Discussion
Tofacitinib is a small molecule drug with the potential to cross the placenta. Animal studies have evaluated its teratogenic and fetotoxic effects in pregnant rats and rabbits. However, these studies used exposure doses significantly higher than those in humans.5 Teratogenic effects include the development of membranous ventricular septal defects and cranial/skeletal malformations or deformations. To mitigate these risks, the manufacturer advises the use of effective contraception during tofacitinib treatment and for a period of 4-6 weeks after the last dose. Despite these recommendations, pregnancies can still occur during tofacitinib treatment.
A previous report has noted a case of preterm labor in pregnancies involving tofacitinib treatment.6 Studies have also indicated a potential association between tofacitinib exposure and congenital malformations and spontaneous abortions. However, the frequency of these occurrences does not appear to be significantly different from that observed in the general population.7 Based on limited clinical data, pregnancy outcomes related to tofacitinib have primarily focused on patients with RA and psoriasis. The comprehensive analysis of pregnancy outcomes in these populations has shown similarities to the general population and to patients receiving biologic therapies for inflammatory diseases, and no definitive conclusions can be drawn.8
To the best of our knowledge, this case report presents the first documented instance of using tofacitinib during pregnancy in a patient with SAPHO syndrome. Interestingly, it was observed that the patient experienced an improvement in SAPHO syndrome symptoms during pregnancy, despite discontinuation of the drug and symptoms recurred 40 days after delivery. It is well-established that several immune disorders, including RA, often exhibit significant improvement during pregnancy due to physiological changes in the hormonal status and immune system of pregnant women.9 Thus, pregnancy appears to confer a protective effect in the management of SAPHO syndrome. Currently, the adverse pregnancy outcomes observed in patients with rheumatic diseases are primarily attributed to autoimmune disease activity during pregnancy, underscoring the importance of appropriate antirheumatic medications to control disease activity and minimize side effects in pregnant patients.10 While unintended pregnancies cannot be completely avoided in the treatment of tofacitinib, this may not interfere with SAPHO syndrome therapy. In the case, the patient successfully delivered a healthy newborn after timely discontinuation of tofacitinib and continued using the medication for SAPHO syndrome without breastfeeding. Importantly, no abnormalities were detected in either the mother or the baby.
We report a case of SAPHO syndrome in which an unintended pregnancy occurred after 5 weeks of continuous tofacitinib treatment during the first trimester. Following the discontinuation of the drug, a healthy newborn was delivered. Although the pregnancy outcome in this case was favorable, given the short duration of tofacitinib exposure, further monitoring of pregnancy outcomes in tofacitinib -treated patients is warranted.