Small intestinal perforation due to a huge gastrointestinal stromal tumor in a kidney transplant recipient

A case report and literature review

Research output: Contribution to journalReview article

Abstract

Background: Gastrointestinal stromal tumors (GISTs) in transplant recipients are very rare and only a handful of cases have been reported to date. Here we present the first known case of a huge GIST in a kidney transplant recipient with perforation of small intestine. Case presentation: A 64-year-old male presented at our hospital with right colic pain; he had received an ABO incompatible kidney transplant 6 years earlier and was treated with cyclosporine, mycophenolate mofetil, and methylprednisolone. Radiological evaluation revealed a huge (11 cm in diameter) solitary tumor at the small intestine without distant metastasis. The small intestinal wall at the tumor location was perforated one week after diagnosis and the patient underwent emergency surgery. The pathological findings were compatible with GIST and the tumor consisted of spindle cells with positive staining for KIT, CD34, and DOG1 and negative or weak staining for desmin and S-100 protein. A mutation in exon 11 of the c-kit gene was also detected. Cyclosporine was withdrawn and imatinib mesylate (400 mg daily) was introduced. However, thereafter, we needed to decrease the dose at 300 mg daily due to severe hyponatremia. Reduced imatinib treatment was well tolerated and recurrence was not observed for 18 months after surgery. Conclusions: The occurrence of GISTs in transplant patients is rare, and huge GISTs should be resected immediately after diagnosis because gastrointestinal tract at the tumor site could be perforated. Imatinib treatment is feasible in transplant recipients under immunosuppression, although immunosuppressive drugs metabolized by CYP3A4 should be used at a reduced dosage or withdrawn.

Original languageEnglish
Article number120
JournalBMC Nephrology
Volume20
Issue number1
DOIs
Publication statusPublished - 2019 Apr 3

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Intestinal Perforation
Gastrointestinal Stromal Tumors
Kidney
Cyclosporine
Small Intestine
Neoplasms
Staining and Labeling
Mycophenolic Acid
Transplants
Cytochrome P-450 CYP3A
Desmin
Colic
S100 Proteins
Hyponatremia
Methylprednisolone
Immunosuppressive Agents
Immunosuppression
Gastrointestinal Tract
Exons
Emergencies

Keywords

  • And imatinib mesylate
  • Gastrointestinal stromal tumor
  • Kidney transplant recipient
  • Spontaneous rupture

ASJC Scopus subject areas

  • Nephrology

Cite this

@article{072ed96d5f454cdd909e8d6dd233973f,
title = "Small intestinal perforation due to a huge gastrointestinal stromal tumor in a kidney transplant recipient: A case report and literature review",
abstract = "Background: Gastrointestinal stromal tumors (GISTs) in transplant recipients are very rare and only a handful of cases have been reported to date. Here we present the first known case of a huge GIST in a kidney transplant recipient with perforation of small intestine. Case presentation: A 64-year-old male presented at our hospital with right colic pain; he had received an ABO incompatible kidney transplant 6 years earlier and was treated with cyclosporine, mycophenolate mofetil, and methylprednisolone. Radiological evaluation revealed a huge (11 cm in diameter) solitary tumor at the small intestine without distant metastasis. The small intestinal wall at the tumor location was perforated one week after diagnosis and the patient underwent emergency surgery. The pathological findings were compatible with GIST and the tumor consisted of spindle cells with positive staining for KIT, CD34, and DOG1 and negative or weak staining for desmin and S-100 protein. A mutation in exon 11 of the c-kit gene was also detected. Cyclosporine was withdrawn and imatinib mesylate (400 mg daily) was introduced. However, thereafter, we needed to decrease the dose at 300 mg daily due to severe hyponatremia. Reduced imatinib treatment was well tolerated and recurrence was not observed for 18 months after surgery. Conclusions: The occurrence of GISTs in transplant patients is rare, and huge GISTs should be resected immediately after diagnosis because gastrointestinal tract at the tumor site could be perforated. Imatinib treatment is feasible in transplant recipients under immunosuppression, although immunosuppressive drugs metabolized by CYP3A4 should be used at a reduced dosage or withdrawn.",
keywords = "And imatinib mesylate, Gastrointestinal stromal tumor, Kidney transplant recipient, Spontaneous rupture",
author = "Ryohei Takahashi and Kazunobu Shinoda and Takashi Ishida and Yasuo Hamamoto and Shinya Morita and Hirotaka Akita and Sotaro Kitaoka and Satoshi Tamaki and Hiroshi Asanuma and Tadashi Yoshida and Masahiro Jinzaki and Kaori Kameyama and Mototsugu Oya",
year = "2019",
month = "4",
day = "3",
doi = "10.1186/s12882-019-1310-5",
language = "English",
volume = "20",
journal = "BMC Nephrology",
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TY - JOUR

T1 - Small intestinal perforation due to a huge gastrointestinal stromal tumor in a kidney transplant recipient

T2 - A case report and literature review

AU - Takahashi, Ryohei

AU - Shinoda, Kazunobu

AU - Ishida, Takashi

AU - Hamamoto, Yasuo

AU - Morita, Shinya

AU - Akita, Hirotaka

AU - Kitaoka, Sotaro

AU - Tamaki, Satoshi

AU - Asanuma, Hiroshi

AU - Yoshida, Tadashi

AU - Jinzaki, Masahiro

AU - Kameyama, Kaori

AU - Oya, Mototsugu

PY - 2019/4/3

Y1 - 2019/4/3

N2 - Background: Gastrointestinal stromal tumors (GISTs) in transplant recipients are very rare and only a handful of cases have been reported to date. Here we present the first known case of a huge GIST in a kidney transplant recipient with perforation of small intestine. Case presentation: A 64-year-old male presented at our hospital with right colic pain; he had received an ABO incompatible kidney transplant 6 years earlier and was treated with cyclosporine, mycophenolate mofetil, and methylprednisolone. Radiological evaluation revealed a huge (11 cm in diameter) solitary tumor at the small intestine without distant metastasis. The small intestinal wall at the tumor location was perforated one week after diagnosis and the patient underwent emergency surgery. The pathological findings were compatible with GIST and the tumor consisted of spindle cells with positive staining for KIT, CD34, and DOG1 and negative or weak staining for desmin and S-100 protein. A mutation in exon 11 of the c-kit gene was also detected. Cyclosporine was withdrawn and imatinib mesylate (400 mg daily) was introduced. However, thereafter, we needed to decrease the dose at 300 mg daily due to severe hyponatremia. Reduced imatinib treatment was well tolerated and recurrence was not observed for 18 months after surgery. Conclusions: The occurrence of GISTs in transplant patients is rare, and huge GISTs should be resected immediately after diagnosis because gastrointestinal tract at the tumor site could be perforated. Imatinib treatment is feasible in transplant recipients under immunosuppression, although immunosuppressive drugs metabolized by CYP3A4 should be used at a reduced dosage or withdrawn.

AB - Background: Gastrointestinal stromal tumors (GISTs) in transplant recipients are very rare and only a handful of cases have been reported to date. Here we present the first known case of a huge GIST in a kidney transplant recipient with perforation of small intestine. Case presentation: A 64-year-old male presented at our hospital with right colic pain; he had received an ABO incompatible kidney transplant 6 years earlier and was treated with cyclosporine, mycophenolate mofetil, and methylprednisolone. Radiological evaluation revealed a huge (11 cm in diameter) solitary tumor at the small intestine without distant metastasis. The small intestinal wall at the tumor location was perforated one week after diagnosis and the patient underwent emergency surgery. The pathological findings were compatible with GIST and the tumor consisted of spindle cells with positive staining for KIT, CD34, and DOG1 and negative or weak staining for desmin and S-100 protein. A mutation in exon 11 of the c-kit gene was also detected. Cyclosporine was withdrawn and imatinib mesylate (400 mg daily) was introduced. However, thereafter, we needed to decrease the dose at 300 mg daily due to severe hyponatremia. Reduced imatinib treatment was well tolerated and recurrence was not observed for 18 months after surgery. Conclusions: The occurrence of GISTs in transplant patients is rare, and huge GISTs should be resected immediately after diagnosis because gastrointestinal tract at the tumor site could be perforated. Imatinib treatment is feasible in transplant recipients under immunosuppression, although immunosuppressive drugs metabolized by CYP3A4 should be used at a reduced dosage or withdrawn.

KW - And imatinib mesylate

KW - Gastrointestinal stromal tumor

KW - Kidney transplant recipient

KW - Spontaneous rupture

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U2 - 10.1186/s12882-019-1310-5

DO - 10.1186/s12882-019-1310-5

M3 - Review article

VL - 20

JO - BMC Nephrology

JF - BMC Nephrology

SN - 1471-2369

IS - 1

M1 - 120

ER -