PMC9361946_01
Patient's bone scan and 68 Ga-DOTATATE-PET-CT at time of TIO diagnosis in 2013. a Bone scan shows uptake of the tracer (Tc99) in the fracture/pseudofractures areas. Patient has a rachitic rosary at the costochondral junctions, which is an evocative sign in adults with TIO. Asterisk highlights fractures and pseudofractures. 68 Ga-DOTATATE-PET-CT (b
68 Ga-DOTATATE-CT and c
68 Ga-DOTATATE-PET) shows the oval pre-sacral lesion with enhanced uptake (white arrows). The lesion is located in the pre-sacral adipose tissue, closely adherent to the anterior wall of sacrum and anteriorly related to the rectus intestinus, which imprints but does not infiltrate. Plane of cleavage with the left piriformis muscle is preserved
198_2022_6516_Fig1_HTML.jpg
PMC9361946_01_198_2022_6516_Fig1_HTML.jpg
Fig1
Figure 1 shows bone scan and 68 Ga-DOTATATE-PET-CT at baseline.
VAS pain change (a) and serum phosphate change (b) from baseline to the last evaluation. Changes in bone scan at baseline (c) and 1 year after burosumab start (d). a shows the VAS trend during 2 years. b shows the serum phosphate levels during the 2 years. Gray arrows indicate burosumab dose escalation. Burosumab was stopped at the beginning of October 2020 due to COVID-19 infection and restarted on 11-11-2020. Bone scan shows the uptake of tracer at fracture sites (left and right foot, ribs, bilateral iliopubic branch) before burosumab onset in September 2019 (c). d shows the reduction of radiotracer uptake particularly at lower limbs and ribs 1 year later
198_2022_6516_Fig2_HTML.jpg
PMC9361946_01_198_2022_6516_Fig2_HTML.jpg
Fig2
After 2 months, she experienced significant pain symptom improvement (visual analogic scale (VAS) [from 0 = no pain to 100 = maximal pain] reduction from 65 to 12 mm, Fig. 2a), which allowed her to walk and stand without crutches. After the negative swab, burosumab was started again at 40 mg/every 4 weeks with normalization of SP levels in 4 months (SP 2.5 mg/dL) (Fig. 2b). Changes in bone scan over time are shown in Fig. 2c.