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Open access

Alessandro Prete, Giada Cosentino, Luca Manetti, Carlo Enrico Ambrosini, Piermarco Papini, Michele Marinò, Liborio Torregrossa, Claudio Marcocci, Rossella Elisei, and Isabella Lupi

Summary

In elderly patients presenting with a solid thyroid mass, the differential diagnosis between benign and malignant lesion is not always straightforward. We present the case of an 85-year-old woman with fever and an enlarged, firm and painful thyroid mass. Blood exams documented a mild thyrotoxicosis with a moderate inflammatory status. Thyroid scintiscan showed an absent uptake of 131I. Ultrasound and CT scan documented a 3 cm hypoechoic nodule with infiltration of the sternocleidomastoid muscle, very suspicious for neoplastic nature. Fine-needle aspiration and tru-cut biopsy were performed. During biopsy, the lesion was partially drained and a brownish fluid was extracted. The culture resulted positive for Klebsiella pneumoniae whereas the pathological analysis of the specimen was not conclusive due to the presence of an intense inflammatory response. A targeted oral antibiotic therapy was then initiated, obtaining only a partial response thus, in order to achieve a definite diagnosis, a minimally invasive hemithyroidectomy was performed. The pathological analysis documented acute suppurative thyroiditis and the clinical conditions of the patient significantly improved after surgical removal of thyroid abscess. In elderly patients with a solid thyroid mass, although neoplastic origin is quite frequent, acute suppurative thyroiditis should be considered as a differential diagnosis.

Learning points:

  • A solid and rapidly growing thyroid mass in elderly patients can hide a multifaceted variety of diseases, both benign and malign.
  • A multidisciplinary team (endocrinologist, surgeon, radiologist and pathologist) could be necessary in order to perform a correct differential diagnosis and therapeutic approach.
  • Surgery can be decisive not only to clarify a clinically uncertain diagnosis, but also to rapidly improve the clinical conditions of the patient.
Open access

Isabella Lupi, Alessandro Brancatella, Mirco Cosottini, Nicola Viola, Giulia Lanzolla, Daniele Sgrò, Giulia Di Dalmazi, Francesco Latrofa, Patrizio Caturegli, and Claudio Marcocci

Summary

Programmed cell death protein 1/programmed cell death protein ligand 1 (PD-1/PD-L1) and cytotoxic T-lymphocyte antigen 4/B7 (CTLA-4/B7) pathways are key regulators in T-cell activation and tolerance. Nivolumab, pembrolizumab (PD-1 inhibitors), atezolizumab (PD-L1 inhibitor) and ipilimumab (CTLA-4 inhibitor) are monoclonal antibodies approved for treatment of several advanced cancers. Immune checkpoint inhibitors (ICIs)-related hypophysitis is described more frequently in patients treated with anti-CTLA-4; however, recent studies reported an increasing prevalence of anti-PD-1/PD-L1-induced hypophysitis which also exhibits slightly different clinical features. We report our experience on hypophysitis induced by anti-PD-1/anti-PD-L1 treatment. We present four cases, diagnosed in the past 12 months, of hypophysitis occurring in two patients receiving anti-PD-1, in one patient receiving anti-PD-1 and anti-CTLA-4 combined therapy and in one patient receiving anti-PD-L1. In this case series, timing, clinical presentation and association with other immune-related adverse events appeared to be extremely variable; central hypoadrenalism and hyponatremia were constantly detected although sellar magnetic resonance imaging did not reveal specific signs of pituitary inflammation. These differences highlight the complexity of ICI-related hypophysitis and the existence of different mechanisms of action leading to heterogeneity of clinical presentation in patients receiving immunotherapy.

Learning points:

  • PD-1/PD-L1 blockade can induce hypophysitis with a different clinical presentation when compared to CTLA-4 blockade.
  • Diagnosis of PD-1/PD-L1 induced hypophysitis is mainly made on clinical grounds and sellar MRI does not show radiological abnormalities.
  • Hyponatremia due to acute secondary adrenal insufficiency is often the principal sign of PD-1/PD-L1-induced hypophysitis and can be masked by other symptoms due to oncologic disease.
  • PD-1/PD-L1-induced hypophysitis can present as an isolated manifestation of irAEs or be in association with other autoimmune diseases