Inflammatory myofibroblastic tumor (IMT) is an uncommon condition that is rarely encountered in the urinary tract. In this report, we present a case of IMT of the bladder in a woman with multiple previous pelvic surgeries. We further review the relevant literature to highlight this rare but important clinical presentation.
Therapeutic plasma exchange was attempted on a 53-year-old female with angioimmunoblastic T-cell lymphoma with associated combined cold/warm immune hemolytic anemia and hyperviscosity syndrome from hypergammaglobulinemia. Her preprocedure hematocrit (Hct) was 16.8%, despite daily red blood cell (RBC) transfusions. Due to concern for cold agglutinins aggregating in the tubing coil within the centrifuge channel, all attempts were made to optimize the ambient temperature in the room and warm the extracorporeal tubing before plasma exchange; however, the maximum room temperature that could be obtained was 308C. The apheresis machine was allowed to equilibrate to the ambient room temperature, and all extracorporeal circuitry was wrapped with hot packs (Jack Frost, Cardinal Health, McGraw Park, IL). The extracorporeal circuit was primed to a Hct of 20% per our internal protocol for a preprocedure Hct less than 20%. A few minutes into the procedure, the apheresis inflow line showed bright pink-red blood instead of the anticipated maroon color seen in the outflow line. The procedure was paused and a syringe sample from the inflow line was centrifuged and showed pink-red supernatant consistent with hemoglobin from hemolysis; a spun Hct was 4%. A photograph (figure, left) shows the fluid in the plasma collection bag, with RBCs sedimenting on the bottom beneath bright pink-red fluid. The procedure was aborted. An examination of the separation channel (figure, right) demonstrates a mottled appearance from maroon-colored aggregates of agglutinated RBCs. While concern was raised whether the hot packs used on the extracorporeal tubing contributed to the hemolysis, the maximum observed temperature of a heat pack wrapped over a thermometer was 104 F (per manufacturer, maximal temperature of 1108F/ 438C may be achieved). Additionally, a blood specimen collected from a healthy individual was wrapped with a hot pack for 60 minutes; no hemolysis was observed. This illustrates the potential technical difficulties associated with plasma exchange for patients with cold agglutinins, although successful exchanges in similar scenarios have been documented. 1 Additional attempts at plasma exchange were not undertaken and the patient's clinical picture progressively deteriorated over the next several weeks until care was withdrawn. CONFLICT OF INTERESTThe authors have disclosed no conflicts of interest. REFERENCE 1. Andrzejewski C, Gault E, Briggs M, et al. Benefit of a 378C extracorporeal circuit in plasma exchange therapy for selected cases with cold agglutinin disease. J Clin Apheresis. 1988;4:13-17.
We have developed a human fetal digit model of response to tip amputation to simulate the clinical phenomenon of digit tip regrowth or regeneration in children. We reported previously that younger estimated gestational age (EGA) digits respond to tip amputation with several layers of cells covering the phalangeal anlage, compared to older digits where such coverage comprises fewer cell layers or is absent. The proximal cut end of excised digits does not demonstrate such a response. We report here that the transcription repressor MSX1 is expressed beneath the developing nailfield in younger human fetal digits (57 to 67 days EGA), as has been shown in the mouse, and that cells expressing MSX1 are found at the digit tip by 4 days postamputation. Further, expression of keratins K14 and K19 is found, suggesting reforming of an epithelium. These findings are consistent with the mouse model of digit tip regeneration where MSX1‐expressing cells are hypothesized to migrate to the amputation site and reconstitute the missing tip, and with the general observation that a wound epidermis is required for either regeneration or wound healing. 57 d EGA digits; left, MSX1 expression, control; left center, MSX1 and center, K19 expression, day 4 posttip amputation; right center, MSX1 and right, K19 expression, day 7 postamputation
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