Among 243 patients who received kidney transplants at our center, two patients suddenly developed a severe debilitating pain syndrome in the lower limbs.
Case no. 1A 49-year-old male patient who received a deceased donor kidney transplant, in therapy with tacrolimus (plus steroids and perioperative basiliximab). One month after transplantation he reported joint pain in his ankles, knees, feet, and hands. The pain became so intense that the patient was forced to use crutches to walk. Clinical examination revealed intense pain at movement, without edema, redness, increase in temperature, or cutaneous trophic alterations. The patient was receiving tacrolimus 5 mg b.i.d. with levels between 5 and 10 ng/ml. There was no clinical or serologic evidence of rheumatic disease or rhabdomyolysis. The rise in serum creatinine was attributed to heavy non-steroidal antiinflamatory drug use. After withdrawing these agents, the serum creatinine decreased to 1.6 mg/dl. Bone radiographies showed osteoporosis at the heads of the knee, ankle, tarsal, and metatarsal bones. An ultrasound scan of the joints highlighted a minimal amount of articular effusion and a mild synovial reaction in the knee and foot joints. Magnetic resonance imaging (MRI) of the left knee showed an area of bone marrow edema in the external condyle of the femur and wearing of the cartilage (Figure 1). Computerized bone mineralometry showed a slight reduction of the bone mass, whereas bone scintigraphy revealed increased radionuclide uptake in the affected joints (Figure 2).
Generalized lymphedema is an extremely rare effect of sirolimus therapy in renal transplant recipients. We describe the development of this complication in a 56-yr-old woman, who was given an experimental protocol with cyclosporine, sirolimus, steroids, and basiliximab. Following the protocol, after one month, the patient was randomized to the "sirolimus only" group, while cyclosporine was completely suspended and the oral steroids were continued. Three months later, the patient was admitted for severe lymphedema of the lower limbs, with significant weight increase, massive ascites and dyspnea, but excellent renal function. A chest radiography and an ultrasound study of the heart showed a moderate pleural and pericardial effusion. An abdominal ultrasound scan showed two small lymphoceles next to the transplanted kidney, confirmed with a CT scan. After sirolimus discontinuation the generalized lymphedema started to improve and three months later all the symptoms had disappeared.
Nodular neck lesions mimicking a thyroid pathology (thyroid nodules or metastatic lymph nodes) are rare but can represent a tough challenge for surgeons who might fall into incorrect surgical approaches, resulting in high morbidity. Pre-operative work-up would help the surgeon to obtain the correct diagnosis, thus, to follow the better surgical approach. Nevertheless, a careful approach would be used for that neurogenic tumour amenable of resection without jeopardising nervous structures.
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