Table 1.
Current Case | Case 1 (10) Negri et al. | Case 2 (10) Negri et al. | Case 3 (10) Negri et al. | Case 4 (10) Negri et al. | |
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Age, Gender | 52, Male | 53, Female | 29, Female | 50, Female | 39, Female |
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Pertinent History | HIV/HBV infection – controlled on ART | Mammary prostheses | Drug-induced CKD Stage III ; Mammary prostheses | Multiple prior implants and fillings | HIV infection – controlled on ART; Multiple prior implants and fillings |
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Filler and Injection Location | PMMA to buttocks and deltoids | PMMA to gluteus | PMMA to gluteus, thighs, and buttocks | PMMA to legs | PMMA to lower extremities |
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Time after PMMA injection to occurrence of hypercalcemia | 5 years (buttocks) 1 month (deltoids) |
4 months | 7 months | 6 months | Not specified, less than 14 months |
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Baseline Labs: | |||||
Serum Ca (mg/dL) | Ca = 14.6* (nl,8.5–10.2) | Ca = 15.2† | Ca = 12 | Ca = 13.2 | Ca = 12 |
iPTH (pg/ml) | iPTH = 3.5 (nl, 12–65) | iPTH = 3.5† | iPTH = 11.6 | iPTH = 15 | iPTH = 16 |
25-D (ng/ml) | 25-D = 17 (nl, 30–100) | 25-D = N/D† | 25-D = 31.2 | 25-D = N/D | 25-D = 9 |
1,25D (pg/ml) | 1,25-D =128 (nl,18–72) | 1,25-D = 55.5† | 1,25-D = 88.3 | 1,25-D = 53.6 | 1,25-D = 94 (nl, 18–60) |
ACE (U/L) | ACE = 200 (nl, 9–67) | ACE = N/D† | ACE = 91 (nl, <40) | ACE = N/D | ACE = N/D |
Cr (mg/dL) | Cr = 2.02 (nl, 0.5–1.1) | Cr = N/D† | Cr = 5.62 | Cr = 2.2 | Cr = N/D |
Alb = N/D† | Alb = N/D | Alb = N/D | Alb = N/D | ||
*Albumin-corrected | †Normal ranges not indicated in case report | ||||
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Relevant physical findings | Prominent muscles diffusely tender to palpation | N/D | N/D | N/D | N/D |
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Relevant imaging | FDG-PET: Uptake in the subcutaneous regions of the proximal thighs, buttocks, and deltoids, without tracer avid lymphadenopathy or masses | N/D | FDG-PET: + metabolic activity at gluteus muscle, isquiotibial quadriceps and internal gemellus of both legs | MRI: nodular images in the subcutaneous tissue compatible with granulomas | FDG-PET: increased activity in the gluteus and thighs between muscles fibers suggesting infiltrates around methacrylate deposits |
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Biopsy findings | Subcutaneous tissue of buttocks and deltoid: Florid giant cell reaction with histiocytic infiltration surrounding globules of amorphous material (PMMA) | Partial excision of gluteal filler: Methacrylate and silicone material associated with a foreign body granulomatous reaction | Sample insufficient | N/D | Muscle biopsy: Granulomas around methacrylate deposits |
Immuno-histochemistry (IHC) | Strong histiocytic expression of CYP27B1 | N/D | N/D | N/D | N/D |
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Treatment | Intravenous (IV) fluids, pamidronate, resumption of antiretroviral therapy; later zoledronic acid + methylprednisolone | Methylprednisone, followed by cyclophosphamide + steroids, then IV fluids, diuretics, calcitionin, then ketoconazole | IV pamidronate, then IV fluids, calcitonin, denosumab; then hemodialysis for progression of renal failure | Methylprednisone | Methylprednisone Intralesional injections of triamcinolone |