Introduction: Belly dancer dyskinesia (BDD) is a visible, involuntary, semicontinuous, waving, and writhing type of abdominal wall movement, which resembles those of a belly dancer. The etiology is unknown, but it is thought to be due to cerebral, spinal, hormonal, or drug-induced effects. Epilepsy as one of the factors of BDD, is considered to have a high risk in pregnancy, where 25–33.3% of seizures will increase during pregnancy. Case summary: A 33-year-old multigravid woman in her 34th week of gestation, who had a prior vertical incision C-section, presented with painless, intermittent rhythmic repetitive waving involuntary movements of the abdominal wall with no involvement of other parts of the body since 1 year ago. She was referred from a local hospital after suffering from generalized absence seizures with bizarre abdominal wall movement and irregular abdominal contraction. Basic laboratory tests, including complete blood count (CBC), liver enzymes [(alanine transaminase (ALT), aspartate transaminase (AST), serum creatinine level, and blood urea nitrogen (BUN)] levels were normal. Head-CT showed left temporal infarction. Head-MRI with contrast showed lacunar infarcts in bilateral centrum semilobar, perivascular space in bilateral corona radiate, and bilateral centrum semilobar. Electroencephalography (EEG) results showed wave slowing on the right and left temporal. During hospitalization, she suffered sudden abdominal pain with increasing abdominal wall movements and hematuria then she underwent an emergency cesarean section, and intraoperatively we found an incomplete uterine rupture. Right now she is treated with trihexyphenidyl, levetiracetam, carbamazepine, and haloperidol. She had significant improvement in terms of frequency and degree of abnormal movement. Discussion: Belly dancer dyskinesia in pregnancy is so rare that only four cases were reported in 2021 worldwide. To our knowledge, there has been no article discussing BDD with incomplete uterine rupture. The etiology of this condition is still multifactorial. Belly dancer dyskinesia is also known to arise from drug induction, which is most commonly caused by antidopaminergic drugs. The management of BDD can be challenging depending on the underlying etiologies. Conclusion: Belly dancer dyskinesia is a rare type of movement disorder with no clearly understood etiologic agent, pathogenesis, and effective treatment. To understand the etiology and possible mechanisms or pathogenesis as well as to determine the best effective treatment, thorough investigation is important.