Publicação
Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association
| Resumo: | Von Willebrand disease (vWD) is the most prevalent hereditary bleeding disorder, affecting 0.6-1.3% of the population. While gastrointestinal bleeding from angiodysplasia is a well-known complication of vWD, the same is not true for Dieulafoy's lesions (DLs). We report the case of a 21-year-old black male with type 1 vWD and 2 previous hospital admissions for severe anemia with no visible blood loss. In both episodes, DLs were identified and treated endoscopically, one in the stomach and another in the duodenum. The patient presented to the emergency department in September 2016 with dizziness, fatigue, and again no visible blood loss. He was hemodynamically stable, and laboratory workup showed a hemoglobin level of 3.4 g/dL. After transfusion of packed red blood cells, intravenous iron, and von Willebrand factor/factor VIII concentrate infusions, the patient underwent upper endoscopy and colonoscopy, which were normal. Small-bowel capsule endoscopy showed dark blood and a fresh clot in the proximal jejunum. At this site, push enteroscopy identified a pulsatile vessel with an overlying minimal mucosal defect, consistent with a DL, type 2b of the Yano-Yamamoto classification, which was successfully treated with adrenaline and 2 hemoclips. The patient remains stable after 18 months of follow-up, with a hemoglobin level of 13.2 g/dL. This is a case of recurrent severe occult gastrointestinal bleeding from multiple DL in a young patient with vWD who is otherwise healthy. Three other cases of DL bleeding in the setting of vWD have been reported in the literature, suggesting a possible association between these 2 entities. |
|---|---|
| Autores principais: | Cardoso, M |
| Outros Autores: | Lourenço, L; Antunes, M; Branco, J; Santos, L; Martins, A; Reis, J |
| Assunto: | Gastrointestinal hemorrhage von Willebrand diseases Dieulafoy's lesion Capsule endoscopy |
| Ano: | 2019 |
| País: | Portugal |
| Tipo de documento: | artigo |
| Tipo de acesso: | acesso aberto |
| Instituição associada: | Hospital Prof. Dr. Fernando Fonseca E.P.E. |
| Idioma: | inglês |
| Origem: | Repositório do Hospital Prof. Doutor Fernando Fonseca |
| _version_ | 1868417261724762112 |
|---|---|
| author | Cardoso, M |
| author2 | Lourenço, L Antunes, M Branco, J Santos, L Martins, A Reis, J |
| author2_role | author author author author author author |
| author_facet | Cardoso, M Lourenço, L Antunes, M Branco, J Santos, L Martins, A Reis, J |
| author_role | author |
| contributor_name_str_mv | Unidade Local de Saúde Amadora / Sintra |
| country_str | PT |
| creators_json_txt | [{\"Person.name\":\"Cardoso, M\"},{\"Person.name\":\"Lourenço, L\"},{\"Person.name\":\"Antunes, M\"},{\"Person.name\":\"Branco, J\"},{\"Person.name\":\"Santos, L\"},{\"Person.name\":\"Martins, A\"},{\"Person.name\":\"Reis, J\"}] |
| datacite.contributors.contributor.contributorName.fl_str_mv | Unidade Local de Saúde Amadora / Sintra |
| datacite.creators.creator.creatorName.fl_str_mv | Cardoso, M Lourenço, L Antunes, M Branco, J Santos, L Martins, A Reis, J |
| datacite.date.Accepted.fl_str_mv | 2019-01-01T00:00:00Z |
| datacite.date.available.fl_str_mv | 2019-12-17T14:54:47Z |
| datacite.date.embargoed.fl_str_mv | 2019-12-17T14:54:47Z |
| datacite.rights.fl_str_mv | http://purl.org/coar/access_right/c_abf2 |
| datacite.subjects.subject.fl_str_mv | Gastrointestinal hemorrhage von Willebrand diseases Dieulafoy's lesion Capsule endoscopy |
| datacite.titles.title.fl_str_mv | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association |
| dc.contributor.none.fl_str_mv | Unidade Local de Saúde Amadora / Sintra |
| dc.creator.none.fl_str_mv | Cardoso, M Lourenço, L Antunes, M Branco, J Santos, L Martins, A Reis, J |
| dc.date.Accepted.fl_str_mv | 2019-01-01T00:00:00Z |
| dc.date.available.fl_str_mv | 2019-12-17T14:54:47Z |
| dc.date.embargoed.fl_str_mv | 2019-12-17T14:54:47Z |
| dc.format.none.fl_str_mv | application/pdf |
| dc.identifier.none.fl_str_mv | http://hdl.handle.net/10400.10/2362 |
| dc.language.none.fl_str_mv | eng |
| dc.publisher.none.fl_str_mv | Karger Publishers |
| dc.rights.none.fl_str_mv | http://purl.org/coar/access_right/c_abf2 |
| dc.subject.none.fl_str_mv | Gastrointestinal hemorrhage von Willebrand diseases Dieulafoy's lesion Capsule endoscopy |
| dc.title.fl_str_mv | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association |
| dc.type.none.fl_str_mv | http://purl.org/coar/resource_type/c_6501 |
| description | Von Willebrand disease (vWD) is the most prevalent hereditary bleeding disorder, affecting 0.6-1.3% of the population. While gastrointestinal bleeding from angiodysplasia is a well-known complication of vWD, the same is not true for Dieulafoy's lesions (DLs). We report the case of a 21-year-old black male with type 1 vWD and 2 previous hospital admissions for severe anemia with no visible blood loss. In both episodes, DLs were identified and treated endoscopically, one in the stomach and another in the duodenum. The patient presented to the emergency department in September 2016 with dizziness, fatigue, and again no visible blood loss. He was hemodynamically stable, and laboratory workup showed a hemoglobin level of 3.4 g/dL. After transfusion of packed red blood cells, intravenous iron, and von Willebrand factor/factor VIII concentrate infusions, the patient underwent upper endoscopy and colonoscopy, which were normal. Small-bowel capsule endoscopy showed dark blood and a fresh clot in the proximal jejunum. At this site, push enteroscopy identified a pulsatile vessel with an overlying minimal mucosal defect, consistent with a DL, type 2b of the Yano-Yamamoto classification, which was successfully treated with adrenaline and 2 hemoclips. The patient remains stable after 18 months of follow-up, with a hemoglobin level of 13.2 g/dL. This is a case of recurrent severe occult gastrointestinal bleeding from multiple DL in a young patient with vWD who is otherwise healthy. Three other cases of DL bleeding in the setting of vWD have been reported in the literature, suggesting a possible association between these 2 entities. |
| dirty | 0 |
| eu_rights_str_mv | openAccess |
| format | article |
| fulltext.url.fl_str_mv | https://repositorio.hff.min-saude.pt/bitstreams/92417d8c-e5e6-4684-a4e0-5e580ca608ce/download |
| id | rhff_fb11abd41a58c7c9926af7b577cdc19b |
| identifier.url.fl_str_mv | http://hdl.handle.net/10400.10/2362 |
| instacron_str | hff |
| institution | Hospital Prof. Dr. Fernando Fonseca E.P.E. |
| instname_str | Hospital Prof. Dr. Fernando Fonseca E.P.E. |
| language | eng |
| network_acronym_str | rhff |
| network_name_str | Repositório do Hospital Prof. Doutor Fernando Fonseca |
| oai_identifier_str | oai:repositorio.hff.min-saude.pt:10400.10/2362 |
| organization_str_mv | urn:organizationAcronym:hff |
| person_str_mv | Cardoso, M Lourenço, L Antunes, M Branco, J Santos, L Martins, A Reis, J |
| publishDate | 2019 |
| publisher.none.fl_str_mv | Karger Publishers |
| reponame_str | Repositório do Hospital Prof. Doutor Fernando Fonseca |
| repository_id_str | urn:repositoryAcronym:rhff |
| service_str_mv | urn:repositoryAcronym:rhff |
| spelling | engKarger Publisherspt_PTVon Willebrand disease (vWD) is the most prevalent hereditary bleeding disorder, affecting 0.6-1.3% of the population. While gastrointestinal bleeding from angiodysplasia is a well-known complication of vWD, the same is not true for Dieulafoy's lesions (DLs). We report the case of a 21-year-old black male with type 1 vWD and 2 previous hospital admissions for severe anemia with no visible blood loss. In both episodes, DLs were identified and treated endoscopically, one in the stomach and another in the duodenum. The patient presented to the emergency department in September 2016 with dizziness, fatigue, and again no visible blood loss. He was hemodynamically stable, and laboratory workup showed a hemoglobin level of 3.4 g/dL. After transfusion of packed red blood cells, intravenous iron, and von Willebrand factor/factor VIII concentrate infusions, the patient underwent upper endoscopy and colonoscopy, which were normal. Small-bowel capsule endoscopy showed dark blood and a fresh clot in the proximal jejunum. At this site, push enteroscopy identified a pulsatile vessel with an overlying minimal mucosal defect, consistent with a DL, type 2b of the Yano-Yamamoto classification, which was successfully treated with adrenaline and 2 hemoclips. The patient remains stable after 18 months of follow-up, with a hemoglobin level of 13.2 g/dL. This is a case of recurrent severe occult gastrointestinal bleeding from multiple DL in a young patient with vWD who is otherwise healthy. Three other cases of DL bleeding in the setting of vWD have been reported in the literature, suggesting a possible association between these 2 entities.application/pdfpt_PTRecurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare AssociationCardoso, MLourenço, LAntunes, MBranco, JSantos, LMartins, AReis, JHostingInstitutionOrganizationalUnidade Local de Saúde Amadora / Sintrae-mailmailto:repositorio@hff.min-saude.ptrepositorio@hff.min-saude.ptISSNIsPartOf2387-1954DOIIsPartOf10.1159/0004909212019-12-17T14:54:47Z20192019-01-01T00:00:00ZHandlehttp://hdl.handle.net/10400.10/2362http://purl.org/coar/access_right/c_abf2open accessGastrointestinal hemorrhagevon Willebrand diseasesDieulafoy's lesionCapsule endoscopy417291 bytesliteraturehttp://purl.org/coar/resource_type/c_6501journal articlehttp://purl.org/coar/access_right/c_abf2application/pdffulltexthttps://repositorio.hff.min-saude.pt/bitstreams/92417d8c-e5e6-4684-a4e0-5e580ca608ce/downloadGE Portuguese journal of gastroenterologyBasel |
| spellingShingle | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association Cardoso, M Gastrointestinal hemorrhage von Willebrand diseases Dieulafoy's lesion Capsule endoscopy |
| status | SINGLETON |
| subject.fl_str_mv | Gastrointestinal hemorrhage von Willebrand diseases Dieulafoy's lesion Capsule endoscopy |
| title | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association |
| title_full | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association |
| title_fullStr | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association |
| title_full_unstemmed | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association |
| title_short | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association |
| title_sort | Recurrent Gastrointestinal Bleeding from Dieulafoy's Lesions in a Patient with Type 1 von Willebrand Disease: A Rare Association |
| topic | Gastrointestinal hemorrhage von Willebrand diseases Dieulafoy's lesion Capsule endoscopy |
| topic_facet | Gastrointestinal hemorrhage von Willebrand diseases Dieulafoy's lesion Capsule endoscopy |
| url | http://hdl.handle.net/10400.10/2362 |
| visible | 1 |