<p>Sanjad-Sakati syndrome (SSS) is a rare autosomal recessive disorder characterized by hypoparathyroidism, growth retardation, and dysmorphic features. However, the hematological profile of affected patients remains poorly characterized. This study aimed to investigate complete blood count (CBC) parameters in patients with SSS compared to healthy controls. This cross-sectional observational study included 19 patients with Sanjad-Sakati syndrome and 31 age- and sex-matched healthy controls. Blood samples were collected for CBC analysis measuring 14 hematological parameters. Statistical comparisons were performed using independent <i>t</i>-tests or Mann–Whitney <i>U</i> tests with Benjamini–Hochberg correction for multiple comparisons. Effect sizes (Cohen’s <i>d</i> or <i>r</i>) were calculated to quantify differences. Patients with SSS showed significantly lower red blood cell count, hemoglobin (10.16 ± 1.86 vs. 13.87 ± 1.13&#xa0;g/dL), hematocrit (32.25 ± 5.15 vs. 38.75 ± 2.75%), MCH, and MCHC compared to controls (all <i>p</i> &lt; 0.01). Conversely, white blood cell count (13.37 ± 5.74 vs. 6.08 ± 1.61 × 10⁹/L), RDW-CV (18.69 ± 4.05 vs. 13.15 ± 0.77%), eosinophils, basophils, and monocytes were significantly elevated in patients (<i>p</i> &lt; 0.001). The largest effect sizes were observed for hemoglobin (<i>d</i> =  − 2.57), RDW (<i>d</i> = 2.17), and WBC (<i>d</i> = 1.95). MCV, platelet count, neutrophils, and lymphocytes showed no significant differences.</p><p><i>Conclusion</i>:&#xa0;Sanjad-Sakati syndrome is associated with significant hematological alterations including microcytic anemia and leukocyte imbalances. Routine CBC monitoring is recommended for clinical management of affected patients.</p>