Shapiro syndrome and periodic hypothermia have been reported approximately fifty 
times in the literature. Shapiro syndrome is defined as the constellation of 
periodic hypothermia and hyperhidrosis along with agenesis of the corpus 
callosum by Shapiro et al. in 1969. Periodic hypothermia is a more broad 
diagnosis with a number of proposed mechanisms; it occurs in patients without 
structural brain abnormalities. Hematologic abnormalities beyond iron-deficiency 
anemia have not been documented in any of the reported cases of Shapiro syndrome 
or periodic hypothermia. Though accidental and therapeutic hypothermia have been 
associated with thrombocytopenia, this is, to our knowledge, the first reported 
case of periodic intrinsic hypothermia causing bicytopenia. In this report, we 
present the case of a patient with Shapiro syndrome who experienced cyclic 
bicytopenia mirroring hypothermic episodes. We address the differential 
diagnosis of bicytopenia, review the mechanisms proposed for cytopenias related 
to hypothermia, and propose possible mechanisms for the finding in this case.