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.