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Progressive multifocal leukoencephalopathy presenting with acute sensorineural hearing loss in an intestinal transplant recipient.
Lippa, AM, Ocwieja, KE, Iglesias, J, Fawaz, R, Elisofon, S, Lee, C, Sharma, TS
Transplant infectious disease : an official journal of the Transplantation Society. 2020;(4):e13304
Abstract
A 20-year-old male presented 3.5 years after intestinal transplantation with rapidly progressive sensorineural hearing loss. Initial brain imaging was consistent with inflammation and/or demyelination. Lumbar puncture was initially non-diagnostic and a broad infectious workup was unrevealing. Three months after presentation, a repeat LP detected JC virus for which tests had not earlier been conducted. He continued to deteriorate despite withdrawal of prior immunosuppression and addition of mirtazapine, maraviroc, and steroids. He died of progressive neurologic decompensation 5 months after his initial presentation. This case highlights progressive multifocal leukoencephalopathy (PML) as a rare complication after solid organ transplantation and acute sensorineural hearing loss as an unusual first presenting symptom of PML. JC virus should be considered in the differential diagnosis of acute sensorineural hearing loss in any immunocompromised patient.
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2.
Intestinal Lymphangiectasia: Insights on Management and Literature Review.
Alshikho, MJ, Talas, JM, Noureldine, SI, Zazou, S, Addas, A, Kurabi, H, Nasser, M
The American journal of case reports. 2016;:512-22
Abstract
BACKGROUND Intestinal lymphangiectasia (IL) is a rare disease characterized by a dilatation of the intestinal lymphatics and loss of lymph fluid into the gastrointestinal tract leading to hypoproteinemia, edema, lymphocytopenia, hypogammaglobinemia, and immunological abnormalities. Iron, calcium, and other serum components (e.g., lipids, fat soluble vitamins) may also be depleted. A literature search revealed more than 200 reported cases of IL. Herein, we report our observations of a patient diagnosed with IL; we also present our conclusion for our review of the published literature. CASE REPORT A 24-year-old male was admitted to Aleppo University Hospital with the complaints of abdominal pain, headache, arthralgia, fever, and rigors. His past medical history was remarkable for frequent episodes of diarrhea, recurrent infections, and swelling in the lower limbs. In addition, he had been hospitalized several times in non-academic hospitals due to edema in his legs, cellulitis, and recurrent infections. In the emergency department, a physical examination revealed a patient in distress. He was weak, dehydrated, pale, and had a high-grade fever. His lower extremities were edematous, swollen, and extremely tender to touch. The overlying skin was erythematous and warm. Moreover, the patient was tachycardic, tacypneic, and moderately hypotensive. The patient was resuscitated with IV fluids, and Tylenol was administered to bring the temperature down. Blood tests showed anemia and high levels of inflammatory markers. The patient's white blood cell count was elevated with an obvious left shift. However, subsequent investigations showed that the patient had IL. Suitable diet modification plans were applied as a long-term management plan. CONCLUSIONS IL is a rare disease of challenging nature due to its systematic effects and lack of comprehensive studies that can evaluate the effectiveness of specific treatments in a large cohort of patients. MCT (medium-chain triglyceride) oils and diet modification strategies are effective in reducing the loss of body proteins and in maintaining near-normal blood levels of immunoglobulins. However, octreotide and MCT oils had no proven role in shrinking edema in our patient.
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3.
Intestinal transplantation: from the laboratory to the clinics.
Pirenne, J, Kawai, M, Kitade, H, Koshiba, T, Takahashi, K, Aerts, R, Monbaliu, D, Coosemans, W, Waer, M
Acta chirurgica Belgica. 2008;(1):52-7
Abstract
The intestine has long been seen as a "forbidden" organ to transplant. This is because the first attempts at Intestinal Transplantation (ITx) were defeated by rejection, technical problems, infection and graft versus host disease. Results of ITx have improved in the short-term (70 to 80% 1-year patient survival) but remain inferior to other solid organ transplants in the long-term (5 years patient survival of 50% or less). Reasons for this difference between intestine and other organ transplants are reviewed. Development of immunomodulatory protocols--e.g. protocols aiming at reducing the rejection response and facilitating engraftment--are described. Our center experience with a consecutive series of five intestinal transplants utilizing a new protolerogenic protocol and low immunosuppression is described. At time of writing, these five patients are rejection-free, nutritionally independent and lead a normal life.
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4.
[Intestinal transplantation in adults: present situation and future prospects].
Gajate Martín, L, Elías Martín, E, Martínez Pérez, A, Carrasco Seral, C, Alvarez Utrera, F, Pablo de Pajares, A, Pinto Corraliza, J, Honrubia Checa, A, Vicente de López, E, Quijano-Collazo, Y, et al
Revista espanola de anestesiologia y reanimacion. 2004;(9):537-48
Abstract
Intestinal transplantation is becoming more firmly established as a treatment for intestinal failure in patients whose home parenteral nutrition regimens have caused serious side effects. Outcomes have improved spectacularly over recent years thanks to the refinement of surgical techniques and the introduction of new immunosuppressants, and also to greater experience in anesthetic and postoperative management of intestinal transplant patients. The main causes of high morbidity and mortality continue to be sepsis and acute rejection of the graft. Both graft and patient survival have improved with the advent of the immunosuppressant regimens based on Tacrolimus, although survival rates are still far below those reported for other solid organ transplants. The first intestinal transplant performed in Spain took place in July 2002 in our hospital and the results were promising. Given this new challenge for anesthesiologists, we decided to review current trends in the perioperative management of patients receiving isolated intestinal transplants, the main complications that arise, treatment strategies, and future prospects.
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5.
Nutritional complications and management of intestinal transplant.
Silver, HJ, Castellanos, VH
Journal of the American Dietetic Association. 2000;(6):680-4, 687-9; quiz 685-6
Abstract
Advances in intestinal transplantation provide a promising alternative to patients with intestinal failure and chronic dependence on total parenteral nutrition. However, many physiologic complications arising from the surgical procedure and high-dose immunosuppression, along with potential for rejection and infection, make successful graft function after transplantation a challenge. Nutrition issues unique to this patient population include recovery of normal intestinal motility and absorptive capacity. Diarrhea and high stomal output, which are common postoperatively, lead to deficits in macronutrients and micronutrients, especially electrolytes. Impaired gastrointestinal function affects ability to wean patients off hyperalimentation and enable them to tolerate nutrients enterally. In pediatric recipients of intestinal transplant, lack of experience with food or prior food aversions can lead to refusal to eat after transplant--additional challenges to achieving oral intake. Early and aggressive nutrition intervention is necessary for resolution of nutritional deficits and health of donor small bowel. This article presents an overview of the surgical procedure of intestinal transplantation and describes the physiologic adaptations that occur after the process. A case study demonstrates the clinical and nutritional hurdles associated with an intestinal transplant in a child and how dietitians can provide nutrition management. The potential role of individual nutrients in recovery of the transplanted bowel is also discussed.