Small intestinal perforation due to a huge gastrointestinal stromal tumor in a kidney transplant recipient: a case report and literature review.


Journal

BMC nephrology
ISSN: 1471-2369
Titre abrégé: BMC Nephrol
Pays: England
ID NLM: 100967793

Informations de publication

Date de publication:
03 04 2019
Historique:
received: 11 01 2019
accepted: 21 03 2019
entrez: 5 4 2019
pubmed: 5 4 2019
medline: 10 5 2020
Statut: epublish

Résumé

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. 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 Cyclosporine was withdrawn and imatinib mesylate (400 mg daily) was introduced.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. 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under immunosuppression, although immunosuppressive drugs metabolized by CYP3A4 should be used at a reduced dosage or withdrawn.

Sections du résumé

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.

Identifiants

pubmed: 30943904
doi: 10.1186/s12882-019-1310-5
pii: 10.1186/s12882-019-1310-5
pmc: PMC6448240
doi:

Substances chimiques

Antineoplastic Agents 0
Imatinib Mesylate 8A1O1M485B
KIT protein, human EC 2.7.10.1
Proto-Oncogene Proteins c-kit EC 2.7.10.1

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

120

Références

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Auteurs

Ryohei Takahashi (R)

Department of Urology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Kazunobu Shinoda (K)

Department of Urology, Keio University School of Medicine, Tokyo, 160-8582, Japan. kshino49@yahoo.co.jp.
Department of Nephrology, Toho University Faculty of Medicine, 7-5-23 Omorinishi Ota-ku, Tokyo, 143-0015, Japan. kshino49@yahoo.co.jp.

Takashi Ishida (T)

Department of Surgery, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Yasuo Hamamoto (Y)

Keio Cancer Center, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Shinya Morita (S)

Department of Urology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Hirotaka Akita (H)

Department of Diagnostic Radiology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Sotaro Kitaoka (S)

Department of Urology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Satoshi Tamaki (S)

Department of Urology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Hiroshi Asanuma (H)

Department of Urology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Tadashi Yoshida (T)

Apheresis and Dialysis Center, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Masahiro Jinzaki (M)

Department of Diagnostic Radiology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Kaori Kameyama (K)

Department of Diagnostic Pathology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

Mototsugu Oya (M)

Department of Urology, Keio University School of Medicine, Tokyo, 160-8582, Japan.

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