Annamaria De Bellis1, Giuseppe Bellastella2, Maria Ida Maiorino2, Ernesto Aitella2, Emma Lucci2, Domenico Cozzolino2, Antonio Bellastella2, Antonio Bizzarro2, Dario Giugliano2, Katherine Esposito2. 1. Department of Cardiothoracic and Respiratory SciencesEndocrinology and Metabolic Diseases Unit, Second University of Naples, Piazza L. Miraglia 2, 80131 Napoli, ItalyDepartment of MedicalSurgical, Neurological, Metabolic and Geriatric Sciences, Endocrinology and Metabolic Diseases UnitDepartment of Clinical and Experimental MedicineSecond University of Naples, Napoli, Italy annamaria.debellis@unina2.it. 2. Department of Cardiothoracic and Respiratory SciencesEndocrinology and Metabolic Diseases Unit, Second University of Naples, Piazza L. Miraglia 2, 80131 Napoli, ItalyDepartment of MedicalSurgical, Neurological, Metabolic and Geriatric Sciences, Endocrinology and Metabolic Diseases UnitDepartment of Clinical and Experimental MedicineSecond University of Naples, Napoli, Italy.
Abstract
BACKGROUND: Some cases of apparently idiopathic GH deficiency (GHD) may be caused by pituitary autoimmunity. OBJECTIVE: To study the variations in pituitary function and antipituitary antibodies (APA) from childhood to transition age in patients with apparently idiopathic GHD. DESIGN: We conducted a longitudinal study. PATIENTS AND METHODS: Pituitary function and APA detection by immunofluorescence were investigated in 24 childhood patients with isolated GHD before starting recombinant GH therapy and after the stopping of this therapy in transition age. Sera of patients positive for APA were processed by double immunofluorescence to identify their pituitary target. RESULTS: At diagnosis, 16 out of 24 patients were APA positive targeting only somatotrophs (group 1), while the remaining eight were APA negative (group 2). When retested off therapy, 12 out of 16 patients in group 1 persisted being APA positive, while the remaining four became negative with recovery of pituitary function. All patients in group 2 persisted being APA negative but still showing GHD. Of the 12 patients persistently APA positive, eight with confirmed GHD showed APA still targeting somatotrophs, whereas four showed APA targeting only gonadotrophs associated with isolated hypogonadotropic hypogonadism (HH). CONCLUSION: Patients with APA at middle but not at high titer in childhood may show a remission of autoimmune GHD in childhood after GH replacement therapy. As APA may shift their target in transition period, an early characterization of APA by double immunofluorescence is advisable in APA positive GHD patients showing delayed puberty, to allow an early diagnosis and an appropriate therapy, thus preventing the progression toward HH.
BACKGROUND: Some cases of apparently idiopathic GH deficiency (GHD) may be caused by pituitary autoimmunity. OBJECTIVE: To study the variations in pituitary function and antipituitary antibodies (APA) from childhood to transition age in patients with apparently idiopathic GHD. DESIGN: We conducted a longitudinal study. PATIENTS AND METHODS: Pituitary function and APA detection by immunofluorescence were investigated in 24 childhood patients with isolated GHD before starting recombinant GH therapy and after the stopping of this therapy in transition age. Sera of patients positive for APA were processed by double immunofluorescence to identify their pituitary target. RESULTS: At diagnosis, 16 out of 24 patients were APA positive targeting only somatotrophs (group 1), while the remaining eight were APA negative (group 2). When retested off therapy, 12 out of 16 patients in group 1 persisted being APA positive, while the remaining four became negative with recovery of pituitary function. All patients in group 2 persisted being APA negative but still showing GHD. Of the 12 patients persistently APA positive, eight with confirmed GHD showed APA still targeting somatotrophs, whereas four showed APA targeting only gonadotrophs associated with isolated hypogonadotropic hypogonadism (HH). CONCLUSION:Patients with APA at middle but not at high titer in childhood may show a remission of autoimmune GHD in childhood after GH replacement therapy. As APA may shift their target in transition period, an early characterization of APA by double immunofluorescence is advisable in APA positive GHD patients showing delayed puberty, to allow an early diagnosis and an appropriate therapy, thus preventing the progression toward HH.
Authors: Sriram Gubbi; Fady Hannah-Shmouni; Joseph G Verbalis; Christian A Koch Journal: Best Pract Res Clin Endocrinol Metab Date: 2019-12-12 Impact factor: 4.690
Authors: Giuseppe Bellastella; Maria Ida Maiorino; Miriam Longo; Paolo Cirillo; Lorenzo Scappaticcio; Maria Teresa Vietri; Antonio Bellastella; Katherine Esposito; Annamaria De Bellis Journal: Int J Mol Sci Date: 2020-02-19 Impact factor: 5.923
Authors: Giuseppe Bellastella; Maria Ida Maiorino; Antonio Bizzarro; Dario Giugliano; Katherine Esposito; Antonio Bellastella; Annamaria De Bellis Journal: Pituitary Date: 2016-12 Impact factor: 4.107