Case report
A 31-year-old woman presented in 2022 with persistent anterior chest
wall pain and palmoplantar pustulosis (PPP, Figure1A ).
Whole-body bone scintigraphy revealed abnormal radioactive uptake at the
sternoclavicular joint (Figure 2 ). The diagnosis of SAPHO
syndrome was made based on clinical manifestations and imaging
examinations. Initial treatment with diclofenac 75mg twice daily for one
month did not lead to symptom improvement. Subsequently, the patient was
treated with adalimumab at a dosage of 40mg every two weeks, resulting
in an initial satisfactory remission. However, after 10 weeks of
treatment, the patient experienced a recurrence of rash. In July, the
patient initiated a trial of tofacitinib treatment at a dose of 5mg
twice daily, which provided significant relief from the rash.
Unexpectedly, after 10 weeks of tofacitinib therapy, the patient
discovered a 5-week pregnancy and promptly discontinued the use of
tofacitinib.
During the patient’s pregnancy, regular monitoring was conducted to
ensure the well-being and development of the fetus. The screening for
chromosomal abnormalities showed the fetus to be at low risk for trisomy
13, 18, and 21. The second and third trimester of pregnancy, fetal
growth parameters including skull length, biparietal diameter, abdominal
perimeter, and femur length remained within the normal ranges.
Continuous monitoring of the fetal heart rate and predicted weight were
also normal. Ultrasound scans conducted at 20 weeks and beyond did not
detect any external or internal fetal defects. At the 39th week of
pregnancy, a cesarean section was performed, and a healthy infant was
delivered. The baby had a birth weight of 2900 g and a length of 51 cm,
with no congenital anomalies or dysfunctions detected.
The patient experienced a notable reduction in sternoclavicular pain and
PPP (Figure 1B ) during pregnancy. However, approximately 40
days after delivery, the symptoms resurfaced (Figure 1C ). The
patient continues to use tofacitinib 5mg twice daily as treatment for
SAPHO syndrome and is still being followed up.