Gougerot-Sjogren-like syndrome under PD-1 inhibitor treatment

A 36-year-old female patient with no significant medical history except hypothyroidism was diagnosed with left parotid acinic cell carcinoma that was treated by radical parotidectomy followed by adjuvant radiotherapy (50 Gy) in 2004. Seven years after the initial diagnosis, the patient developed histologically confirmed metastases on the left adrenal gland and the lung, for which systemic therapy was indicated. The patient participated in two successive studies and received lapatinib and then an EZH-2 inhibitor. In March 2016, the patient's adrenal lesion progressed, and she started treatment with pembrolizumab, 200 mg, every 3 weeks. At the time of the 11th injection, the patient experienced a retinal detachment that was linked to her myopia. The detachment was treated by surgery and amoxicillin for 7 days. Subsequently, the patient developed an oral candidiasis that was treated with systemic fungizone. At the time of the 13th injection, in December 2016, the patient experienced a debilitating grade 2 dry-eye syndrome that was associated with grade 1 conjunctival hyperemia, grade 2 xerostomia, and grade 1 skin rash on both hands. Therefore, serum protein electrophoresis (SPEP) and determination of antinuclear antibodies (ANA) [particularly Sjogren's syndrome A/Sjogren's syndrome B (SSA/SSB)] were carried out. A salivary gland biopsy could not be conducted because of the patient's radiation history. The SPEP and the ANA were normal. Concomitantly, the CT-scan showed a stable disease using the RECIST criteria v1.1.

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