Anisakiasis mimics cancer recurrence: Two cases of extragastrointestinal anisakiasis suspected to be recurrence of gynecological cancer on PET-CT and molecular biological investigation

Yuya Nogami, Yoko Nishimura, Kouji Banno, Atsushi Suzuki, Nobuyuki Susumu, Taizo Hibi, Koji Murakami, Taketo Yamada, Hiromu Sugiyama, Yasuyuki Morishima, Daisuke Aoki

Research output: Contribution to journalComment/debate

6 Citations (Scopus)

Abstract

Background: We report two cases of anisakiasis lesions that were initially suspected to be recurrence of gynecological cancer by positron emission tomography-computed tomography (PET-CT). Both cases were extragastrointestinal anisakiasis that is very rare. Case presentation: The first case was a patient with endometrial cancer. At 19 months after surgery, a new low density area of 2 cm in diameter in liver segment 4 was found on follow-up CT. In PET-CT, the lesion had abnormal 18fluoro-deoxyglucose (FDG) uptake with elevation in the delayed phase, with no other site showing FDG uptake. Partial liver resection was performed. A pathological examination revealed no evidence of malignancy, but showed necrotic granuloma with severe eosinophil infiltration and an irregular material with a lumen structure in the center. Parasitosis was suspected and consultation with the National Institute of Infectious Diseases (NIID) showed the larvae to be Anisakis simplex sensu stricto by genetic examination. The second case was a patient with low-grade endometrial stromal sarcoma (LG-ESS). At 8 months after surgery, swelling of the mediastinal lymph nodes was detected on CT and peripheral T-cell lymphoma was diagnosed by biopsy. A new peritoneal lesion with abnormal FDG uptake was detected on pre-treatment PET-CT and this lesion was increased in size on post-treatment PET-CT. Tumorectomy was performed based on suspected dissemination of LG-ESS recurrence. The findings in a pathological examination were similar to the first case and we again consulted the NIID. The larvae was identified as Anisakis pegreffi, which is a rare pathogen in humans. Having experienced these rare cases, we investigated the mechanisms of FDG uptake in parasitosis lesions by immunohistochemical staining using antibodies to glucose transporter type 1 (GLUT-1) and hexokinase type 2 (HK-2). While infiltrated eosinophils were negative, macrophages demonstrated positive for both antibodies. Therefore, mechanisms behind FDG uptake may involve macrophages, which is common among various granulomas. This is the first report to investigate parasitosis in such a way. Conclusion: These cases suggest that anisakiasis is a potential differential diagnosis for a lesion with FDG uptake in PET-CT, and that it is difficult to distinguish this disease from a recurrent tumor using PET-CT alone.

Original languageEnglish
Article number31
JournalBMC Medical Imaging
Volume16
Issue number1
DOIs
Publication statusPublished - 2016

Fingerprint

Anisakiasis
Recurrence
Endometrial Stromal Tumors
Anisakis
Neoplasms
Granuloma
Eosinophils
Communicable Diseases
Larva
Glucose Transporter Type 1
Macrophages
Peripheral T-Cell Lymphoma
Hexokinase
Antibodies
Liver
Deoxyglucose
Endometrial Neoplasms
Positron Emission Tomography Computed Tomography
Differential Diagnosis
Referral and Consultation

Keywords

  • Anisakis
  • Extragastrointestinal anisakiasis
  • Genetic examination
  • Glucose transporter type 1
  • Gynecological cancer
  • Hexokinase type 2
  • PCR
  • PET-CT

ASJC Scopus subject areas

  • Radiology Nuclear Medicine and imaging

Cite this

Anisakiasis mimics cancer recurrence : Two cases of extragastrointestinal anisakiasis suspected to be recurrence of gynecological cancer on PET-CT and molecular biological investigation. / Nogami, Yuya; Nishimura, Yoko; Banno, Kouji; Suzuki, Atsushi; Susumu, Nobuyuki; Hibi, Taizo; Murakami, Koji; Yamada, Taketo; Sugiyama, Hiromu; Morishima, Yasuyuki; Aoki, Daisuke.

In: BMC Medical Imaging, Vol. 16, No. 1, 31, 2016.

Research output: Contribution to journalComment/debate

Nogami, Yuya ; Nishimura, Yoko ; Banno, Kouji ; Suzuki, Atsushi ; Susumu, Nobuyuki ; Hibi, Taizo ; Murakami, Koji ; Yamada, Taketo ; Sugiyama, Hiromu ; Morishima, Yasuyuki ; Aoki, Daisuke. / Anisakiasis mimics cancer recurrence : Two cases of extragastrointestinal anisakiasis suspected to be recurrence of gynecological cancer on PET-CT and molecular biological investigation. In: BMC Medical Imaging. 2016 ; Vol. 16, No. 1.
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abstract = "Background: We report two cases of anisakiasis lesions that were initially suspected to be recurrence of gynecological cancer by positron emission tomography-computed tomography (PET-CT). Both cases were extragastrointestinal anisakiasis that is very rare. Case presentation: The first case was a patient with endometrial cancer. At 19 months after surgery, a new low density area of 2 cm in diameter in liver segment 4 was found on follow-up CT. In PET-CT, the lesion had abnormal 18fluoro-deoxyglucose (FDG) uptake with elevation in the delayed phase, with no other site showing FDG uptake. Partial liver resection was performed. A pathological examination revealed no evidence of malignancy, but showed necrotic granuloma with severe eosinophil infiltration and an irregular material with a lumen structure in the center. Parasitosis was suspected and consultation with the National Institute of Infectious Diseases (NIID) showed the larvae to be Anisakis simplex sensu stricto by genetic examination. The second case was a patient with low-grade endometrial stromal sarcoma (LG-ESS). At 8 months after surgery, swelling of the mediastinal lymph nodes was detected on CT and peripheral T-cell lymphoma was diagnosed by biopsy. A new peritoneal lesion with abnormal FDG uptake was detected on pre-treatment PET-CT and this lesion was increased in size on post-treatment PET-CT. Tumorectomy was performed based on suspected dissemination of LG-ESS recurrence. The findings in a pathological examination were similar to the first case and we again consulted the NIID. The larvae was identified as Anisakis pegreffi, which is a rare pathogen in humans. Having experienced these rare cases, we investigated the mechanisms of FDG uptake in parasitosis lesions by immunohistochemical staining using antibodies to glucose transporter type 1 (GLUT-1) and hexokinase type 2 (HK-2). While infiltrated eosinophils were negative, macrophages demonstrated positive for both antibodies. Therefore, mechanisms behind FDG uptake may involve macrophages, which is common among various granulomas. This is the first report to investigate parasitosis in such a way. Conclusion: These cases suggest that anisakiasis is a potential differential diagnosis for a lesion with FDG uptake in PET-CT, and that it is difficult to distinguish this disease from a recurrent tumor using PET-CT alone.",
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AU - Nogami, Yuya

AU - Nishimura, Yoko

AU - Banno, Kouji

AU - Suzuki, Atsushi

AU - Susumu, Nobuyuki

AU - Hibi, Taizo

AU - Murakami, Koji

AU - Yamada, Taketo

AU - Sugiyama, Hiromu

AU - Morishima, Yasuyuki

AU - Aoki, Daisuke

PY - 2016

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N2 - Background: We report two cases of anisakiasis lesions that were initially suspected to be recurrence of gynecological cancer by positron emission tomography-computed tomography (PET-CT). Both cases were extragastrointestinal anisakiasis that is very rare. Case presentation: The first case was a patient with endometrial cancer. At 19 months after surgery, a new low density area of 2 cm in diameter in liver segment 4 was found on follow-up CT. In PET-CT, the lesion had abnormal 18fluoro-deoxyglucose (FDG) uptake with elevation in the delayed phase, with no other site showing FDG uptake. Partial liver resection was performed. A pathological examination revealed no evidence of malignancy, but showed necrotic granuloma with severe eosinophil infiltration and an irregular material with a lumen structure in the center. Parasitosis was suspected and consultation with the National Institute of Infectious Diseases (NIID) showed the larvae to be Anisakis simplex sensu stricto by genetic examination. The second case was a patient with low-grade endometrial stromal sarcoma (LG-ESS). At 8 months after surgery, swelling of the mediastinal lymph nodes was detected on CT and peripheral T-cell lymphoma was diagnosed by biopsy. A new peritoneal lesion with abnormal FDG uptake was detected on pre-treatment PET-CT and this lesion was increased in size on post-treatment PET-CT. Tumorectomy was performed based on suspected dissemination of LG-ESS recurrence. The findings in a pathological examination were similar to the first case and we again consulted the NIID. The larvae was identified as Anisakis pegreffi, which is a rare pathogen in humans. Having experienced these rare cases, we investigated the mechanisms of FDG uptake in parasitosis lesions by immunohistochemical staining using antibodies to glucose transporter type 1 (GLUT-1) and hexokinase type 2 (HK-2). While infiltrated eosinophils were negative, macrophages demonstrated positive for both antibodies. Therefore, mechanisms behind FDG uptake may involve macrophages, which is common among various granulomas. This is the first report to investigate parasitosis in such a way. Conclusion: These cases suggest that anisakiasis is a potential differential diagnosis for a lesion with FDG uptake in PET-CT, and that it is difficult to distinguish this disease from a recurrent tumor using PET-CT alone.

AB - Background: We report two cases of anisakiasis lesions that were initially suspected to be recurrence of gynecological cancer by positron emission tomography-computed tomography (PET-CT). Both cases were extragastrointestinal anisakiasis that is very rare. Case presentation: The first case was a patient with endometrial cancer. At 19 months after surgery, a new low density area of 2 cm in diameter in liver segment 4 was found on follow-up CT. In PET-CT, the lesion had abnormal 18fluoro-deoxyglucose (FDG) uptake with elevation in the delayed phase, with no other site showing FDG uptake. Partial liver resection was performed. A pathological examination revealed no evidence of malignancy, but showed necrotic granuloma with severe eosinophil infiltration and an irregular material with a lumen structure in the center. Parasitosis was suspected and consultation with the National Institute of Infectious Diseases (NIID) showed the larvae to be Anisakis simplex sensu stricto by genetic examination. The second case was a patient with low-grade endometrial stromal sarcoma (LG-ESS). At 8 months after surgery, swelling of the mediastinal lymph nodes was detected on CT and peripheral T-cell lymphoma was diagnosed by biopsy. A new peritoneal lesion with abnormal FDG uptake was detected on pre-treatment PET-CT and this lesion was increased in size on post-treatment PET-CT. Tumorectomy was performed based on suspected dissemination of LG-ESS recurrence. The findings in a pathological examination were similar to the first case and we again consulted the NIID. The larvae was identified as Anisakis pegreffi, which is a rare pathogen in humans. Having experienced these rare cases, we investigated the mechanisms of FDG uptake in parasitosis lesions by immunohistochemical staining using antibodies to glucose transporter type 1 (GLUT-1) and hexokinase type 2 (HK-2). While infiltrated eosinophils were negative, macrophages demonstrated positive for both antibodies. Therefore, mechanisms behind FDG uptake may involve macrophages, which is common among various granulomas. This is the first report to investigate parasitosis in such a way. Conclusion: These cases suggest that anisakiasis is a potential differential diagnosis for a lesion with FDG uptake in PET-CT, and that it is difficult to distinguish this disease from a recurrent tumor using PET-CT alone.

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KW - Extragastrointestinal anisakiasis

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KW - Glucose transporter type 1

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