Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Aug 6, 2019; 7(15): 2049-2057
Published online Aug 6, 2019. doi: 10.12998/wjcc.v7.i15.2049
ALK-positive anaplastic large cell lymphoma presenting multiple lymphomatous polyposis: A case report and literature review
Makoto Saito, Koh Izumiyama, Reiki Ogasawara, Akio Mori, Takeshi Kondo, Masanori Tanaka, Masanobu Morioka, Kencho Miyashita, Mishie Tanino
Makoto Saito, Koh Izumiyama, Reiki Ogasawara, Akio Mori, Takeshi Kondo, Masanori Tanaka, Masanobu Morioka, Department of Internal Medicine and Hematology, Aiiku Hospital, Sapporo 0640804, Japan
Kencho Miyashita, Department of Gastroenterology, Aiiku Hospital, Sapporo 0640804, Japan
Mishie Tanino, Department of Surgical Pathology, Asahikawa Medical University Hospital (formerly Department of Cancer Pathology, Hokkaido University, Faculty of Medicine), Asahikawa 0788510, Japan
Author contributions: All authors collected and analyzed the patient’s clinical data; Miyashita K involved in endoscopic procedure; Tanino M involved in pathological procedure; Saito M designed and wrote the manuscript; and all authors agree to be accountable for all aspects of the work.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest in this work.
CARE Checklist (2016) statement: The manuscript was prepared according to the CARE Checklist (2016).
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Makoto Saito, MD, PhD, Doctor, Department of Internal Medicine and Hematology, Aiiku Hospital, Minami 4 Nishi 25 Chuo-ku, Sapporo 0640804, Japan. ikyoku@aiiku-hp.or.jp
Telephone: +8-111-5632211
Received: February 18, 2019
Peer-review started: February 20, 2019
First decision: May 31, 2019
Revised: June 11, 2019
Accepted: June 20, 2019
Article in press: June 21, 2019
Published online: August 6, 2019
Abstract
BACKGROUND

Anaplastic large cell lymphoma (ALCL) is a type of T-cell lymphoma that can be divided into two categories: anaplastic lymphoma kinase-positive (ALK+) and ALK-negative. Gastrointestinal ALK+ ALCL is rare. Multiple lymphomatous polyposis (MLP) is thought to be a representative form of gastrointestinal lesion in mantle cell lymphoma, and T-cell lymphomas seldom show this feature. Here, we report the first known case of ALK+ ALCL with gastroduodenal involvement to present with MLP.

CASE SUMMARY

The patient was a 43-year-old man who was complained of a mass in the left inguinal area and was performed open biopsy. ALK+ ALCL was diagnosed pathologically. Computed tomography scan demonstrated multiple lymph node lesions in the abdomen - pelvis/inguinal region, and scattered nodular lesions in both lung fields. He did not complain of gastrointestinal symptoms. While, esophagogastroduodenoscopy identified MLP lesions from the antrum of the stomach to the descending portion of the duodenum and mild thickened folds on the corpus of the stomach, and biopsy showed invasion of ALK+ ALCL. We treated this patient with six cycles of CHOEP (Cyclophosphamide, Doxorubicin, Vincristine, Etoposide, and Prednisone) chemotherapy. At the conclusion of treatment, there was complete remission. Numerous white scars were found on the stomach, endoscopically consistent with a remission image of lymphoma. The endoscopic features of this case were thought to be similar to those of MCL.

CONCLUSION

The macroscopic/endoscopic features of gastrointestinal ALK+ ALCL may be more similar to those of B-cell lymphomas rather than T-cell lymphomas.

Keywords: Anaplastic large-cell lymphoma, Anaplastic lymphoma kinase, Multiple lymphomatous polyposis, T-cell lymphoma, Gastrointestinal involvement

Core tip: Anaplastic large cell lymphoma (ALCL) encompasses two distinct categories: anaplastic lymphoma kinase-positive (ALK+) and ALK-negative. ALK+ ALCL cases rarely involve the gut. However, in a very small number of case reports, gastrointestinal ALK+ ALCL exhibits a fungating growth pattern, similar to that of B-cell lymphomas rather than T-cell lymphomas. Multiple lymphomatous polyposis (MLP) is thought to be a typical form of gastrointestinal lesion in mantle cell lymphoma, but it develops in other B-cell lymphomas. T-cell lymphomas seldom present with MLP. Here, we report the first known case in the world of ALK+ ALCL with gastroduodenal involvement presenting with MLP.