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.