Life Inventory, 15D 15-Dimentional