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      Posttransplant Lymphoproliferative Disorder involving the Larynx

      case-report

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          ABSTRACT

          Aim:

          Our goal is to present an unusual case of posttransplant lymphoproliferative disorder (PTLD) involving the larynx. We aim to expand the literature regarding head and neck manifestations involving PTLD and for otolaryngologists to include PTLD in their differential diagnosis of laryngeal lesions in patients who have a history of solid organ transplantation.

          Background

          Posttransplant lymphoproliferative disorder occurs in immunocompromised patients following solid organ transplantation. Head and neck manifestations most commonly involve Waldeyer's ring with the larynx and trachea being relatively uncommon sites of disease. However, lesions of the larynx can cause acute airway obstruction and rare fatalities have been reported in the literature.

          Case report

          We present the case of a 51-year-old female with a history of renal transplantation, who presented to the office after an incidental supraglottic lesion was discovered during intubation for cochlear implant placement. A mucosalized lesion arising from the right aryepiglottic fold with dynamic obstruction of the airway was noted on office endoscopic examination. Imaging revealed extension of the mass into the right posterolateral hypopharynx. The patient was taken to the operating room for microdirect laryngoscopy and CO 2 laser excision of the mass. The lesion was excised with negative gross margins. Final pathology revealed plasmacytoma-like PTLD and the patient was referred back to her transplant team for modulation of her immunosuppressive therapy.

          Conclusion

          We present the case of an incidental supraglottic lesion in a posttransplant patient. Special emphasis should be given to the tissues of Waldeyer's ring and larynx.

          Clinical significance

          Posttransplant lymphoproliferative disorder, while rare, should be kept in the differential diagnosis of pharyngeal and airway lesions in patients with a history of solid organ transplantation. Expansile lesions can cause acute airway obstruction. Urgent evaluation and treatment of theses lesions is necessary in these circumstances.

          How to cite this article

          Kohli N, Wasserman JM. Posttransplant Lymphoproliferative Disorder involving the Larynx. Int J Head Neck Surg 2016;7(1):53-56.

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          Most cited references12

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          Posttransplant lymphoproliferative disorders in liver transplantation: a 20-year experience.

          To evaluate the incidence of posttransplant lymphoproliferative disease (PTLD) and the risk factors and the impact of this complication on survival outcomes in a large cohort of liver transplant recipients at a single institution. Liver transplantation has been accepted as a therapeutic option for patients with end-stage liver disease since 1983, in large part due to the availability and reliance on the use of nonspecifically directed immunosuppression. However, as predicted and subsequently verified in 1968, an increased incidence of certain de novo malignancies has been observed, particularly with regards to lymphoid neoplasms. While many reports have confirmed and clarified the nature of PTLD, the literature is fraught with conflicting experience and outcomes with PTLD. Four thousand consecutive patients who underwent liver transplants between February 1981 and April 1998 were included in this analysis and were followed to November 2001. The effect of recipient age at the time of transplant, recipient gender, diagnosis, baseline immunosuppression, grading of PTLD, and association with Epstein-Barr virus were compared. The causes of death were also examined. Treatment for PTLD varied over the 20-year period, but all included massive reduction or elimination of baseline immunosuppression. The 1-year patient survival for liver transplant patients with PTLD was 85%, while the overall patient survival for the entire cohort was 53%. The actuarial 20-year survival was estimated at 45%. The overall median time to PTLD presentation was 10 months, and children had an incidence of PTLD that was threefold higher than adults. Patient survival was better in children, in patients transplanted in the era of tacrolimus immunosuppression, in patients with polymorphic PTLD, and in those with limited disease. Interestingly, neither the presence or absence of Epstein-Barr virus nor the timing of PTLD presentation appeared to influence overall patient survival. Patients transplanted for alcohol-related liver disease had a similar incidence of PTLD but had a higher risk of mortality. While PTLD continues to pose problems in patients receiving liver transplants, improvements in patient survival have been observed over time. While it is too early to assess the impact of new advances in prophylaxis, diagnosis, and treatment, such approaches are based on an increased knowledge of the pathophysiology of PTLD.
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            Posttransplant lymphoproliferative disorders: summary of Society for Hematopathology Workshop.

            Twenty cases of posttransplant lymphoproliferative disorders arising in solid organ allograft recipients (18 patients) or bone marrow allograft recipients (2 patients: 1 autologous; 1 allogeneic) were selected for presentation at the Society for Hematopathology Workshop. In the course of the Workshop discussions, based both on the submitted cases and the combined experience of the participants, it was possible to agree on several distinctive categories of PTLD. These include (1) early lesions, (2) polymorphic posttransplant lymphoproliferative disorders (PTLDs), (3) monomorphic PTLDs (B- and T-cell lymphomas), (4) plasmacytoma-like lesions, and (5) T-cell-rich large B-cell lymphoma/Hodgkin's disease-like lesions. Monomorphic lesions should be classified according to a recognized classification of non-Hodgkin's lymphoma, although specified in the report as PTLD. Polymorphic lesions should be carefully evaluated for clonality; by immunophenotyping; and, if necessary, analysis of antigen-receptor and Epstein-Barr virus (EBV) genomes. Minimal pathological evaluation should include routine morphology, immunophenotyping on fresh tissue (flow cytometry or frozen section), and preservation of tissue for molecular genetic analysis. Analysis of the presence of EBV can be useful in establishing whether early or equivocal lesions represent PTLD (EBV+) or unrelated processes, but is not required in most cases. The pathologist can make an important contribution to the management of patients with PTLD by providing a complete diagnostic evaluation of the biopsy specimens (this is the least expensive part of the care of a transplant patients, not a place to try to cut costs) and making sure the attending physicians understand the special issues in management of PTLD.
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              • Record: found
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              • Article: not found

              Five years' experience in renal transplantation with immunosuppressive drugs: survival, function, complications, and the role of lymphocyte depletion by thoracic duct fistula.

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                Author and article information

                Contributors
                Role: Resident
                Role: Private Practitioner
                Journal
                IJHNS
                International Journal of Head and Neck Surgery
                IJHNS
                Jaypee Brothers Medical Publishers
                0975-7899
                0976-0539
                January-March 2016
                : 7
                : 1
                : 53-56
                Affiliations
                [1 ] SUNY Downstate Medical Center, Brooklyn, New York, USA
                [2 ] The Voice and Swallowing Center ENT and Allergy Associates, Hackensack, New Jersey, USA
                Author notes
                Nikita Kohli, Resident, SUNY Downstate Medical Center, Brooklyn, New York, USA Phone: 718-270-1638, e-mail: nikitavkohli@ 123456gmail.com
                Article
                10.5005/jp-journals-10001-1265
                b499573e-7174-4153-9733-adba1dc14242
                Copyright © 2016; Jaypee Brothers Medical Publishers (P) Ltd.

                Creative Commons Attribution 4.0

                History
                Categories
                CASE REPORT
                Custom metadata
                ijhns-2016-7-53.pdf

                General medicine,Pathology,Surgery,Sports medicine,Anatomy & Physiology,Orthopedics
                Immunocompromised,Adult,Laryngology,Airway obstruction,Posttransplant lymphoproliferative disorder

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