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[PMID]:28906349
[Au] Autor:Youn JK; Park SH; Han JW; Oh C; Kim HY; Jung SE
[Ad] Endereço:Department of Pediatric Surgery, Seoul National University College of Medicine, Seoul, Korea.
[Ti] Título:Intestinal obstruction due to kaposiform hemangioendothelioma in a 1-month-old infant: A case report.
[So] Source:Medicine (Baltimore);96(37):e6974, 2017 Sep.
[Is] ISSN:1536-5964
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:RATIONALE: Kaposiform hemangioendothelioma (KHE) is an aggressive vascular tumor, mainly occurring in infants and young children and previously reported cases were mainly cutaneous or visceral form. Intestinal kaposiform hemangioma was first reported in 2012. Intestinal type KHE showed better prognosis if the lesion was limited in the gastrointestinal tract and coagulopathy was not accompanied. Since the number of reported cases is small, further study for treatment options and prognosis need to be done. PATIENT CONCERNS: We described the case of a 1-month-old female who had abdominal distention and bilious vomiting. DIAGNOSES: She was suspected as intestinal obstruction after diagnostic work up. INTERVENTIONS: Surgical exploration was performed and jejunal obstruction with a mass was identified. Small bowel segmental resection and anastomosis was performed. OUTCOMES: The patient discharged with symptom free. Through the pathological examination, the mass was identified as intestinal type KHE. LESSONS: Intestinal KHE can cause bowel obstruction and be managed successfully with complete surgical resection. More cases should be reported and further evaluation for treatment options and prognosis evaluation is necessary.
[Mh] Termos MeSH primário: Hemangioendotelioma/complicações
Hemangioendotelioma/cirurgia
Obstrução Intestinal/etiologia
Obstrução Intestinal/cirurgia
Síndrome de Kasabach-Merritt/complicações
Síndrome de Kasabach-Merritt/cirurgia
Sarcoma de Kaposi/complicações
Sarcoma de Kaposi/cirurgia
[Mh] Termos MeSH secundário: Diagnóstico Diferencial
Feminino
Hemangioendotelioma/diagnóstico
Hemangioendotelioma/patologia
Seres Humanos
Lactente
Obstrução Intestinal/diagnóstico
Obstrução Intestinal/patologia
Síndrome de Kasabach-Merritt/diagnóstico
Síndrome de Kasabach-Merritt/patologia
Sarcoma de Kaposi/diagnóstico
Sarcoma de Kaposi/patologia
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE
[Em] Mês de entrada:1709
[Cu] Atualização por classe:171004
[Lr] Data última revisão:
171004
[Sb] Subgrupo de revista:AIM; IM
[Da] Data de entrada para processamento:170915
[St] Status:MEDLINE
[do] DOI:10.1097/MD.0000000000006974


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[PMID]:28767598
[Au] Autor:Liu X; Yang Z; Tan H; Xu L; Sun Y; Si S; Liu L; Zhou W; Huang J
[Ad] Endereço:Department of Hepatobiliary Surgery, China-Japan Friendship Hospital, Beijing, China.
[Ti] Título:Giant liver hemangioma with adult Kasabach-Merritt syndrome: Case report and literature review.
[So] Source:Medicine (Baltimore);96(31):e7688, 2017 Aug.
[Is] ISSN:1536-5964
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:RATIONALE: Adult Kasabach-Merritt syndrome associated with giant liver hemangioma is rare; to date, most reports have been single-case reports, and no multi-case reports or literature reviews are available. DIAGNOSES: We conducted a retrospective analysis of 5 cases of adult Kasabach-Merritt syndrome associated with giant liver hemangioma treated at our hospital between 2011 and 2016. All 5 patients had varying severities of leukopenia, anemia, thrombocytopenia, prolonged prothrombin time, and hypofibrinogenemia. INTERVENTIONS: All the patients underwent surgery: 2 patients had left hemihepatectomy; 1 had enucleation; 1 had a right hemihepatectomy; and 1 had a left trisectionectomy. OUTCOMES: The 5 patients had an average operative time of 6.9 hours and an average blood loss of 3200 mL. One patient developed a biliary fistula (grade II) after the operation. There was no mortality among 5 patients. The white blood cell counts, hemoglobin, platelets, and prothrombin times of all 5 patients returned to normal after the operation. To date, a total of 11 cases of adult Kasabach-Merritt syndrome associated with giant liver hemangioma have been reported, of which 8 patients underwent surgery, and their platelets and coagulation returned to normal after the operation. LESSONS: Adult Kasabach-Merritt syndrome associated with giant liver hemangioma is uncommon, and surgical treatment is risky. However, resection of the tumor corrected the abnormalities in hematological and coagulative systems.
[Mh] Termos MeSH primário: Síndrome de Kasabach-Merritt/complicações
Síndrome de Kasabach-Merritt/cirurgia
Neoplasias Hepáticas/complicações
Neoplasias Hepáticas/cirurgia
[Mh] Termos MeSH secundário: Adulto
Feminino
Seres Humanos
Síndrome de Kasabach-Merritt/sangue
Síndrome de Kasabach-Merritt/diagnóstico por imagem
Fígado/diagnóstico por imagem
Fígado/cirurgia
Neoplasias Hepáticas/sangue
Neoplasias Hepáticas/diagnóstico por imagem
Masculino
Meia-Idade
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE; REVIEW
[Em] Mês de entrada:1708
[Cu] Atualização por classe:171013
[Lr] Data última revisão:
171013
[Sb] Subgrupo de revista:AIM; IM
[Da] Data de entrada para processamento:170803
[St] Status:MEDLINE
[do] DOI:10.1097/MD.0000000000007688


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[PMID]:28486787
[Au] Autor:Ji Y; Chen S; Xiang B; Li K; Xu Z; Yao W; Lu G; Liu X; Xia C; Wang Q; Li Y; Wang C; Yang K; Yang G; Tang X; Xu T; Wu H
[Ad] Endereço:Division of Oncology, Department of Pediatric Surgery, West China Hospital of Sichuan University, Chengdu, China.
[Ti] Título:Sirolimus for the treatment of progressive kaposiform hemangioendothelioma: A multicenter retrospective study.
[So] Source:Int J Cancer;141(4):848-855, 2017 Aug 15.
[Is] ISSN:1097-0215
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:Kaposiform hemangioendothelioma (KHE) is an aggressive disease with high morbidity and mortality. The aim of this study was to retrospectively evaluate the efficacy and safety of sirolimus for the treatment of progressive KHE. A multicenter, retrospective cohort study was conducted in patients with progressive KHE treated with sirolimus. A total of 52 patients were analyzed. Thirty-seven (71%) patients exhibited Kasabach-Merritt phenomenon (KMP) and were significantly younger than the patients without KMP [95% confidence interval (CI), 14.39-41.61; p < 0.001]. Patients without KMP were all treated with sirolimus alone, whereas 21 KMP patients with severe symptoms received short-term combination therapy with prednisolone. Overall, 96% and 98% of patients showed improved relief of notable symptoms and/or improved complications at 6 and 12 months after treatment, respectively. After sirolimus treatment, significant decreases in mean severity scores occurred at 6 months (95% CI, 2.23-2.54, p < 0.001) and 12 months (95% CI, 1.53-1.90, p < 0.001). Compared to KMP patients, patients without KMP showed a response that was similar to but less pronounced during the 12 months of treatment (95% CI, 40.87-53.80; p < 0.001). For subgroup analysis of KMP patients, there were no significant differences in tumor shrinkage between those treated with combination therapy and those receiving sirolimus alone (95% CI, 18.11-25.02; p > 0.05). No patients permanently discontinued treatment due to toxicity-related events, and no drug-related deaths occurred. Sirolimus was effective and safe for the treatment of progressive KHE. Sirolimus may be considered as a first-line therapy or as part of a multidisciplinary approach for the treatment of KHE.
[Mh] Termos MeSH primário: Hemangioendotelioma/tratamento farmacológico
Síndrome de Kasabach-Merritt/tratamento farmacológico
Prednisolona/administração & dosagem
Sarcoma de Kaposi/tratamento farmacológico
Sirolimo/administração & dosagem
[Mh] Termos MeSH secundário: Adolescente
Adulto
Idoso
Idoso de 80 Anos ou mais
Protocolos de Quimioterapia Combinada Antineoplásica/administração & dosagem
Protocolos de Quimioterapia Combinada Antineoplásica/uso terapêutico
Criança
Pré-Escolar
Feminino
Hemangioendotelioma/complicações
Seres Humanos
Lactente
Recém-Nascido
Síndrome de Kasabach-Merritt/complicações
Masculino
Meia-Idade
Prednisolona/uso terapêutico
Estudos Retrospectivos
Sarcoma de Kaposi/complicações
Sirolimo/uso terapêutico
Resultado do Tratamento
Adulto Jovem
[Pt] Tipo de publicação:JOURNAL ARTICLE; MULTICENTER STUDY
[Nm] Nome de substância:
9PHQ9Y1OLM (Prednisolone); W36ZG6FT64 (Sirolimus)
[Em] Mês de entrada:1709
[Cu] Atualização por classe:170915
[Lr] Data última revisão:
170915
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:170510
[St] Status:MEDLINE
[do] DOI:10.1002/ijc.30775


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[PMID]:28297758
[Au] Autor:Ke ZY; Yang SJ
[Ad] Endereço:Department of Pathology, Xijing Hospital, Fourth Military Medical University, Xi'an 710032, China.
[Ti] Título:[Role of master transcriptional factor Prox-1 in lymphatic endothelial differentiation of Kaposiform hemangioendothelioma].
[So] Source:Zhonghua Bing Li Xue Za Zhi;46(3):176-181, 2017 Mar 08.
[Is] ISSN:0529-5807
[Cp] País de publicação:China
[La] Idioma:chi
[Ab] Resumo:To analyze the clinical and pathological features of Kaposiform hemangioendothelioma (KHE), and to investigate the role of master transcriptional factor Prox-1 in the regulation of lymphatic differentiation. Nine cases of KHE (during the period from October 2009 to June 2016) were collected with clinical and pathological data. H&E stained section review and immunohistochemietry using the Dako EnVision method were performed. There were 6 female and 3 male patients with age ranging from 2 months to 8 years (median 3 years and 4 months). The patients presented with either single subcutaneous soft tissue mass, or bone tumors, with the duration of disease onset ranging from 1 month to 1 year. The sites of involvement included the skins of neck (2 cases), nose root (1 case), inguinal (1 case), thigh root (1 case), humerus (2 cases), lumbar vertebrae(1 case), and mesentery (1 case). These tumors were histologically composed of nodules of densely packed spindle or ovoid cells and deformed small blood vessels in an invasive growth pattern. The tumor cells were immunohistochemically positive for both blood vessels and lymphatic endothelial markers, including Prox-1, the master transcriptional factor, and VEGFR-3. With followed-up from 1 to 60 months (median 26 months), two patients died of the disease, while the remaining patients were alive without recurrence. KHE is a rare vascular tumor with at least partial lymphatic endothelial differentiation, in which Prox-1 may act as a master regulator for such differentiation. KHE is an aggressive tumor of intermediate malignant potential, with local invasion and recurrence tendency, and long term follow-up is required.
[Mh] Termos MeSH primário: Neoplasias Ósseas/patologia
Hemangioendotelioma/genética
Hemangioendotelioma/patologia
Proteínas de Homeodomínio/genética
Síndrome de Kasabach-Merritt/genética
Síndrome de Kasabach-Merritt/patologia
Sarcoma de Kaposi/genética
Sarcoma de Kaposi/patologia
Neoplasias Cutâneas/patologia
Proteínas Supressoras de Tumor/genética
Receptor 3 de Fatores de Crescimento do Endotélio Vascular/genética
[Mh] Termos MeSH secundário: Biomarcadores Tumorais
Criança
Pré-Escolar
Feminino
Marcadores Genéticos
Fator 1-alfa Nuclear de Hepatócito
Proteínas de Homeodomínio/metabolismo
Seres Humanos
Masculino
Recidiva Local de Neoplasia
Proteínas Supressoras de Tumor/metabolismo
Receptor 3 de Fatores de Crescimento do Endotélio Vascular/metabolismo
Neoplasias Vasculares/genética
Neoplasias Vasculares/patologia
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Nm] Nome de substância:
0 (Biomarkers, Tumor); 0 (Genetic Markers); 0 (Hepatocyte Nuclear Factor 1-alpha); 0 (Homeodomain Proteins); 0 (Tumor Suppressor Proteins); 0 (prospero-related homeobox 1 protein); EC 2.7.10.1 (FLT4 protein, human); EC 2.7.10.1 (Vascular Endothelial Growth Factor Receptor-3)
[Em] Mês de entrada:1707
[Cu] Atualização por classe:170724
[Lr] Data última revisão:
170724
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:170316
[St] Status:MEDLINE
[do] DOI:10.3760/cma.j.issn.0529-5807.2017.03.007


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[PMID]:28102622
[Au] Autor:Pascal S; Bettex Q; Andre N; Petit P; Casanova D; Degardin N
[Ad] Endereço:Pediatric Plastic Surgery Department, Timone Enfants University Hospital, Marseille, France.
[Ti] Título:Successful surgical management of congenital Kasabach-Merritt syndrome.
[So] Source:Pediatr Int;59(1):89-92, 2017 Jan.
[Is] ISSN:1442-200X
[Cp] País de publicação:Australia
[La] Idioma:eng
[Ab] Resumo:Since the first description of Kasabach-Merritt syndrome (KMS) in 1940, many treatments have been proposed combining pharmacologic and non-pharmacologic approaches, which can be effective on the pathology but can have adverse and unpredictable side-effects with long-term use. Herein we describe the solely surgical treatment of Kasabach-Merritt syndrome in a neonate with a severe and rapidly progressive thrombocytopenia. The patient's condition normalized at 7 days postoperatively, with rapid increase in platelet count and normalization of d-dimer concentration.
[Mh] Termos MeSH primário: Síndrome de Kasabach-Merritt/cirurgia
[Mh] Termos MeSH secundário: Seres Humanos
Recém-Nascido
Síndrome de Kasabach-Merritt/complicações
Masculino
Trombocitopenia/complicações
Resultado do Tratamento
Ultrassonografia Doppler
[Pt] Tipo de publicação:CASE REPORTS
[Em] Mês de entrada:1705
[Cu] Atualização por classe:170510
[Lr] Data última revisão:
170510
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:170120
[St] Status:MEDLINE
[do] DOI:10.1111/ped.13171


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[PMID]:28027747
[Au] Autor:Ryu YJ; Choi YH; Cheon JE; Kim WS; Kim IO; Park JE; Kim YJ
[Ad] Endereço:Department of Radiology, Seoul National University Hospital, Seoul, Republic of Korea; Department of Radiology, Seoul National University College of Medicine, Seoul, Republic of Korea.
[Ti] Título:Imaging findings of Kaposiform Hemangioendothelioma in children.
[So] Source:Eur J Radiol;86:198-205, 2017 Jan.
[Is] ISSN:1872-7727
[Cp] País de publicação:Ireland
[La] Idioma:eng
[Ab] Resumo:PURPOSE: Kaposiform hemangioendothelioma (KHE) is a rare, aggressive vascular tumor that typically occurs during infancy or early childhood. Though several case reports have discussed the imaging findings of KHE, larger comprehensive studies are lacking. The purpose of this study was to evaluate the imaging findings of KHE in children. MATERIALS AND METHODS: A total of twelve cases of pathologically proven KHE were collected by searching our institution's pathology database for children diagnosed between January 2004 and April 2016 (6 male, 6 female; median age: 3 months; age range 7days - 18 years). CT (n=7) and MRI (n=9) findings were retrospectively evaluated. The location, morphology, enhancement, and growth pattern were analyzed. RESULTS: KHEs involved various locations: superficial or deep soft tissue of the extremities (n=4); abdomen (n=3; 2 cases, pancreas; 1 case, small bowel), neck and mediastinum (n=1); chest wall, diaphragm, and pericardium (n=1); abdominal wall (n=1); and head (IAC, CP angle) (n=2). Eight of 10 cases (80%) with CT/MR findings were locally invasive and involved two or more adjacent organs. Three cases presented as well-defined solid masses, and seven were associated with infiltrative lesions of the surrounding areas with (n=4) or without definite solid regions (n=3). In nine patients with MRI, all KHEs were heterogeneous and hyperintense to muscles on T2-weighted images (T2WI), while four KHEs consisted of some regions that were nearly isointense to muscle. Eight of the 10 cases with imaging exhibited heterogeneous intense enhancement, while only one demonstrated mild enhancement. Signal voids (n=2), engorged vessels (n=1), calcification (n=3), hemorrhage (n=1), or bone changes (n=4) were infrequently observed. Four patients (33%) had Kasabach-Merritt phenomenon, and recurrence was observed in two cases. CONCLUSION: KHEs occurred in various locations, affected mostly infants, and generally exhibited intense heterogeneous enhancement. In more than half of the included cases, KHEs were highly infiltrative and locally invasive with ill-defined margins. Awareness of these features should prompt radiologists to include KHE in the differential diagnosis for pediatric masses.
[Mh] Termos MeSH primário: Neoplasias de Cabeça e Pescoço/patologia
Hemangioendotelioma/patologia
Síndrome de Kasabach-Merritt/patologia
Sarcoma de Kaposi/patologia
Neoplasias Cutâneas/patologia
Neoplasias Vasculares/patologia
[Mh] Termos MeSH secundário: Adolescente
Criança
Pré-Escolar
Diagnóstico Diferencial
Feminino
Seres Humanos
Lactente
Recém-Nascido
Imagem por Ressonância Magnética
Masculino
Imagem Multimodal
Recidiva Local de Neoplasia/patologia
Estudos Retrospectivos
Tomografia Computadorizada por Raios X
[Pt] Tipo de publicação:EVALUATION STUDIES; JOURNAL ARTICLE
[Em] Mês de entrada:1703
[Cu] Atualização por classe:170817
[Lr] Data última revisão:
170817
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161229
[St] Status:MEDLINE


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[PMID]:27723921
[Au] Autor:Triana P; Dore M; Cerezo VN; Cervantes M; Sánchez AV; Ferrero MM; González MD; Lopez-Gutierrez JC
[Ad] Endereço:Department of Pediatric Surgery, Hospital Universitario La Paz, Madrid, Spain.
[Ti] Título:Sirolimus in the Treatment of Vascular Anomalies.
[So] Source:Eur J Pediatr Surg;27(1):86-90, 2017 Feb.
[Is] ISSN:1439-359X
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:mTOR inhibitors are showing promising results in the management of vascular anomalies. Although current controlled trials remain to be completed, many individual experiences are being published. We present our series of children with complex vascular anomalies treated with sirolimus. A retrospective review of 41 patients treated with sirolimus between January 2011 and December 2015 was performed: 15% ( = 6) had vascular tumors (4 kaposiform hemangioendotheliomas, 1 PTEN) and 85% ( = 35) had malformations (13 generalized lymphatic anomalies/Gorham-Stout diseases [GSD], 1 kaposiform lymphangiomatosis [KLA], 11 large lymphatic malformations (LMs) in critical areas, 2 lymphedemas, 4 venous malformations, and 4 aggressive arteriovenous malformations [AVM]). Several variables were collected: type of vascular anomaly, duration of treatment, dosage, response, and secondary effects. There was a female predominance (1.4:1). All patients received sirolimus, at initial dosage of 0.8 mg/m /12 hour. Overall successful response rate was 80.4% of cases, presenting improvement in radiologic imaging and reduction of symptoms, at a median time of 10 weeks. Patients showing no response included four AVMs, one GSD, one LM, one KLA, and one unknown tumor. Sirolimus was well tolerated, even in neonates, with insignificant side effects. No patients had complete resolution and no patients worsened on therapy. Thirty patients remain under treatment at the present moment. Sirolimus has become a new therapeutic option for patients with vascular anomalies that do not respond to other treatments. Unfortunately, important questions as what is the most appropriate dosage and for how long should the patient be treated remain unanswered. An international registry followed by customized controlled trials is mandatory to clarify the future of this therapy.
[Mh] Termos MeSH primário: Hemangioendotelioma/tratamento farmacológico
Síndrome de Kasabach-Merritt/tratamento farmacológico
Anormalidades Linfáticas/tratamento farmacológico
Inibidores de Proteínas Quinases/uso terapêutico
Sarcoma de Kaposi/tratamento farmacológico
Sirolimo/uso terapêutico
Malformações Vasculares/tratamento farmacológico
[Mh] Termos MeSH secundário: Adolescente
Adulto
Criança
Pré-Escolar
Feminino
Seguimentos
Seres Humanos
Lactente
Masculino
Meia-Idade
Estudos Retrospectivos
Resultado do Tratamento
Adulto Jovem
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Nm] Nome de substância:
0 (Protein Kinase Inhibitors); W36ZG6FT64 (Sirolimus)
[Em] Mês de entrada:1707
[Cu] Atualização por classe:170705
[Lr] Data última revisão:
170705
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161011
[St] Status:MEDLINE
[do] DOI:10.1055/s-0036-1593383


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[PMID]:27701822
[Au] Autor:Schaefer BA; Wang D; Merrow AC; Dickie BH; Adams DM
[Ad] Endereço:Cancer and Blood Disease Institute, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio.
[Ti] Título:Long-term outcome for kaposiform hemangioendothelioma: A report of two cases.
[So] Source:Pediatr Blood Cancer;64(2):284-286, 2017 Feb.
[Is] ISSN:1545-5017
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:Kaposiform hemangioendothelioma (KHE) is a rare aggressive vascular tumor of skin and deep soft tissues that typically presents in infancy and may be associated with a potentially life-threatening coagulopathy known as Kasabach-Merrit phenomenon (KMP). Recent advances in medical therapy have successfully treated many patients. However, our knowledge regarding the natural history of these lesions and optimum surveillance strategies remains rudimentary. We report two young women who had KHE with KMP treated in infancy and presented in adolescence with comorbidities related to their KHE tumor. This presentation supports the need for long-term surveillance in these patients.
[Mh] Termos MeSH primário: Hemangioendotelioma/mortalidade
Hemangioendotelioma/terapia
Síndrome de Kasabach-Merritt/mortalidade
Síndrome de Kasabach-Merritt/terapia
Sarcoma de Kaposi/mortalidade
Sarcoma de Kaposi/terapia
[Mh] Termos MeSH secundário: Terapia Combinada
Feminino
Hemangioendotelioma/patologia
Seres Humanos
Lactente
Síndrome de Kasabach-Merritt/patologia
Prognóstico
Sarcoma de Kaposi/patologia
Taxa de Sobrevida
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE
[Em] Mês de entrada:1706
[Cu] Atualização por classe:170621
[Lr] Data última revisão:
170621
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161005
[St] Status:MEDLINE
[do] DOI:10.1002/pbc.26224


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[PMID]:27571125
[Au] Autor:Dapaah-Siakwan F; Bryan C; Walker LS; Del Moral T
[Ad] Endereço:Department of Pediatrics, Division of Neonatology, University of Miami, Holtz Children's Hospital, Miami, FL.
[Ti] Título:A Rare Presentation of Isolated Congenital Splenic Hemangioma With Kasabach-Merritt Syndrome.
[So] Source:J Pediatr Hematol Oncol;39(1):e29-e32, 2017 Jan.
[Is] ISSN:1536-3678
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:INTRODUCTION: Splenic hemangiomas (SHs) are the most common benign neoplasms of the spleen. However, they are rare in the newborn period. We present an extremely rare case of congenital SH complicated by Kasabach-Merritt syndrome. CASE PRESENTATION: A 2.93 kg male infant was delivered at term with a prenatal diagnosis of a left infrarenal mass diagnosed by ultrasound at 35 weeks of gestation. Magnetic resonance imaging demonstrated a well-defined splenic mass with multiple flow voids and scattered areas of high intensity suggestive of hemorrhage. He developed anemia, thrombocytopenia, and coagulopathy which required transfusion with packed red cells, platelets, cryoprecipitate, and fresh frozen plasma. Excision biopsy of the spleen led to resolution of anemia, thrombocytopenia, and coagulopathy. The diagnosis of SH was confirmed by histopathology. At 2 months outpatient follow-up, the patient was growing well without any evidence of tumor recurrence. CONCLUSIONS: Congenital SH is a rare entity that can be fatal if the potential complication of Kasabach-Merritt syndrome is not anticipated, evaluated, and promptly treated. Our patient had a favorable outcome with early surgical excision of the SH.
[Mh] Termos MeSH primário: Hemangioma/congênito
Síndrome de Kasabach-Merritt/etiologia
Neoplasias Esplênicas/congênito
[Mh] Termos MeSH secundário: Anemia/etiologia
Transfusão de Componentes Sanguíneos
Diagnóstico Diferencial
Coagulação Intravascular Disseminada/etiologia
Coagulação Intravascular Disseminada/terapia
Diagnóstico Precoce
Hemangioma/complicações
Hemangioma/diagnóstico por imagem
Hemangioma/cirurgia
Seres Humanos
Recém-Nascido
Síndrome de Kasabach-Merritt/diagnóstico
Imagem por Ressonância Magnética
Masculino
Sepse/diagnóstico
Esplenectomia
Neoplasias Esplênicas/complicações
Neoplasias Esplênicas/diagnóstico por imagem
Neoplasias Esplênicas/cirurgia
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE
[Em] Mês de entrada:1709
[Cu] Atualização por classe:170904
[Lr] Data última revisão:
170904
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:160830
[St] Status:MEDLINE


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[PMID]:28152371
[Au] Autor:Serra JT; Orozco MJ; Prato FJ; Sánchez V; Basqueira AJ; Brocca CA
[Ti] Título:[Myelophthisis and kasabach merrit syndrome as initial manifestation of splenic angiosarcoma].
[So] Source:Rev Fac Cien Med Univ Nac Cordoba;73(4):297-301, 2016.
[Is] ISSN:1853-0605
[Cp] País de publicação:Argentina
[La] Idioma:spa
[Ab] Resumo:Primary splenic angiosarcoma is an extremely agressive and rare neoplasm. Manifestations as bone marrow invasion and coagulation disorders have been reported isolatedly. A 26 years-old woman presented with abdominal pain; several anemia and thrombocytopenia associated to leukoerythroblastic reaction were found in the laboratory. Consumpion coagulopathy signs and microangiopathy as schistocytes, prolonged prothrombine time, decreased fibrinogen and increased D dimer were also present. Imaging findings included a lobulated, enlarged spleen, with spontanously hyperdense areas, and heterogeneous nodules with intense, irregular enhancement after contrast administration. There were hepatic and pulmonary metastases, as well as bone lesions with conspicuous vessels. Clinical features of Kasabach-Merrit syndrome and imaging vascular neoplasm characteristics suggest a primary splenic angiosarcoma. Splenectomy and bone marrow biopsy confirmed the diagnosis of primary splenic angiosarcoma in metastatic stage.
[Mh] Termos MeSH primário: Anemia Mielopática/etiologia
Hemangiossarcoma/complicações
Síndrome de Kasabach-Merritt/etiologia
Neoplasias Esplênicas/complicações
[Mh] Termos MeSH secundário: Adulto
Anemia Mielopática/diagnóstico
Biópsia
Feminino
Hemangiossarcoma/diagnóstico
Hemangiossarcoma/cirurgia
Seres Humanos
Síndrome de Kasabach-Merritt/diagnóstico
Esplenectomia
Neoplasias Esplênicas/diagnóstico
Neoplasias Esplênicas/cirurgia
[Pt] Tipo de publicação:CASE REPORTS; JOURNAL ARTICLE
[Em] Mês de entrada:1705
[Cu] Atualização por classe:170503
[Lr] Data última revisão:
170503
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:170203
[St] Status:MEDLINE



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