RESUMO
Various immune-related adverse events (irAEs), including fulminant type 1 diabetes (FT1D), are known to be associated with immune checkpoint inhibitors (ICIs). We experienced two lung adenocarcinoma cases who developed fulminant type 1 diabetes long after discontinuation of ICI therapies. One, a 74-year-old male, received nivolumab and developed fulminant type 1 diabetes 44 days after the last infusion. The other, an 85-year-old male, received atezolizumab and developed fulminant type 1 diabetes 171 days after the last infusion. Clinical ICI treatment guidelines recommend laboratory tests during ICI treatments but the necessity of tests in patients whose ICI therapy has been discontinued is not clearly described. These cases indicate that blood glucose monitoring should be continued at least for several months, and that patients should be informed of the possibility of fulminant type 1 diabetes after ICI discontinuation, because fulminant type 1 diabetes progresses rapidly and can be life-threatening if not promptly recognized.
Assuntos
Anticorpos Monoclonais Humanizados/efeitos adversos , Diabetes Mellitus Tipo 1 , Inibidores de Checkpoint Imunológico , Neoplasias Pulmonares/tratamento farmacológico , Nivolumabe/efeitos adversos , Idoso , Idoso de 80 Anos ou mais , Glicemia , Automonitorização da Glicemia , Diabetes Mellitus Tipo 1/induzido quimicamente , Humanos , Inibidores de Checkpoint Imunológico/efeitos adversos , Neoplasias Pulmonares/complicações , MasculinoAssuntos
Diabetes Mellitus Tipo 2/complicações , Músculos Isquiossurais/diagnóstico por imagem , Infarto/diagnóstico por imagem , Músculo Quadríceps/diagnóstico por imagem , Biópsia , Proteína C-Reativa/metabolismo , Creatina Quinase/metabolismo , Nefropatias Diabéticas/etiologia , Nefropatias Diabéticas/terapia , Retinopatia Diabética/etiologia , Diagnóstico Diferencial , Febre/etiologia , Músculos Isquiossurais/irrigação sanguínea , Humanos , Infarto/etiologia , Infarto/metabolismo , Infarto/patologia , Falência Renal Crônica/etiologia , Falência Renal Crônica/terapia , Imageamento por Ressonância Magnética , Masculino , Pessoa de Meia-Idade , Músculo Esquelético/irrigação sanguínea , Músculo Esquelético/diagnóstico por imagem , Músculo Esquelético/patologia , Piomiosite/diagnóstico , Músculo Quadríceps/irrigação sanguínea , Músculo Quadríceps/patologia , Diálise Renal , Coxa da PernaRESUMO
CONTEXT: Tumors producing IGF-2 (IGF-2oma) are a major cause of spontaneous hypoglycemia. The treatment mainstay is surgical resection. Many case reports note resolution of hypoglycemia after IGF-2oma resection; however, outcomes are variable according to tumor type. We report a case of resolving hypoglycemia, observed on continuous glucose monitoring, after resection of an IGF-2-producing solitary fibrous tumor of pleura and review the current literature. CASE REPORT: A 69-year-old woman presented with impaired consciousness because of hypoglycemia. An IGF-2oma was diagnosed as the cause for hypoglycemia because of decreased serum insulin and IGF-1, the presence of a pleural tumor, and a high-molecular-weight form of serum IGF-2 detected by Western immunoblot. Surgical resection was performed; pathological examination demonstrated a solitary fibrous tumor with low-grade malignancy. Continuous glucose monitoring showed reversal of hypoglycemia after tumor resection. Approximately 2 years after resection, the patient has no signs of tumor recurrence or hypoglycemia. CONCLUSIONS: An IGF-2-producing solitary fibrous tumor of pleura in this case caused hypoglycemia. From a search of the literature of 2004-2014, 32 cases of IGF-2oma with hypoglycemia that underwent radical surgery were identified; in 19 (59%) patients, hypoglycemia was reversed, and there was no subsequent recurrence. The remaining 13 (41%) patients experienced tumor recurrence or metastasis an average of 43 months after initial tumor resection. The tumor of the present case was a low-grade malignancy. Regular follow-up with biomarker monitoring of glucose metabolism and assessment of hypoglycemic symptomatology, in conjunction with imaging tests, is important for detecting possible tumor recurrence and metastasis.