Chronic active EBV infection in refractory enteritis with longitudinal ulcers with a cobblestone appearance: an autopsied case report Article Swipe
Yosuke Aihara
,
Kei Moriya
,
Naotaka Shimozato
,
Shinsaku Nagamatsu
,
Shinya Kobayashi
,
Masakazu Uejima
,
Hideki Matsuo
,
Eiwa Ishida
,
Hideo Yagi
,
Toshiya Nakatani
,
Hitoshi Yoshiji
,
Eiryo Kikuchi
·
YOU?
·
· 2021
· Open Access
·
· DOI: https://doi.org/10.1186/s12876-020-01589-1
YOU?
·
· 2021
· Open Access
·
· DOI: https://doi.org/10.1186/s12876-020-01589-1
Background Chronic active Epstein–Barr virus infection (CAEBV) is defined as Epstein–Barr virus (EBV)-positive T/NK cell-related neoplasia, and its major clinical symptom is systemic inflammation presenting as infectious mononucleocytosis, whereas enteritis and diarrhea are minor clinical symptoms. The complex mixture of tumorigenic processes of EBV-positive cells and physical symptoms of systemic inflammatory disease constitutes the varied phenotypes of CAEBV. Herein, we describe a case of CAEBV that was initially diagnosed as Crohn’s disease (CD) based on ileal ulcers and clinical symptoms of enteritis. Case presentation A 19-year-old woman complained of abdominal pain and fever. Blood examination showed normal blood cell counts without atypical lymphocyte but detected modest inflammation, hypoalbuminemia, slight liver dysfunction, and evidence of past EBV infection. The esophagogastroduodenoscopic findings were normal. However, colonoscopy revealed a few small ulcers in the terminal ileum. The jejunum and ileum also exhibited various forms of ulcers, exhibiting a cobblestone appearance, on capsule endoscopy. Based on these clinical findings, she was strongly suspected with CD. In the course of treatment by steroid and biologics for refractory enteritis, skin ulcers appeared about 50 months after her initial hospital visit. Immunohistology of her skin biopsy revealed proliferation of EBV-encoded small RNA (EBER)-positive atypical lymphocytes. We retrospectively assessed her previous ileal ulcer biopsy before treatment and found many EBER-positive lymphocytes. Blood EBV DNA was also positive. Therefore, she was diagnosed with extranodal NK/T-cell lymphoma with CAEBV-related enteritis rather than CD. She was treated with cyclosporine and prednisolone combination therapy for CAEBV-related systemic inflammation and chemotherapy for malignant lymphoma. Unfortunately, her disease continued to progress, leading to multiple organ failure and death at the age of 23 years. Conclusion Clinicians need to remember the possibility of CAEBV as a differential diagnosis of refractory enteritis. Enteritis with intestinal ulcer is a rare symptom of CAEBV, and it is impossible to acquire a definitive diagnosis by ulcer morphology only. In cases where the possibility of CAEBV remains, tissue EBVR expression should be checked by in situ hybridization and blood EBV DNA.
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Metadata
- Type
- article
- Language
- en
- Landing Page
- https://doi.org/10.1186/s12876-020-01589-1
- https://bmcgastroenterol.biomedcentral.com/track/pdf/10.1186/s12876-020-01589-1
- OA Status
- gold
- Cited By
- 10
- References
- 17
- Related Works
- 10
- OpenAlex ID
- https://openalex.org/W3118714660
All OpenAlex metadata
Raw OpenAlex JSON
- OpenAlex ID
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https://openalex.org/W3118714660Canonical identifier for this work in OpenAlex
- DOI
-
https://doi.org/10.1186/s12876-020-01589-1Digital Object Identifier
- Title
-
Chronic active EBV infection in refractory enteritis with longitudinal ulcers with a cobblestone appearance: an autopsied case reportWork title
- Type
-
articleOpenAlex work type
- Language
-
enPrimary language
- Publication year
-
2021Year of publication
- Publication date
-
2021-01-06Full publication date if available
- Authors
-
Yosuke Aihara, Kei Moriya, Naotaka Shimozato, Shinsaku Nagamatsu, Shinya Kobayashi, Masakazu Uejima, Hideki Matsuo, Eiwa Ishida, Hideo Yagi, Toshiya Nakatani, Hitoshi Yoshiji, Eiryo KikuchiList of authors in order
- Landing page
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https://doi.org/10.1186/s12876-020-01589-1Publisher landing page
- PDF URL
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https://bmcgastroenterol.biomedcentral.com/track/pdf/10.1186/s12876-020-01589-1Direct link to full text PDF
- Open access
-
YesWhether a free full text is available
- OA status
-
goldOpen access status per OpenAlex
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https://bmcgastroenterol.biomedcentral.com/track/pdf/10.1186/s12876-020-01589-1Direct OA link when available
- Concepts
-
Medicine, Internal medicine, Gastroenterology, Enteritis, Biopsy, Ileitis, Lymphoma, Pathology, Immunology, Crohn's disease, DiseaseTop concepts (fields/topics) attached by OpenAlex
- Cited by
-
10Total citation count in OpenAlex
- Citations by year (recent)
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2025: 4, 2024: 2, 2022: 2, 2021: 2Per-year citation counts (last 5 years)
- References (count)
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17Number of works referenced by this work
- Related works (count)
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10Other works algorithmically related by OpenAlex
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| abstract_inverted_index.age | 267 |
| abstract_inverted_index.and | 17, 31, 46, 78, 92, 112, 136, 169, 209, 239, 247, 263, 297, 328 |
| abstract_inverted_index.are | 33 |
| abstract_inverted_index.but | 104 |
| abstract_inverted_index.few | 127 |
| abstract_inverted_index.for | 171, 243, 249 |
| abstract_inverted_index.her | 181, 187, 202, 253 |
| abstract_inverted_index.its | 18 |
| abstract_inverted_index.she | 156, 221 |
| abstract_inverted_index.the | 54, 131, 163, 266, 276, 313 |
| abstract_inverted_index.was | 67, 157, 217, 222, 235 |
| abstract_inverted_index.(CD) | 73 |
| abstract_inverted_index.Case | 83 |
| abstract_inverted_index.DNA. | 331 |
| abstract_inverted_index.EBVR | 319 |
| abstract_inverted_index.T/NK | 14 |
| abstract_inverted_index.also | 138, 218 |
| abstract_inverted_index.case | 63 |
| abstract_inverted_index.cell | 99 |
| abstract_inverted_index.many | 211 |
| abstract_inverted_index.need | 273 |
| abstract_inverted_index.pain | 91 |
| abstract_inverted_index.past | 115 |
| abstract_inverted_index.rare | 293 |
| abstract_inverted_index.situ | 326 |
| abstract_inverted_index.skin | 174, 188 |
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| abstract_inverted_index.that | 66 |
| abstract_inverted_index.were | 121 |
| abstract_inverted_index.with | 160, 224, 228, 237, 288 |
| abstract_inverted_index.Based | 151 |
| abstract_inverted_index.Blood | 94, 214 |
| abstract_inverted_index.CAEBV | 65, 279, 316 |
| abstract_inverted_index.about | 177 |
| abstract_inverted_index.after | 180 |
| abstract_inverted_index.based | 74 |
| abstract_inverted_index.blood | 98, 329 |
| abstract_inverted_index.cases | 311 |
| abstract_inverted_index.cells | 45 |
| abstract_inverted_index.death | 264 |
| abstract_inverted_index.forms | 141 |
| abstract_inverted_index.found | 210 |
| abstract_inverted_index.ileal | 76, 204 |
| abstract_inverted_index.ileum | 137 |
| abstract_inverted_index.liver | 110 |
| abstract_inverted_index.major | 19 |
| abstract_inverted_index.minor | 34 |
| abstract_inverted_index.only. | 309 |
| abstract_inverted_index.organ | 261 |
| abstract_inverted_index.small | 128, 194 |
| abstract_inverted_index.these | 153 |
| abstract_inverted_index.ulcer | 205, 290, 307 |
| abstract_inverted_index.virus | 5, 12 |
| abstract_inverted_index.where | 312 |
| abstract_inverted_index.woman | 87 |
| abstract_inverted_index.CAEBV, | 296 |
| abstract_inverted_index.CAEBV. | 58 |
| abstract_inverted_index.active | 3 |
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| abstract_inverted_index.biopsy | 189, 206 |
| abstract_inverted_index.counts | 100 |
| abstract_inverted_index.course | 164 |
| abstract_inverted_index.fever. | 93 |
| abstract_inverted_index.ileum. | 133 |
| abstract_inverted_index.modest | 106 |
| abstract_inverted_index.months | 179 |
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| abstract_inverted_index.rather | 231 |
| abstract_inverted_index.should | 321 |
| abstract_inverted_index.showed | 96 |
| abstract_inverted_index.slight | 109 |
| abstract_inverted_index.tissue | 318 |
| abstract_inverted_index.ulcers | 77, 129, 175 |
| abstract_inverted_index.varied | 55 |
| abstract_inverted_index.visit. | 184 |
| abstract_inverted_index.years. | 270 |
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| abstract_inverted_index.Herein, | 59 |
| abstract_inverted_index.acquire | 302 |
| abstract_inverted_index.capsule | 149 |
| abstract_inverted_index.checked | 323 |
| abstract_inverted_index.complex | 38 |
| abstract_inverted_index.defined | 9 |
| abstract_inverted_index.disease | 52, 72, 254 |
| abstract_inverted_index.failure | 262 |
| abstract_inverted_index.initial | 182 |
| abstract_inverted_index.jejunum | 135 |
| abstract_inverted_index.leading | 258 |
| abstract_inverted_index.mixture | 39 |
| abstract_inverted_index.normal. | 122 |
| abstract_inverted_index.steroid | 168 |
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| abstract_inverted_index.ulcers, | 143 |
| abstract_inverted_index.various | 140 |
| abstract_inverted_index.whereas | 29 |
| abstract_inverted_index.without | 101 |
| abstract_inverted_index.Abstract | 0 |
| abstract_inverted_index.However, | 123 |
| abstract_inverted_index.appeared | 176 |
| abstract_inverted_index.assessed | 201 |
| abstract_inverted_index.atypical | 102, 197 |
| abstract_inverted_index.clinical | 20, 35, 79, 154 |
| abstract_inverted_index.describe | 61 |
| abstract_inverted_index.detected | 105 |
| abstract_inverted_index.diarrhea | 32 |
| abstract_inverted_index.evidence | 113 |
| abstract_inverted_index.findings | 120 |
| abstract_inverted_index.hospital | 183 |
| abstract_inverted_index.lymphoma | 227 |
| abstract_inverted_index.multiple | 260 |
| abstract_inverted_index.physical | 47 |
| abstract_inverted_index.previous | 203 |
| abstract_inverted_index.remains, | 317 |
| abstract_inverted_index.remember | 275 |
| abstract_inverted_index.revealed | 125, 190 |
| abstract_inverted_index.strongly | 158 |
| abstract_inverted_index.symptoms | 48, 80 |
| abstract_inverted_index.systemic | 23, 50, 245 |
| abstract_inverted_index.terminal | 132 |
| abstract_inverted_index.Crohn’s | 71 |
| abstract_inverted_index.Enteritis | 287 |
| abstract_inverted_index.NK/T-cell | 226 |
| abstract_inverted_index.abdominal | 90 |
| abstract_inverted_index.biologics | 170 |
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| abstract_inverted_index.diagnosis | 283, 305 |
| abstract_inverted_index.enteritis | 30, 230 |
| abstract_inverted_index.exhibited | 139 |
| abstract_inverted_index.findings, | 155 |
| abstract_inverted_index.infection | 6 |
| abstract_inverted_index.initially | 68 |
| abstract_inverted_index.lymphoma. | 251 |
| abstract_inverted_index.malignant | 250 |
| abstract_inverted_index.positive. | 219 |
| abstract_inverted_index.processes | 42 |
| abstract_inverted_index.progress, | 257 |
| abstract_inverted_index.suspected | 159 |
| abstract_inverted_index.symptoms. | 36 |
| abstract_inverted_index.treatment | 166, 208 |
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| abstract_inverted_index.Conclusion | 271 |
| abstract_inverted_index.Therefore, | 220 |
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| abstract_inverted_index.definitive | 304 |
| abstract_inverted_index.endoscopy. | 150 |
| abstract_inverted_index.enteritis, | 173 |
| abstract_inverted_index.enteritis. | 82, 286 |
| abstract_inverted_index.exhibiting | 144 |
| abstract_inverted_index.expression | 320 |
| abstract_inverted_index.extranodal | 225 |
| abstract_inverted_index.impossible | 300 |
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| abstract_inverted_index.infectious | 27 |
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| abstract_inverted_index.lymphocyte | 103 |
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| abstract_inverted_index.refractory | 172, 285 |
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| abstract_inverted_index.EBV-encoded | 193 |
| abstract_inverted_index.appearance, | 147 |
| abstract_inverted_index.cobblestone | 146 |
| abstract_inverted_index.colonoscopy | 124 |
| abstract_inverted_index.combination | 241 |
| abstract_inverted_index.constitutes | 53 |
| abstract_inverted_index.examination | 95 |
| abstract_inverted_index.possibility | 277, 314 |
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| abstract_inverted_index.cyclosporine | 238 |
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| abstract_inverted_index.inflammatory | 51 |
| abstract_inverted_index.lymphocytes. | 198, 213 |
| abstract_inverted_index.prednisolone | 240 |
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| abstract_inverted_index.EBER-positive | 212 |
| abstract_inverted_index.hybridization | 327 |
| abstract_inverted_index.inflammation, | 107 |
| abstract_inverted_index.proliferation | 191 |
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| abstract_inverted_index.Epstein–Barr | 4, 11 |
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| abstract_inverted_index.mononucleocytosis, | 28 |
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| cited_by_percentile_year.min | 93 |
| corresponding_author_ids | https://openalex.org/A5101728731 |
| countries_distinct_count | 1 |
| institutions_distinct_count | 12 |
| corresponding_institution_ids | https://openalex.org/I64926303 |
| sustainable_development_goals[0].id | https://metadata.un.org/sdg/3 |
| sustainable_development_goals[0].score | 0.8700000047683716 |
| sustainable_development_goals[0].display_name | Good health and well-being |
| citation_normalized_percentile.value | 0.67855193 |
| citation_normalized_percentile.is_in_top_1_percent | False |
| citation_normalized_percentile.is_in_top_10_percent | False |