Clinical presentation of celiac disease (CD) in terms of gender have demonstrated differences between symptoms among women and men. This study evaluated the differences in the clinical presentation of CD among Brazilian men and women at the time of diagnosis.We investigated data on symptoms and clinical signs of 240 patients (80 [33.3%] men and 160 [66.6%] women) aged >18 years who were diagnosed with CD during 2000-2017. The overall mean age at diagnosis was 38.3 ± 13.28 years, with the mean age being 38.1 ± 14.48 years for men and 38.4 ± 12.68 years for women (p = 0.16). Details regarding other previously diagnosed autoimmune diseases were enquired with the patient. All data were collected at the time of diagnosis of CD.Symptoms such as anxiety/depression (OR = 7.9), lack of appetite (OR = 3.5), flatulence (OR = 1.9), malaise (OR = 2.5), nausea (OR = 3.1), and vomiting (OR = 6.5) were more frequent in females than in males. Only steatorrhea was more frequent in men (OR = 2.6). Clinical signs such as anemia (OR = 2.8), short stature (OR >20), and weight loss (OR = 2.4) were also more frequent in women. Other findings such as diarrhea, abdominal distension or pain, and anorexia did not reveal differences in terms of gender. Regarding concomitant autoimmune diseases, dermatitis herpetiformis was more frequent in men (OR = 2.6), and thyroiditis was more frequent in women (OR = 10.5).Our study has demonstrated that men and women with CD presented differences in clinical presentation at the time of diagnosis. Women exhibited more gastrointestinal symptoms and clinical signs than men.