Case report: gastrointestinal hemorrhage caused by a pancreas transplant arteriovenous fistula with large psuedoanuerysm 9 years after transplantation

Reported cases of arteriovenous fistulae in transplant recipients are uncommon. We present a case of an arteriovenous fistula associated with a large pseudoaneurysm in the root of the small bowel mesentery of a pancreas transplant. Uniquely, in our case, the arteriovenous fistula presented with an episode of gastrointestinal (GI) hemorrhage 9 years postoperatively. Radiographic imaging including coronal computed tomography angiogram and conventional angiogram demonstrated an arteriovenous fistula in the patient's pancreas transplant between the distal superior mesenteric artery (SMA) and superior mesenteric vein (SMV) with 6 cm aneurysmal dilatation. The tremendous flow in the fistula in the root of the graft small intestine mesentery led to graft duodenal mucosal congestion and lower GI hemorrhage. After successful embolization of the SMA–SMV fistula and pseudoaneurysm using interventional radiographic techniques, the arteriovenous fistula remained thrombosed. The patient had no further episodes of GI bleeding and her endoscopic evaluation was otherwise negative. The presence of arteriovenous fistulae and pseudoaneurysms in pancreas transplant recipients is uncommon, but has been previously documented. This case is further distinguished from previous reports by the notable 9-year interval between transplantation and the onset of hemorrhage. Historically, symptomatic vascular malformations have been associated with significant patient morbidity and mortality. Successful patient management involves timely and accurate diagnosis and intervention.     

Secondary hyperaldosteronism in a patient with polyarteritis nodosa and renal artery aneurysms

Polyarteritis nodosa (PAN) is a systemic necrotizing vasculitis that affects medium- and small-sized arteries. We report the case of a 32-year-old female with PAN in which renal involvement was revealed by a secondary hyperaldosteronism. Hypokaliemia and arterial hypertension preceded rupture of renal artery aneurysm by several months. We believe that hyperreninemia resulted from diffuse renal necrotizing vasculitis with occlusive but non-stenotic lesions. Angiography or CT scan should be performed systematically in PAN to screen for aneurysms so as to be able to consider prophylactic treatment by embolization and intensification of the general treatment. Hyperaldosteronism may reveal renal involvement in PAN and warrants an angiography if it has not yet been done.     

Subclinical renal involvement in essential cryoglobulinemic vasculitis and classic polyarteritis nodosa

ObjectiveRenal vasculitis is usually associated with anti-neutrophil cytoplasmic antibodies (ANCAs). However, non-ANCA patients constitute a rarely studied variant of renal vasculitis. The aim of the present study was to demonstrate the features of renal involvement in patients with primary systemic non-ANCA associated vasculitis (NAAV) and compare essential cryoglobulinemic vasculitis (ECV) with classic polyarteritis nodosa (PAN).MethodsThe study included 30 patients with primary systemic non-ANCA associated vasculitis (NAAV). Fifteen with ECV and another 15 patients with classic PAN. The patients were recruited from the Rheumatology and Internal medicine departments and outpatient clinics of Cairo University Hospitals. The patients had no or mild renal involvement at entry and the ANCA was negative as tested by immunoflourescence and ELISA. Renal biopsy was performed for all the patients and histopathologically studied.ResultsRenal biopsy abnormalities were seen in six females. One patient with PAN showed renal vasculitis and membranoproliferative glomerulonephritis (MPGN) and was HBV and ANA positive. The patient had negative HCV and cryoglobulins. Five patients with ECV-associated HCV had findings; one had chronic interstitial nephritis and was HBV positive. The other four were HBV negative with MPGN in two, focal proliferative and crescentic GN in one patient each.ConclusionsIncreased understanding of the manifestations of systemic vasculitis is likely to provide the basis for the use of more selective immunomodulatory therapies in the future. It is our hope that this study will raise awareness of the non ANCA-associated vasculitic renal involvement.     

Polyarteritis nodosa presenting as massive upper gastrointestinal hemorrhage

We report the first case of massive upper gastrointestinal hemorrhage as the initial presentation of polyarteritis nodosa (PAN), which is an uncommon form of systemic necrotizing vasculitis that may involve many organ systems and could affect any age group. Abdominal pain is the most common sign of gastrointestinal involvement. Gastrointestinal bleeding occurs less frequently in approximately 6% of cases. Reported cases of gastrointestinal hemorrhage have been in the form of coffee ground emesis, melena, or hematochezia. Such bleeding complications have resulted from ischemic ulceration or perforation of the small or large intestine. However, we are unaware of previous reports showing massive hematemesis to be the initial presentation of PAN.     

Colonic lymphoma as a cause of massive rectal bleeding in a cardiac transplant recipient

Lower gastrointestinal hemorrhage in immunosuppressed transplant recipients is associated with significant morbidity and mortality. This hemorrhage is often massive and frequently due to ulceration of the colon by opportunistic organisms requiring early diagnosis and resection. We report a heart transplant recipient with massive rectal bleeding from an unsuspected lymphoproliferative lesion of the sigmoid colon treated successfully with prompt surgical resection and reduction of immunosuppression. Although lymphoproliferative lesions are common in transplant recipients, particularly in those immunosuppressed with cyclosporine, we believe massive rectal bleeding to be a unique presentation of such a lesion.     

Surviving gastrointestinal infarction due to polyarteritis nodosa: a rare event.

Poly arteritis nodosa (PAN) is a systemic vasculitis with a male: female ratio of 2:1 and a peak incidence in the fifth decade. Small to medium-sized arteries are involved by focal transmural inflammatory necrosis. Aneurysms with inflammatory destruction of the media also occur. The most frequently involved organs are the kidney, heart, lung, liver, and gastrointestinal tract. There are few reported cases of ischemic necrosis of the intestine and even fewer survivors. A 22-year-old woman was transferred to St. Thomas Hospital (Nashville, TN) after resection of 80 per cent of the small bowel for ischemic necrosis. She had a history of juvenile onset diabetes mellitus, recurrent abdominal pain, and splinter hemorrhages. Emergency aortogram and selective mesenteric arteriogram were performed. The celiac artery was not visualized and small aneurysms were present in the mesenteric and renal arteries. The patient was successfully resuscitated from a cardiac arrest in x ray from a cardiac tamponade. Laparotomy was performed to determine the viability of the bowel. The celiac, hepatic, and splenic arteries were found to be chronically occluded. Pathology of these arteries revealed a nonspecific arteritis. At a third operation, several more inches of small bowel were removed. Characteristic changes of PAN were present on all small bowel specimens. She was treated with high-dose cyclophosphamide and steroids for 6 months and has continued on low-dose cyclophosphamide. She is now 36 months from her original operation and is doing well on oral nutrition. Intestinal hemorrhage from aneurysm rupture or gangrene with perforation are gastrointestinal complications of PAN that the surgeon may be called upon to treat.(ABSTRACT TRUNCATED AT 250 WORDS)     

Diffuse Alveolar Hemorrhage in a 39-year-old Woman: Unusual Initial Presentation of Microscopic Polyangiitis

Microscopic polyangiitis (MPA) is a necrotizing vasculitis involving the small vessels without granulomatous inflammation. Most MPA initially presents with renal involvement without pulmonary involvement. Isolated and initially presenting alveolar hemorrhage is very rare. The patient was a 39-year-old female with a progressive cough, dyspnea, and blood-tinged sputum for the previous 5 days. We determined that her condition was MPA though VATS lung biopsy and renal biopsy. After 2 months of steroid therapy, the chest lesions had improved. We report here a rare case of MPA with isolated and initial involvement of the lung with a review of the literature.     

Post-transplant Lymphoproliferative Disorder Presenting as Breast Nodule and Intestinal Obstruction in a Renal Transplant Recipient: A Case Report

Post-transplant lymphoproliferative disease (PTLD) is a life-threatening complication among kidney transplant recipients. The clinical presentation of patients with PTLD is highly variable. The type of PTLD and the area of involvement determine its presentation, which includes constitutional symptoms such as fever, weight loss, fatigue, and other symptoms related to dysfunction of involved organs, or compression of surrounding structures. Most present with extranodal masses involving the gastrointestinal tract (stomach, intestine), lungs, skin, liver, central nervous system, and the allograft itself. In our case, a 33-year-old woman developed Epstein-Barr virus–negative PTLD plasmablastic lymphoma (PbL) in her right breast and small intestine presenting as intestinal obstruction, 15 years after renal transplant. Her condition was managed with intestinal mass resection and chemotherapy. A follow-up positron emission tomography scan showed near-complete resolution. Thus, PTLD should always be kept in mind in renal transplant recipients who present with features of a mass effect involving any organ.     

Sequential Liver,Kidney, and Autologous Stem Cell Transplant for AL Amyloidosis: A Case Report

AL type amyloidosis is a systemic disease characterized by the accumulation of amyloid fibrils that can affect many organs such as the skin, gastrointestinal tract, heart, lungs, liver, and kidney. The most frequently involved organ in amyloidosis is the kidney, but cardiac amyloidosis with the poor prognosis is amyloid organ involvement. In this study, we present the treatment of a 40-year-old female patient with acute Budd-Chiari syndrome and very severe proteinuria with sequential liver, kidney, and autologous stem cell transplant after the diagnosis of systemic amyloidosis. To reduce the effects of massive proteinuria and very severe hypoalbuminemia, bilateral renal artery embolization was performed first. After the evaluation of the patient, she underwent liver transplant from a deceased donor, and then kidney transplant was performed from her son 1 month later. Afterward, the patient was discharged without any problems and underwent chemotherapy and stem cell transplant for primary AL amyloidosis. She was followed up without any problem in terms of liver, kidney, and stem cell at the 24th postoperative month. This case shows that autologous stem cell transplant after kidney and liver transplant may be a good treatment option in a selected patient with stem cell involvement diagnosed as having AL amyloidosis.     

Pulmonary Rhizopus infection in a diabetic renal transplant recipient

Infectious complications after renal transplantation remain a major cause of morbidity and mortality. Mucormycosis is a rare infection in renal transplant recipients; however, mortality is exceedingly high. Risk factors predisposing to this disease include prolonged neutropenia, diabetes, and patients who are immunosuppressed (Singh N, Gayowski T, Singh J, Yu LV. Invasive gastrointestinal zygomycosis in a liver transplant recipient: case report and review of zygomycosis in solid-organ transplant recipients, Clin Infect Dis 1995: 20: 617). Life-threatening infections can occur, as this fungus has the propensity to invade blood vessel endothelium, resulting in hematological dissemination. We report a case of cavitary Rhizopus lung infection, 2 months after renal transplantation, where the patient was treated successfully with Amphotericin B and surgical resection of the lesions with preservation of his allograft function. In this era of intensified immunosuppression, we may see an increased incidence of mucormycosis in transplant population. Invasive diagnostic work-up is mandatory in case of suspicion; Amphotericin B and, in selected cases, surgical resection are the mainstays of therapy.     

De Novo Lupus Nephritis in a Renal Transplanted Child: A Case Report

De novo lupus nephritis (LN) is a rare complication in renal transplantation recipients. We present the clinical manifestations of de novo LN in a 12-year-old boy who received a cadaveric renal transplant. The cause of end-stage renal disease was prune belly syndrome with renal dysplasia. His immunosuppressive drugs included tacrolimus, mycophenolate sodium, and prednisolone. After 3 years of treatment, he developed nephrotic syndrome (NS) without other symptoms of systemic lupus erythematosus (SLE). The renal pathology of the transplanted kidney showed suspicious acute cellular rejection and LN World Health Organization class IV-G (A/C). Antinuclear antibody was positive, but anti-dsDNA and anti-Smith were negative. The serum complements were initially normal. Pulse methylprednisolone was given and the dosages of all immunosuppressive drugs increased; notwithstanding, his edema and hypoalbuminemia worsened. Repeated biopsy of the transplanted kidney was done. A full-house pattern was documented under immunofluorescent examination which confirmed LN WHO class IV-G (A/C) without evidence of rejection. He then developed macrophage-associated hemophagocytic syndrome and cytomegalovirus pneumonia. He ultimately developed pulmonary hemorrhage and died owing to severe pneumonia. De novo LN should be considered in renal transplant recipients with new onset of NS despite there not being any other clinical manifestations of SLE.     

Unexpectedly high incidence of cytomegalovirus infection in apparent peptic ulcers in renal transplant recipients

Gastroduodenal ulcers in renal transplant recipients are usually accepted as being acid-peptic in origin. In a series of 573 renal transplant recipients there was histologic material available for examination from eight patients with gastroduodenal ulcers and three patients with gastric erosions. All ulcers had originally been diagnosed as peptic or stress ulcers. However, on review, five of them proved to contain cytomegalovirus (CMV); CMV was also present in all the stomachs with erosions. We suggest that CMV infection plays as important a role in upper gastroduodenal ulcers and erosions in renal transplant recipients as it does in similar lesions elsewhere in the gastrointestinal tract of these immunosuppressed patients.     

Bowel Obstruction after Biliopancreatic Diversion: A Deceptive Complication

Background: Although obesity surgery is now practiced in most of the world, many general surgeons, faced with an emergency, are not experienced in the diagnostic problems associated with these techniques, or about the most suitable treatment to resolve the acute pathology while preserving the weight loss. The biliopancreatic diversion (BPD), because of its complexity, could cause a delay in the diagnosis and therapy, with possible catastrophic consequences for the patient. Methods: We report 3 patients with bowel obstruction after BPD. In the first patient intestinal occlusion was due to an adhesion obstructing the alimentary tract; in the other two patients the occlusion was localized to the biliopancreatic tract, due to a serrate stenosis of the entero-entero anastomosis in one patient and due to volvulus of the biliopancreatic loop in the other patient. Results: Signs and symptoms were different according to whether the obstruction was in the alimentary tract or the biliopancreatic tract. In all cases a prompt gastrointestinal x-ray with barium and ultrasound scan and/or CT scan induced us to a mandatory laparotomy with resolution of the obstruction. Conclusions: After BPD, diagnosis of an intestinal obstruction must be made promptly. Even colleagues who express doubts must be persuaded to perform immediately an upper gastrointestinal tract x-ray and an U/S or CT scan. In this way, it may be possible to avoid intestinal resection and catastrophic complications.     

Parvovirus B19 microepidemic in renal transplant recipients with thrombotic microangiopathy and allograft vasculitis

Parvovirus B-19 (B-19) can lead to various clinical scenarios in renal transplant recipients. Here, we report a B-19 microepidemic that occurred between January and March 2007, involving renal transplant recipients from a single center in Tabriz, Iran. We observed 6 patients in whom there was a temporal association between active B-19 infection and thrombotic microangiopathy and intrarenal small and medium-sized vessel vasculitis. Patients typically presented with deteriorating renal allograft function and anemia, and laboratory findings revealed thrombotic microangiopathy. Ultimately, extensive endothelial injury and renal allograft vasculitis that mimicked a vascular rejection ensued. In conclusion, B-19-related thrombotic microangiopathy may precede allograft vasculitis in renal transplant recipients. A high index of suspicion is required for early diagnosis and treatment of B-19 infection. To the best of our knowledge, this series represents the first report of B-19-related renal allograft vasculitis in the English literature.     

Intra‐abdominal infections in solid organ transplant recipients: Guidelines from the American Society of Transplantation Infectious Diseases Community of Practice

This new guideline from the AST IDCOP reviews intra‐abdominal infections (IAIs), which cause substantial morbidity and mortality among abdominal SOT recipients. Each transplant type carries unique risks for IAI, though peritonitis occurs in all abdominal transplant recipients. Biliary infections, bilomas, and intra‐abdominal and intrahepatic abscesses are common after liver transplantation and are associated with the type of biliary anastomosis, the presence of vascular thrombosis or ischemia, and biliary leaks or strictures. IAIs after kidney transplantation include renal and perinephric abscesses and graft‐site candidiasis, which is uncommon but may require allograft nephrectomy. Among pancreas transplant recipients, duodenal anastomotic leaks can have catastrophic consequences, and polymicrobial abscesses can lead to graft loss and death. Intestinal transplant recipients are at the highest risk for sepsis, infection due to multidrug‐resistant organisms, and death from IAI, as the transplanted intestine is a contaminated, highly immunological, pathogen‐rich organ. Source control and antibiotics are the cornerstone of the management of IAIs. Empiric antimicrobial regimens should be tailored to local susceptibility patterns and pathogens with which the patient is known to be colonized, with subsequent optimization once the results of cultures are reported.     

Four cases of CMV ureteritis: emergence of a new pattern of disease?

Although cytomegalovirus (CMV) infection is common in renal transplant recipients, urinary tract involvement has been rare. Only six cases of symptomatic ureteritis have been reported in renal transplant recipients and all within the last five years. We describe an additional four cases of CMV ureteritis. The presentation of CMV ureteritis is obstructive nephropathy often in the absence of systemic illness. Presentation may also mimic allograft rejection with minimal obstructive symptoms. We hypothesize that CMV ureteritis is an emerging complication of CMV disease, possibly due to changes in transplant practice.     

The Long-Term Prognosis of Renal Transplant in Patients With Systemic Vasculitis

Little information is available about the long-term outcome of renal transplantation in patients with systemic vasculitis (SV). We compared the outcomes of 19 renal transplant recipients with SV with those of 38 controls matched for time of transplantation, age, gender and source of donor. The mean post-transplant follow-up was 58 +/- 57 months for vasculitic patients and 61 +/- 49 months for controls. The actuarial 10-year patient survival was 87% in vasculitic patients and 90% in controls, death-censored graft survival were 84% and 100%, respectively. The risks of acute and chronic rejection, and arterial hypertension were not significantly different between the two groups. Infection was significantly more frequent in vasculitic patients (74% vs. 34%; p = 0.01). Seven patients (36.8%) had a recurrence of vasculitis in mean 45 months after renal transplant (0.076/patients/year). After recurrence, one patient had an irreversible humoral rejection, another died from hemophagocytosis and another restarted dialysis 1 year later. Long-term patient and renal allograft survival in vasculitic patients was good. Although graft function recovered in most relapsers after reinforcement of immunosuppression, one patient died and two lost graft function.     

De novo gastrointestinal tumours after renal transplantation: role of CMV and EBV viruses

The development of new and more effective immunosuppressive agents has provided long-term survival for transplant recipients, thereby increasing the risk of de novo malignancy in chronic immunocompromised hosts. While de novo post-transplant lymphoproliferative diseases and skin cancer has been shown to have an increased incidence in long-term surviving solid organ transplant recipients, the association with gastrointestinal (GI) cancer is controversial. Over 12 yr, 20 patients (5%) out of 395 renal transplant recipients developed 23 de novo tumours; 11 skin cancer and 12 non-skin cancer. Four patients (1%) developed de novo tumours of the GI tract (three colon, and one gastric cancer). Immediately after tumour's diagnosis, immunosuppressive therapy was reduced; all patients were shifted from cyclosporine to Rapamicine within 30 d. The tumour was surgically resected with curative intent in three cases, while one patient had only palliative surgery because of metastatic disease. The post-operative courses was uneventful. All patients maintained normal graft function. However, three out of four patients (75%) died of progression of the neoplasm, within a median time from the diagnosis of 12 months. Further, we investigated a possible correlations between de novo GI cancer and HCV, HBV status, infections, cytomegalovirus (CMV) and Epstein-Barr virus (EBV) reactivation, episodes of rejection, and blood transfusions. All cases with GI de novo cancers reported in this paper developed CMV and EBV reactivation within three months after transplantation. Thereafter we suggest a closer follow-up for de novo GI cancer in renal transplants with early CMV and EBV reactivation in order to avoid delayed diagnosis.     

Erosive enterocolitis in mycophenolate mofetil-treated renal-transplant recipients with persistent afebrile diarrhea

BACKGROUND: Diarrhea is the most frequently reported adverse event in mycophenolate mofetil (MMF)-treated transplant patients. The aim of this study was to explore the gastrointestinal tract in MMF-treated renal transplant recipients with persistent afebrile diarrhea to characterize its nature and etiology. METHODS: Renal transplant recipients with persistent afebrile diarrhea (daily fecal output >200 g) were prospectively investigated for infections, morphologic, and functional (gastrointestinal motility and intestinal absorptive capacity) integrity of the gastrointestinal tract; 26 patients met the inclusion criteria. RESULTS: All but one patient had an erosive enterocolitis. Seventy percent of the patients had malabsorption of nutrients, contributing to the diarrhea. In +/-60%, an infectious origin was demonstrated and successfully treated with antimicrobial agents without changes in immunosuppressive regimen. In +/-40%, no infection occurred, but a Crohn's disease-like pattern of inflammation was noted. These patients also had a less pronounced bile-acid malabsorption but a significant faster colonic transit time, correlating with the trough level of mycophenolic acid (MPA). Cessation of MMF, however, was associated with allograft rejection in one third of these patients. CONCLUSIONS: Persistent afebrile diarrhea in renal transplant recipients is characterized by erosive enterocolitis, which is of infectious origin in +/-60%. In +/-40%, a Crohn's disease-like (entero-)colitis was present. Because reduction or cessation of MMF was the only effective therapy, MPA or one of its metabolites may be suggested as a possible cause. However, reduction or cessation of MMF was associated with an increased risk for rejection.     

Isolated celiac artery aneurysm with splenic artery stenosis as a rare presentation of polyarteritis nodosum: a case report and review of the literature

Polyarteritis nodosum (PAN) is a vasculitic disease that affects small to medium arteries. It often presents as a systemic and multiorgan disease that affects the skin, joints, gastrointestinal tract, kidneys, and nervous system. Nonspecific signs and symptoms are the hallmarks of PAN. It rarely affects single vessels, although there have been case reports of PAN involving single intra-abdominal vessels. The authors present a case of a 42-year-old white man who presented with mild epigastric pain but was otherwise asymptomatic. The workup ultimately revealed a rare case of an isolated celiac artery aneurysm with a concomitant stenosis of the splenic artery secondary to PAN. The patient was treated with high-dose steroid therapy, with resultant resolution of the vasculitis and a decreased size of the aneurysm.