Case Reports in Transplantation The latest articles from Hindawi Publishing Corporation © 2016 , Hindawi Publishing Corporation . All rights reserved. Successful Stem Cell Transplantation in a Patient with Pretransplant Hepatic Inflammatory Pseudotumour Mon, 28 Nov 2016 07:54:03 +0000 Inflammatory pseudotumours (IPT) are rare benign neoplasms of unknown aetiology. We present a case of hepatic IPT which was incidentally discovered in a patient with relapsed B-acute lymphoblastic leukaemia (B-ALL) undergoing pretransplant workup. After investigation to exclude an infective cause she underwent a reduced intensity conditioning stem cell transplant (SCT) successfully and currently remains well and in remission. On repeat liver MRI after SCT, the IPT was seen to be resolving. To the best of our knowledge this is the first report of an adult patient with hepatic IPT successfully undergoing SCT. The reduction in size of the IPT after SCT also suggests an inflammatory rather than an infective aetiology for IPT. Sanjay de Mel, Zarni Soe, Benjamin Wong, Ching Ching Ong, Lynette Teo, and Lip Kun Tan Copyright © 2016 Sanjay de Mel et al. All rights reserved. ST Elevation Infarction after Heart Transplantation Induced by Coronary Spasms and Mural Thrombus Detected by Optical Coherence Tomography Thu, 17 Nov 2016 09:38:23 +0000 The case illustrates the possible link between coronary spasms, intraluminal thrombus formation, and widespread organized and layered thrombi in HTx patients. Furthermore, the case underlines the clinical value of OCT as a novel method for high-resolution vessel imaging in heart-transplanted (HTx) patients with coronary spasms and suspected coronary artery disease. Coronary spasms and sudden death are frequent complications after HTx. The underlying mechanisms leading to these complications are unknown. The present case displays the clinical course of a 19-year-old HTx patient who was hospitalized due to acute myocardial infarction induced by severe coronary spasms. The patients remained unstable on conservative therapy. Therefore, an optical coherence tomography (OCT) was performed and revealed massive, organized thrombi in the left main coronary artery, the circumflex coronary artery, and the left anterior descending coronary artery. The patient was stabilized after percutaneous coronary intervention. As a mural thrombus often goes undetected by coronary angiography, OCT may prove benefit in HTx patients with myocardial infarction or suspected coronary spasms. Tor Skibsted Clemmensen, Niels Ramsing Holm, Hans Eiskjær, Steen Hvitfeldt Poulsen, Michael Maeng, Christian Juhl Terkelsen, and Evald Høj Christiansen Copyright © 2016 Tor Skibsted Clemmensen et al. All rights reserved. Massive Ascites in a Renal Transplant Patient after Laparoscopic Fenestration of a Lymphocele Mon, 07 Nov 2016 13:54:38 +0000 Retroperitoneal lymphocele is a common complication of renal transplantation. Here, we report the case of a 67-year-old woman with massive ascites after fenestration surgery for a lymphocele that developed following renal transplantation. She had been on continuous ambulatory peritoneal dialysis for 9 years. Living donor renal transplantation was performed and an intrapelvic lymphocele subsequently developed. The lymphocele did not resolve after aspiration therapy; therefore, laparoscopic fenestration was performed. Although the lymphocele disappeared, massive ascites appeared in its stead. Half a year later, the ascites was surgically punctured, which then gradually resolved and disappeared 6 weeks later. Aspiration therapy should be considered in patients on long-term peritoneal dialysis, although laparoscopic fenestration is safe and effective. Shohei Kawaguchi, Takahiro Nohara, Takashi Shima, Satoko Matsuyama, Chikako Nose, Junya Yamahana, Yoshifumi Kadono, Chikashi Seto, Masahiko Kawabata, and Atsushi Mizokami Copyright © 2016 Shohei Kawaguchi et al. All rights reserved. Fatal Donor-Derived Carbapenem-Resistant Klebsiella pneumoniae Infection in a Combined Kidney-Pancreas Transplantation Sun, 16 Oct 2016 08:03:32 +0000 Carbapenem-resistant Klebsiella pneumoniae (CR-KP) infections in solid organ transplant recipients are associated with high morbidity and mortality. We report a case of a fatal donor-derived CR-KP infection in a combined kidney-pancreas transplant. Given the short interval of time between donor hospitalization and organ procurement, information concerning the donor CR-KP positivity arrived only 72 hours after transplant. Based on this experience, we believe that knowledge of the donor’s CR-KP status should be mandatory before procurement and, if positive, pancreas donation should be contraindicated. Giovanni Varotti, Ferdinando Dodi, Anna Marchese, Alessia Terulla, Massimo Bertocchi, and Iris Fontana Copyright © 2016 Giovanni Varotti et al. All rights reserved. Immune Complex Mediated Glomerulonephritis with Acute Thrombotic Microangiopathy following Newly Detected Hepatitis B Virus Infection in a Kidney Transplant Recipient Sun, 09 Oct 2016 14:06:06 +0000 Hepatitis B virus (HBV) presents a risk to patients and staff in renal units. To minimise viral transmission, there are international and UK guidelines recommending HBV immunisation for patients commencing renal replacement therapy (RRT) and HBV surveillance in kidney transplant recipients. We report the case of a 56-year-old male who was immunised against HBV before starting haemodialysis. He received a deceased donor kidney transplant three years later, at which time there was no evidence of HBV infection. After a further six years he developed an acute kidney injury; allograft biopsy revealed an acute thrombotic microangiopathy (TMA) with glomerulitis, peritubular capillaritis, and C4d staining. Due to a “full house” immunoprofile, tests including virological screening were undertaken, which revealed acute HBV infection. Entecavir treatment resulted in an improvement in viral load and kidney function. HBV genotyping demonstrated a vaccine escape mutant, suggesting “past resolved” infection that reactivated with immunosuppression, though posttransplant acquisition cannot be excluded. This is the first reported case of acute HBV infection associated with immune complex mediated glomerulonephritis and TMA. Furthermore, it highlights the importance of HBV surveillance in kidney transplant recipients, which although addressed by UK guidelines is not currently practiced in all UK units. Tracey Salter, Hannah Burton, Sam Douthwaite, William Newsholme, Catherine Horsfield, and Rachel Hilton Copyright © 2016 Tracey Salter et al. All rights reserved. Strongyloides Hyperinfection Syndrome Combined with Cytomegalovirus Infection Thu, 15 Sep 2016 12:21:30 +0000 The mortality in Strongyloides hyperinfection syndrome (SHS) is alarmingly high. This is particularly common in bone marrow, renal, and other solid organ transplant (SOT) patients, where figures may reach up to 50–85%. Immunosuppressives, principally corticosteroids, are the primary triggering factor. In general, the clinical features of Strongyloides stercoralis hyperinfection are nonspecific; therefore, a high index of suspicion is required for early diagnosis and starting appropriate therapy. Although recurrent Gram-negative sepsis and meningitis have been previously reported, the combination of both cytomegalovirus (CMV) and strongyloidiasis had rarely been associated. We here describe a patient who survived SHS with recurrent Escherichia coli (E. coli) urosepsis and CMV infection. Fatehi Elnour Elzein, Mohammed Alsaeed, Sulafa Ballool, and Ashraf Attia Copyright © 2016 Fatehi Elnour Elzein et al. All rights reserved. High on Cannabis and Calcineurin Inhibitors: A Word of Warning in an Era of Legalized Marijuana Tue, 09 Aug 2016 07:48:46 +0000 Tacrolimus, a potent immunosuppressant medication, acts by inhibiting calcineurin, which eventually leads to inhibition of T-cell activation. The drug is commonly used to prevent graft rejection in solid organ transplant and graft-versus-host disease in hematopoietic stem cell transplant patients. Tacrolimus has a narrow therapeutic index with variable oral bioavailability and metabolism via cytochrome P-450 3A enzyme. Toxicity can occur from overdosing or from drug-drug interactions with the simultaneous administration of cytochrome P-450 3A inhibitors and possibly P-glycoprotein inhibitors. Tacrolimus toxicity can be severe and may include multiorgan damage. We present a case of suspected tacrolimus toxicity in a postallogeneic hematopoietic stem cell transplant patient who was concurrently using oral marijuana. This case represents an important and growing clinical scenario with the increasing legalization and use of marijuana throughout the United States. Naomi Hauser, Tanmay Sahai, Rocco Richards, and Todd Roberts Copyright © 2016 Naomi Hauser et al. All rights reserved. Preserved Renal Function in Kidney Transplantation over a Thrombosed Aortobifemoral Bypass Graft: The Role of Retrograde Flow and Early Thrombolysis Thu, 04 Aug 2016 13:18:32 +0000 Aortobifemoral bypass (ABFB) thrombosis is not uncommon, and when the artery of a renal graft is implanted on a bypass the risk of graft loss is high. We report the case of a 48-year-old woman with a previous history of ABFB under antiplatelet therapy and a kidney allograft implanted on the vascular prosthesis, who presented with acute limb ischemia and severe renal impairment. Imaging techniques revealed a complete thrombosis of the proximal left arm of the ABFB. However, a faint retrograde flow over the graft was observed thanks to the recanalization of distal left bypass by collateral native arteries. This unusual situation not previously reported in a kidney transplant setting, together with an early diagnosis, allowed graft survival until an early local thrombolysis resolved the problem. Two years later, renal function remains normal. Saúl Pampa-Saico, Sara Jiménez-Alvaro, Fernando Caravaca-Fontán, Ana Fernández-Rodríguez, Maite Rivera-Gorrín, Juan Sánchez, Antonio Chinchilla, and Roberto Marcén Copyright © 2016 Saúl Pampa-Saico et al. All rights reserved. Eculizumab for the Treatment of Severe Antibody-Mediated Rejection: A Case Report and Review of the Literature Sun, 10 Jul 2016 12:43:51 +0000 In renal transplantation, treatment options for antibody-mediated rejection are limited. Here, we report a case of severe AMR treated with eculizumab. A 50-year-old woman known for end stage kidney disease secondary to IgA nephropathy received a kidney transplant from a 50-year-old deceased donor. At 5 months after transplantation, she presented with acute graft dysfunction and biopsy showed a severe antibody-mediated rejection associated with thrombotic microangiopathy. Despite an aggressive conventional immunosuppressive regimen, signs of rejection persisted and the patient was treated with 3 doses of eculizumab. Following the therapy, markers of TMA improved and graft function stabilized. However, ongoing signs of rejection remained in the repeated biopsy. In kidney transplantation, eculizumab is an expensive treatment and its role in the treatment of antibody-mediated rejection remains to be determined. Duy Tran, Anne Boucher, Suzon Collette, Alexis Payette, Virginie Royal, and Lynne Senécal Copyright © 2016 Duy Tran et al. All rights reserved. Rapid Resolution of Cholangitic Abscess and Biliary Sepsis in a Liver Transplant Recipient after Hepatic Artery Revascularization Wed, 01 Jun 2016 08:02:10 +0000 Hepatic arterial flow is paramount in preserving biliary integrity. We present an interesting clinical scenario of a liver transplant recipient with biliary anastomotic stricture who developed biliary abscess and sepsis after Endoscopic Retrograde Cholangiopancreatography. The abscess did not respond to maximal medical management, percutaneous drainage, and adequate endoscopic biliary drainage. Clinically, patient continued to deteriorate and imaging identified hepatic artery stenosis which was treated with percutaneous intra-arterial stenting. Revascularization and perfusion of infected area led to rapid resolution of abscess and sepsis. This case emphasizes the anatomic basis of biliary ductal pathology. An important educational point is to understand that interrupted hepatic arterial supply can lead to biliary complications in liver transplant recipients and early correction of perfusion deficit should be pursued in such cases. In nonresolving hepatobiliary infections after liver transplantation, hepatic arterial compromise should be looked for and if present promptly treated. Reperfusion of biliary system in our patient led to improved antibiotics penetration, resolution of abscess and sepsis, and healing of biliary stricture. Sushrut Trakroo and Kamran Qureshi Copyright © 2016 Sushrut Trakroo and Kamran Qureshi. All rights reserved. Early Posttransplant Isolated v1 Lesion Does Not Need to Be Treated and Does Not Lead to Increased Fibrosis Tue, 17 May 2016 09:44:45 +0000 Acute vascular rejection (AVR) is characterized by intimal arteritis in addition to tubulitis and interstitial inflammation. It is associated with a poorer prognosis compared to tubulointerstitial rejection (AIR) and AVR is associated with a higher rate of graft loss than AIR. The prognosis and treatment of arteritis without tubulitis and interstitial inflammation (isolated v1 lesion) are still controversial. We report a case of a patient who had a biopsy of the kidney allograft for evaluation of slow graft function. The biopsy revealed an isolated v1 lesion. However, we chose not to augment immunosuppression. The patient’s kidney allograft function improved over time with close monitoring. Repeat biopsy a year later showed no evidence of endothelialitis and relatively unchanged fibrosis and no other abnormalities. Although it is suggested that most cases of isolated v1 lesions will respond to corticosteroids or T cell depleting therapies, some cases will improve with conservative management. Further studies are needed to determine which cases could be managed conservatively. Irfan Moinuddin, Bijin Thajudeen, Amy Sussman, Machaiah Madhrira, Erika Bracamonte, Mordecai Popovtzer, and Pradeep V. Kadambi Copyright © 2016 Irfan Moinuddin et al. All rights reserved. Application of the Liver Maximum Function Capacity Test in Acute Liver Failure: A Helpful Tool for Decision-Making in Liver Transplantation? Wed, 04 May 2016 11:42:20 +0000 Background. Despite aggressive intensive medical management acute liver failure (ALF) may require high-urgency liver transplantation (LTx). Available prognostic scores do not apply for all patients; reliable tools to identify individuals in need of LTx are highly required. The liver maximum function capacity test (LiMAx) might represent an appropriate option. Referring to a case of ALF after Amanita phalloides-intoxication the potential of the LiMAx-test in this setting is discussed. Presentation of Case. LiMAx was performed in a 27-year-old patient prior to and after high-urgency LTx. In accordance with clinical appearance of hepatic encephalopathy, coagulopathy, and acute kidney failure, the LiMAx-test constituted a fulminant course of ALF with hardly any detectable metabolic activity. Following LTx with a marginal donor organ (95% hepatosteatosis), uptake of liver function was demonstrated by postoperative increase of the LiMAx-value. The patient was discharged from hospital on postoperative day 26. Discussion. ALF often is associated with a critical state of the patient that requires almost immediate decision-making regarding further therapy. Application of a noninvasive liver function test might help to determine the prognosis of ALF and support decision-making for or against LTx as well as acceptance of a critical donor organ in case of a critically ill patient. Florian Wolfgang Rudolf Vondran, Carsten Schumacher, Kai Johanning, Björn Hartleben, Wolfgang Knitsch, Olaf Wiesner, Elmar Jaeckel, Michael Peter Manns, Juergen Klempnauer, Hueseyin Bektas, and Frank Lehner Copyright © 2016 Florian Wolfgang Rudolf Vondran et al. All rights reserved. First Autologous Cord Blood Therapy for Pediatric Ischemic Stroke and Cerebral Palsy Caused by Cephalic Molding during Birth: Individual Treatment with Mononuclear Cells Sat, 30 Apr 2016 11:06:36 +0000 Intracranial laceration due to traumatic birth injury is an extremely rare event affecting approximately one newborn per a population of 4.5 million. However, depending on the mode of injury, the resulting brain damage may lead to lifelong sequelae, for example, cerebral palsy for which there is no cure at present. Here we report a rare case of neonatal arterial ischemic stroke and cerebral palsy caused by fetal traumatic molding and parietal depression of the head during delivery caused by functional cephalopelvic disproportion due to a “long pelvis.” This patient was treated by autologous cord blood mononuclear cells (45.8 mL, cryopreserved, TNC ) with a remarkable recovery. Active rehabilitation was provided weekly. Follow-up examinations were at 3, 18, 34, and 57 months. Generous use of neonatal head MRI in case of molding, craniofacial deformity, and a sentinel event during parturition is advocated to enhance diagnosis of neonatal brain damage as a basis for fast and potentially causative treatment modalities including autologous cord blood transplantation in a timely manner. A. Jensen and E. Hamelmann Copyright © 2016 A. Jensen and E. Hamelmann. All rights reserved. False Positive B-Cells Crossmatch after Prior Rituximab Exposure of the Kidney Donor Thu, 28 Apr 2016 11:38:29 +0000 Crossmatching is essential prior to kidney transplantation to confirm compatibility between the donor and the recipient, particularly to prevent acute antibody-mediated rejection. An unexpected positive crossmatch may be obtained in recipients with an autoimmune disease or preexisting antibodies not detected by single-antigen bead array due to complement interference or who have been previously treated by desensitization protocols such as rituximab, antithymocyte globulin, or intravenous immunoglobulins. We report donor and recipient investigations that revealed unexpected positive B-cells crossmatch, probably due to donor cells, as the donor had received rituximab therapy shortly before organ harvesting, in a context of severe idiopathic thrombocytopenic purpura. We consequently detected unexpected Class II IgG complement-dependent cytotoxicity for all sera tested. Other laboratory investigations failed to elucidate the reasons for this recipient-related positivity. Judith Desoutter, Marie-Joëlle Apithy, Ségolène Bartczak, and Nicolas Guillaume Copyright © 2016 Judith Desoutter et al. All rights reserved. Lung Transplant Recipient with Pulmonary Alveolar Proteinosis Sun, 24 Apr 2016 08:40:17 +0000 Pulmonary alveolar proteinosis (PAP) is a progressive lung disease characterized by accumulated surfactant-like lipoproteinaceous material in the alveoli and distal bronchioles. This accumulation is the result of impaired clearance by alveolar macrophages. PAP has been described in 11 solid organ transplant recipients, 9 of whom were treated with mammalian target of rapamycin inhibitors. We report a case of a lung transplant recipient treated with prednisone, mycophenolate mofetil (MMF), and tacrolimus who ultimately developed PAP, which worsened when MMF was replaced with everolimus. Sofya Tokman, M. Frances Hahn, Hesham Abdelrazek, Tanmay S. Panchabhai, Vipul J. Patel, Rajat Walia, and Ashraf Omar Copyright © 2016 Sofya Tokman et al. All rights reserved. Different Management Options for Transplant Ureteral Obstructions within an Inguinal Hernia Sun, 10 Apr 2016 08:20:34 +0000 Ureteral obstruction secondary to an inguinal hernia with transplant ureteral component is an extremely rare entity with only several case reports found in literature. In all previously reported cases, management of the obstruction involved temporary drainage with ureteral stenting or nephrostomy tube placements followed by delayed definitive repair. We present two case reports, here one being the first one managed by immediate definitive repair via ureteral reimplant and herniorrhaphy and a second case by delayed definitive repair after percutaneous nephrostomy tube placement. Both patients continued to do well postoperatively with normalization of renal function on follow-up. Felix Cheung, Merrit Marion Debartolo, Leonard Michael Copertino, April Adams Szafran, Chelsea Caron Estrada, Patrick Gerard Lynch, and Frank Sam Darras Copyright © 2016 Felix Cheung et al. All rights reserved. Successful Treatment of Focal Segmental Glomerulosclerosis after Kidney Transplantation with Plasma Exchange and Abatacept in a Patient with Juvenile Rheumatoid Arthritis Sun, 20 Mar 2016 13:51:15 +0000 Recurrent focal segmental glomerulosclerosis (FSGS) after renal transplantation is difficult to treat. Recently a series of four patients unresponsive to plasma exchange (PE) and rituximab, who were successfully treated with abatacept, has been reported. We present a 26-year-old Caucasian patient who suffered from juvenile rheumatoid arthritis and developed severe proteinuria eleven days after transplantation. An allograft biopsy was suggestive of recurrent focal segmental glomerulosclerosis. He did not respond to PE therapy. A first dose of abatacept produced partial remission. Four weeks later proteinuria again increased and a second biopsy showed progression of disease. After another ineffective course of PE he was given a second dose of abatacept, which was followed by rapid, complete, and sustained resolution of proteinuria. This treatment caused a significant increase in BK and JC viremia. Whether abatacept ameliorated proteinuria via an effect on podocytes or on the patient’s primary disease remains speculative. Hannelore Sprenger-Mähr, Emanuel Zitt, Afschin Soleiman, and Karl Lhotta Copyright © 2016 Hannelore Sprenger-Mähr et al. All rights reserved. Treatment of Cytomegalovirus Infection with Cidofovir and CMV Immune Globulin in a Lung Transplant Recipient Wed, 13 Jan 2016 13:07:39 +0000 Cytomegalovirus (CMV) infection after lung transplantation is associated with increased risk for pneumonitis and bronchiolitis obliterans as well as allograft rejection and opportunistic infections. Ganciclovir is the mainstay of prophylaxis and treatment but CMV infections can be unresponsive. Apart from direct antiviral drugs, CMV immunoglobulin (CMVIG) preparations may be considered but are only licensed for prophylaxis. A CMV-seronegative 42-year-old man with cystic fibrosis received a lung from a CMV-seropositive donor. Intravenous ganciclovir prophylaxis was delayed until day 12 due to acute postoperative renal failure and was accompanied by five doses of CMVIG (10 g). By day 16, CMV-DNA was detectable and rising; CMV-specific T-cells were undetectable. Switch from ganciclovir to foscarnet prompted a transient decrease in CMV viral load, but after increasing again to reach 3600 copies/mL foscarnet was changed to intravenous cidofovir and CMVIG was restarted. CMV load continued to fluctuate and declined slowly, whereas CMV-specific T-cells were detected five months later and increased thereafter. At last follow-up, the patient was in very good clinical condition with no evidence of bronchiolitis obliterans. No side effects of this treatment were observed. In this hard-to-treat case, the combination of cidofovir with off-label use of CMVIG contributed to a successful outcome. Heinrike Wilkens and Martina Sester Copyright © 2016 Heinrike Wilkens and Martina Sester. All rights reserved. Septic Shock Induced by Bacterial Prostatitis with Morganella morganii subsp. morganii in a Posttransplantation Patient Mon, 21 Dec 2015 11:54:52 +0000 Bacterial infection is a common complication after Hematopoietic Stem Cell Transplantation (HSCT). Morganella morganii is ubiquitous Gram-negative facultative anaerobe, which may cause many kinds of opportunistic infection. Herein we report a case of a 55-year-old man who presented with frequent urination, urgency, and mild pain that comes and goes low in the abdomen and around the anus. The patient had a medical history of chronic prostatitis for 4 years. He received HLA-matched sibling allo-HSCT because of angioimmunoblastic T-cell lymphoma 29 months ago. The routine examination of prostatic fluid showed increased leukocytes and the culture of prostatic fluid showed Morganella morganii subsp. morganii. The patient developed chills and fever 18 hours after examination. Both urine culture and blood culture showed Morganella morganii subsp. morganii. The patient was successfully treated with antibiotic therapy and septic shock management. Taken together, Morganella morganii should be considered a possible pathogen when immunocompromised patients develop prostatitis. Also, prostatic massage could be a possible trigger of septic shock induced by Morganella morganii subsp. morganii in a posttransplantation patient. Xiaofan Li and Jianhui Chen Copyright © 2015 Xiaofan Li and Jianhui Chen. All rights reserved. Gastrointestinal Bleeding and Diffuse Skin Thickening as Kaposi Sarcoma Clinical Presentation Thu, 10 Dec 2015 12:20:36 +0000 A 56-year-old African patient received a kidney from a deceased donor with 4 HLA mismatches in April 2013. He received immunosuppression with basiliximab, tacrolimus, mycophenolate mofetil, and prednisone. Immediate diuresis and a good allograft function were soon observed. Six months later, the serum creatinine level increased to 2.6 mg/dL. A renal allograft biopsy revealed interstitial fibrosis and tubular atrophy grade II. Toxicity of calcineurin inhibitor was assumed and, after a switch for everolimus, renal function improved. However, since March 2014, renal function progressively deteriorated. A second allograft biopsy showed no new lesions. Two months later, the patient was admitted due to anuria, haematochezia with anaemia, requiring 5 units of packed red blood cells, and diffuse skin thickening. Colonoscopy showed haemorrhagic patches in the colon and the rectum; histology diagnosis was Kaposi sarcoma (KS). A skin biopsy revealed cutaneous involvement of KS. Rapid clinical deterioration culminated in death in June 2014. This case is unusual as less than 20 cases of KS with gross gastrointestinal bleeding have been reported and only 6 cases had the referred bleeding originating in the lower gastrointestinal tract. So, KS should be considered in differential diagnosis of gastrointestinal bleeding in some kidney transplant patients. Sara Querido, Henrique Silva Sousa, Tiago Assis Pereira, Rita Birne, Patrícia Matias, Cristina Jorge, André Weigert, Teresa Adragão, Margarida Bruges, and Domingos Machado Copyright © 2015 Sara Querido et al. All rights reserved. Utility of Iron Staining in Identifying the Cause of Renal Allograft Dysfunction in Patients with Sickle Cell Disease Tue, 01 Dec 2015 09:28:03 +0000 Sickle cell nephropathy (SCN) is associated with iron/heme deposition in proximal renal tubules and related acute tubular injury (ATI). Here we report the utility of iron staining in differentiating causes of renal allograft dysfunction in patients with a history of sickle cell disease. Case 1: the patient developed acute allograft dysfunction two years after renal transplant. Her renal biopsy showed ATI, supported by patchy loss of brush border and positive staining of kidney injury molecule-1 in proximal tubular epithelial cells, where diffuse increase in iron staining (2+) was present. This indicated that ATI likely resulted from iron/heme toxicity to proximal tubules. Electron microscope confirmed aggregated sickle RBCs in glomeruli, indicating a recurrent SCN. Case 2: four years after renal transplant, the patient developed acute allograft dysfunction and became positive for serum donor-specific antibody. His renal biopsy revealed thrombotic microangiopathy (TMA) and diffuse positive C4d stain in peritubular capillaries. Iron staining was negative in the renal tubules, implying that TMA was likely associated with acute antibody-mediated rejection (AAMR, type 2) rather than recurrent SCN. These case reports imply that iron staining is an inexpensive but effective method in distinguishing SCN-associated renal injury in allograft kidney from other etiologies. Yingchun Wang, Mona Doshi, Salman Khan, Wei Li, and Ping L. Zhang Copyright © 2015 Yingchun Wang et al. All rights reserved. Treatment of Recurrent Posttransplant Lymphoproliferative Disorder with Autologous Blood Stem Cell Transplant Wed, 25 Nov 2015 12:49:46 +0000 Background. Posttransplant lymphoproliferative disorders (PTLDs) occur after solid organ transplantation. Treatment guidelines include reduction in immunosuppression (RIS), radiation, rituximab, chemotherapy, and immunological agents. We present a rare case of recurrent diffuse large B-cell lymphoma presenting as a PTLD in a heart transplant patient treated with autologous blood stem cell transplant (ASCT) after failure of conventional therapy. Case Presentation. A 66-year-old male presented with a neck mass. He has a history of Hodgkin’s disease status after staging laparotomy with splenectomy and heart transplantation due to dilated nonischemic cardiomyopathy 8 years prior to the development of PTLD. His examination was remarkable for right submandibular swelling. An excisional biopsy confirmed the diagnosis of diffuse large B-cell NHL. Patient received RIS, rituximab, chemotherapy, and radiation therapy with a complete remission. His lymphoma relapsed and he subsequently was treated with RICE salvage chemotherapy and consolidative high-dose chemotherapy with BEAC regimen followed by ASCT resulting in a complete remission. Conclusion. Patients with PTLD present a difficult therapeutic challenge. In this case, the patient’s prior history of Hodgkin’s disease, splenectomy, and a heart transplant appear to be unique features, the significance of which is unclear. ASCT might be a promising therapy for patients with relapsed or refractory PTLD. Bharat Malhotra, Ahmad K. Rahal, Hussam Farhoud, Dennis F. Moore Jr., and K. James Kallail Copyright © 2015 Bharat Malhotra et al. All rights reserved. ELISA-Based Crossmatching Allowing the Detection of Emerging Donor-Specific Anti-HLA Antibodies through the Use of Stored Donors’ Cell Lysates Sun, 08 Nov 2015 12:42:49 +0000 About forty years ago the complement-dependent crossmatch assay (CDC-CM) was developed as standard procedure in order to select recipients without donor-specific antibodies directed against human leukocyte antigens of their given donors since the negative outcome of pretransplant crossmatching represents one of the most important requirements for a successful kidney graft survival. However, as a functional assay the CDC-CM strongly depends on the availability of donors’ isolated lymphocytes and in particular on their vitality highly limiting its applicability for recipients treated with special drugs and therapeutic antibodies or suffering from underlying autoimmune diseases. In the great majority of these cases ELISA-based crossmatching has been demonstrated to be an adequate alternative procedure nevertheless leading to valid results. With these case reports we show for the first time that ELISA-based crossmatching is suitable to demonstrate the upcoming donor-specific anti-HLA antibodies as a consequence of allografting using deep-frozen deceased donor’s material such as blood or spleen detergent lysate. Thus, this ELISA-based procedure first provides the option to routinely perform crossmatching using stored material of deceased donors in order to substitute or at least to complement virtual crossmatching, that is, the comparison of the recipients’ anti-HLA antibody specificities with the donors’ historically identified HLA types. G. Schlaf, K. Stöhr, A. Rothhoff, and W. Altermann Copyright © 2015 G. Schlaf et al. All rights reserved. Premalignant and Malignant Skin Lesions in Two Recipients of Vascularized Composite Tissue Allografts (Face, Hands) Tue, 13 Oct 2015 07:07:23 +0000 Recipients of solid organ transplants (RSOT) have a highly increased risk for developing cutaneous premalignant and malignant lesions, favored by the lifelong immunosuppression. Vascularized composite tissue allografts (VCA) have been introduced recently, and relevant data are sparse. Two patients with skin cancers (one with basal cell carcinoma and one with squamous cell carcinomas) have been so far reported in this patient group. Since 2000 we have been following 9 recipients of VCA (3 face, 6 bilateral hands) for the development of rejection and complications of the immunosuppressive treatment. Among the 9 patients, one face-grafted recipient was diagnosed with nodular-pigmented basal cell carcinoma of her own facial skin 6 years after graft, and one patient with double hand allografts developed disseminated superficial actinic porokeratosis, a potentially premalignant dermatosis, on her skin of the arm and legs. Similar to RSOT, recipients of VCA are prone to develop cutaneous premalignant and malignant lesions. Prevention should be applied through sun-protective measures, regular skin examination, and early treatment of premalignant lesions. Jean Kanitakis, Palmina Petruzzo, Aram Gazarian, Sylvie Testelin, Bernard Devauchelle, Lionel Badet, Jean-Michel Dubernard, and Emmanuel Morelon Copyright © 2015 Jean Kanitakis et al. All rights reserved. Absent Right Side Iliac Arterial System, an Intraoperative Surprise during Live Related Recipient Renal Transplantation Tue, 11 Aug 2015 11:36:15 +0000 Introduction. Renal transplantation has become the standard of care for patients with end stage renal disease. We present a rare case of an absent right sided iliac arterial system encountered during recipient renal transplantation. The presence of such vascular anomaly intraoperatively can present a technically challenging situation to the surgeon. Case Presentation. During a routine renal transplantation of a 34-year-old man, we encountered a complete absence of right side iliac arterial system and a prominent branch arising from left hemipelvis and coursing to the right lower limb and the urinary bladder. The artery to the bladder was divided and anastomosed end to end to the donor renal artery. Intraoperatively the renal perfusion and the urine output were good. A posttransplant magnetic resonance angiogram done six weeks later revealed good vascular supply to the kidney and the lower limb. Conclusion. Absent iliac artery on one or both sides is a rare phenomenon. The presence of it during an unanticipated renal transplant surgery can pose a significant technical challenge to the surgeons. We advocate routine assessment of pelvic vasculature before recipient renal transplant surgery so as to avoid a difficult situation like this. Vijay Radhakrishnan, Rana Kumar, Datson George, and G. P. Abraham Copyright © 2015 Vijay Radhakrishnan et al. All rights reserved. Unusual Causes of Abrupt Anuria Early Post-Renal Transplantation Mon, 13 Jul 2015 11:27:54 +0000 Renal transplantation using living donors has superior outcomes in comparison to deceased donor transplantation and results in immediate allograft function in a majority of cases. Rarely may allograft be nonfunctional from the beginning, or anuria is noted after a period of good urine output. Surgical causes for anuria should be high on the differential diagnosis in immediate-to-early posttransplant period, especially in an unsensitized recipient. We present two unusual causes of early onset anuria after living related renal transplantation where early surgical reexploration salvaged renal allografts with excellent long term outcomes. Gurudev Konana Chennabasappa, Sonika Puri, Vijay Varma, and Mahesh Eswarappa Copyright © 2015 Gurudev Konana Chennabasappa et al. All rights reserved. Anti-Erythropoietin Antibody Associated Pure Red Cell Aplasia Resolved after Liver Transplantation Thu, 09 Jul 2015 09:48:07 +0000 Patients undergoing antiviral therapy for chronic hepatitis C often develop anemia secondary to ribavirin and interferon. Recombinant erythropoietin has been used to improve anemia associated with antiviral therapy and to minimize dose reductions, which are associated with decreased rates of sustained virologic response. A rare potential side effect of recombinant erythropoietin is anti-erythropoietin antibody associated pure red cell aplasia. In chronic kidney disease patients with this entity, there have been good outcomes associated with renal transplant and subsequent immunosuppression. In this case, a chronic liver disease patient developed anti-erythropoietin associated pure red cell aplasia and recovered after liver transplantation and immunosuppression. It is unclear whether it is the transplanted organ, the subsequent immunosuppression, or the combination that contributed to the response. In conclusion, anti-erythropoietin associated pure red cell aplasia is a serious complication of erythropoietin therapy, but this entity should not be considered a contraindication for solid organ transplantation. Annie K. Hung, Jennifer Guy, Caroline M. Behler, and Eugene E. Lee Copyright © 2015 Annie K. Hung et al. All rights reserved. Transplantation of Horseshoe Kidney from Living, Genetically Unrelated Donor Thu, 09 Jul 2015 07:08:39 +0000 We report a case of renal transplantation using a horseshoe kidney from a living, genetically unrelated donor. The recipient was a 60-year-old man with diabetic nephropathy, and the donor was the 63-year-old wife of the recipient with a horseshoe kidney free of complications. Computed tomography showed two renal arteries and one renal vein on the left side, and the isthmus was perfused by several accessory arteries and veins. To demarcate the boundary of the isthmus, the left renal artery was ligated and cannulated for in situ perfusion. Furthermore, the isthmus was clamped, and the boundary of the isthmus was confirmed. The kidney was divided at the left margin of the perfused boundary. The cut ends of the isthmus were closed by sutures. The left kidney was transplanted into the right iliac fossa of the recipient. Asymptomatic fluid collection occurred on the cut surface at the isthmus of the donor, and this fluid decreased in due course. On the other hand, the recipient experienced no surgical complication or rejection, while maintaining serum creatinine levels of 2.00–2.20 mg/dL over a 22-month follow-up period. Horseshoe kidneys may be used for transplantation in selected cases after a detailed preoperative evaluation. Kazuro Kikkawa, Takahito Wakamiya, Hiroki Kusumoto, Nagahide Matsumura, Yasuo Kohjimoto, and Isao Hara Copyright © 2015 Kazuro Kikkawa et al. All rights reserved. Constrictive Pericarditis in the Presence of Remaining Remnants of a Left Ventricular Assist Device in a Heart Transplanted Patient Thu, 21 May 2015 12:31:50 +0000 Constrictive pericarditis (CP) is a severe subform of pericarditis with various causes and clinical findings. Here, we present the unique case of CP in the presence of remaining remnants of a left ventricular assist device (LVAD) in a heart transplanted patient. A 63-year-old man presented at the Heidelberg Heart Center outpatient clinic with progressive dyspnea, fatigue, and loss of physical capacity. Heart transplantation (HTX) was performed at another heart center four years ago and postoperative clinical course was unremarkable so far. Pharmacological cardiac magnetic resonance imaging (MRI) stress test was performed to exclude coronary ischemia. The test was negative but, accidentally, a foreign body located in the epicardial adipose tissue was found. The foreign body was identified as the inflow pump connection of an LVAD which was left behind after HTX. Echocardiography and cardiac catheterization confirmed the diagnosis of CP. Surgical removal was performed and the epicardial tubular structure with a diameter of 30 mm was carefully removed accompanied by pericardiectomy. No postoperative complications occurred and the patient recovered uneventfully with a rapid improvement of symptoms. On follow-up 3 and 6 months later, the patient reported about a stable clinical course with improved physical capacity and absence of dyspnea. R. Rivinius, M. Helmschrott, V. Koch, F. Sedaghat-Hamedani, P. Fortner, F. F. Darche, D. Thomas, A. Ruhparwar, B. Schmack, M. Karck, M. Akhavanpoor, C. Erbel, C. A. Gleissner, S. J. Buss, D. Mereles, P. Ehlermann, H. A. Katus, and A. O. Doesch Copyright © 2015 R. Rivinius et al. All rights reserved. Invasive Microascus trigonosporus Species Complex Pulmonary Infection in a Lung Transplant Recipient Thu, 14 May 2015 11:18:52 +0000 Because of the high incidence of morbidity and mortality associated with invasive fungal infections, antifungal prophylaxis is often used in solid organ transplant recipients. However, this prophylaxis is not universally effective and may contribute to the selection of emerging, resistant pathogens. Here we present a rare case of invasive infection caused by Microascus trigonosporus species complex in a human, which developed during voriconazole prophylaxis in a lung transplant recipient. Nebulized liposomal amphotericin B was used in addition to systemic therapy in order to optimize antifungal drug exposure; this regimen appeared to reduce the patient’s fungal burden. Despite this apparent improvement, the patient’s pulmonary status progressively declined in the setting of multiple comorbidities, ultimately leading to respiratory failure and death. Kelly E. Schoeppler, Martin R. Zamora, Noelle M. Northcutt, Gerard R. Barber, Gayle O’Malley-Schroeder, and Dennis M. Lyu Copyright © 2015 Kelly E. Schoeppler et al. All rights reserved.