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2.
Medicine (Baltimore) ; 103(6): e37137, 2024 Feb 09.
Artigo em Inglês | MEDLINE | ID: mdl-38335421

RESUMO

RATIONALE: Intraosseous hemangioma is a rare benign vascular tumor of the bone that can affect any body part; however, the most common site is the vertebra, followed by calvarial bones. PATIENT CONCERNS: We present a case of intraosseous hemangioma in a 23-year-old male who presented a feeling of fullness in the throat for 3 months. The hyoid bone level had a hard mass of about 5 cm. Fine needle aspiration showed 5 mL dark bloody aspirates. Magnetic resonance image showed a 5.3 cm mixed signal intensity lesion in the hyoid body. DIAGNOSIS: Histopathologic examination showed intraosseous hemangioma with aneurysmal bone cyst (ABC)-like changes in the hyoid bone. INTERVENTIONS: The mass was completely removed without significant problems. OUTCOMES: Complete mass excision and symptomatic improvements were achieved, and no subsequent relapses were observed. LESSONS: The authors experienced a case of intraosseous hemangioma with ABC-like changes. There has been no case report of intraosseous hemangioma in the hyoid bone. This case showed a spectral pattern of the ABC-like changes developing from the underlying bone tumor as a secondary change. ABC-like changes in bone tumors can mislead the diagnosis. Careful examination of the tumor is essential for the correct diagnosis of ABC or ABC-like changes.


Assuntos
Cistos Ósseos Aneurismáticos , Neoplasias Ósseas , Hemangioma , Lesões do Pescoço , Crânio/anormalidades , Coluna Vertebral/anormalidades , Malformações Vasculares , Neoplasias Vasculares , Masculino , Humanos , Adulto Jovem , Adulto , Osso Hioide/diagnóstico por imagem , Osso Hioide/cirurgia , Cistos Ósseos Aneurismáticos/diagnóstico por imagem , Cistos Ósseos Aneurismáticos/cirurgia , Crânio/patologia , Neoplasias Ósseas/diagnóstico por imagem , Neoplasias Ósseas/cirurgia , Hemangioma/diagnóstico por imagem , Hemangioma/cirurgia , Coluna Vertebral/patologia
3.
J Pak Med Assoc ; 74(2): 394-397, 2024 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-38419245

RESUMO

Sturge- Weber syndrome (SWS), is a rare neuro-cutaneous angiomatosis which affects male and females alike. The clinical manifestations include angiomas, haemangiomas of the lips, tongue and palatine region. The oral manifestations are usually unilateral and are susceptible to bleed. Patients can also present with macroglossia and maxillary bone hypertrophy which can lead to malocclusion of the oral cavity. Food accumulation due to occlusion can cause growth of bacteria which can intensify infections and can cause gingival hyperplasia. A case of a middle-aged 39 year old female was reported in the Ziauddin Hospital, Karachi on 2nd of February,2022 with the presenting complaints of intermittent fever and drowsiness for 10 days. On examination she had massive tongue enlargement, drooling, malocclusion, difficulty in eating and breathing. She was a known case of Sturgeweber syndrome. Based on the clinical and radiological findings, she was managed along the lines of prelaryngeal soft tissue and submandibular infection.


Assuntos
Hemangioma , Macroglossia , Macroglossia/congênito , Má Oclusão , Síndrome de Sturge-Weber , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Adulto , Síndrome de Sturge-Weber/complicações , Síndrome de Sturge-Weber/diagnóstico , Macroglossia/etiologia , Hipertrofia
4.
Indian J Pathol Microbiol ; 67(1): 102-106, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-38358197

RESUMO

Background: Splenic sclerosing angiomatoid nodular transformation (SANT) is a rare benign nodular lesion in the red medulla of the spleen. In the past, SANT has not been consistently recognized as the name for this condition and was often misdiagnosed for other conditions. In recent years, SANT has been acknowledged by most scholars as multiple reports have been published. Aim: To assess the clinicopathological features of SANT to identify the histological characteristics of SANT to improve diagnosis and clinical treatment. Materials and Methods: We assessed 25 cases of SANT diagnosed at Zhongshan Hospital affiliated with Fudan University from September 2014 to October 2021, including 14 men and 11 women, aged 24-62 years old. Results: Fourteen cases were complicated with benign tumors of the liver, pancreas, kidney, uterus, and prostate. One case was complicated with renal clear cell carcinoma, and one was complicated with hepatocellular carcinoma. The gross neoplasm is multinodular and well defined. Histologically, angiomatoid nodules are composed of fattened, round, or irregular blood vessels, with or without red blood cells in the lumen, with unequal red blood cell extravasation, and fibrocytes around the nodules. The hemangiomatous nodules were positive for CD31 and CD34, while the vascular wall smooth muscle cells and fibrocytes around the nodules were positive for SMA. Conclusion: The diagnosis of SANT requires a combination of immunohistochemical and histological features, and early splenectomy is crucial for treatment.


Assuntos
Hemangioma , Histiocitoma Fibroso Benigno , Esplenopatias , Neoplasias Esplênicas , Masculino , Humanos , Feminino , Adulto Jovem , Adulto , Pessoa de Meia-Idade , Esplenopatias/diagnóstico , Esplenopatias/cirurgia , Esplenopatias/patologia , Esplenectomia , Hemangioma/diagnóstico , Hemangioma/patologia , Neoplasias Esplênicas/diagnóstico , Neoplasias Esplênicas/cirurgia , Neoplasias Esplênicas/patologia
5.
Einstein (Sao Paulo) ; 22: eRC0267, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-38324846

RESUMO

Littoral cell angioma is an extremely rare splenic vascular tumor originating from the cells lining the splenic red pulp sinuses. Approximately 150 cases of littoral cell angioma have been reported since 1991. Its clinical manifestation is usually asymptomatic and is mostly diagnosed as an incidental finding through abdominal imaging. Herein, we present a case of littoral cell angioma in a 41-year-old woman with no previous comorbidities, which initially presented as a nonspecific splenic lesion diagnosed on imaging in the emergency room. The patient was treated through laparoscopic intervention.


Assuntos
Hemangioma , Neoplasias Esplênicas , Humanos , Feminino , Adulto , Neoplasias Esplênicas/diagnóstico por imagem , Neoplasias Esplênicas/cirurgia , Hemangioma/diagnóstico por imagem , Hemangioma/cirurgia , Tomografia Computadorizada por Raios X/métodos
6.
Rev Med Liege ; 79(2): 65-67, 2024 Feb.
Artigo em Francês | MEDLINE | ID: mdl-38356420

RESUMO

Subglottic haemangioma can cause stridor in young children, and sometimes be life-threatening. Larynx ultrasound is a useful, non-irradiating screening test, but the diagnosis must be confirmed by bronchial fibroscopy and injected chest CT scan. Nowadays propranolol is the first-line treatment. If treated early, the prognosis is excellent.


L'hémangiome sous-glottique peut être responsable d'un stridor chez le jeune enfant et, parfois, menacer le pronostic vital. L'échographie du larynx est un examen utile et non irradiant pour le dépistage, mais le diagnostic sera confirmé par une fibroscopie bronchique et un scanner thoracique avec injection de produit de contraste. Le traitement en première intention est le propranolol. Lors d'une prise en charge précoce, le pronostic est excellent.


Assuntos
Hemangioma , Neoplasias Laríngeas , Criança , Humanos , Lactente , Pré-Escolar , Traqueia , Sons Respiratórios/etiologia , Propranolol/uso terapêutico , Hemangioma/complicações , Hemangioma/diagnóstico , Resultado do Tratamento , Neoplasias Laríngeas/complicações , Neoplasias Laríngeas/diagnóstico
7.
Cells ; 13(3)2024 Jan 29.
Artigo em Inglês | MEDLINE | ID: mdl-38334645

RESUMO

We previously published that in patients with infantile hemangioma (IH) at the onset (T0) colony forming unit-fibroblasts (CFU-Fs) are present in in vitro cultures from PB. Herein, we characterize these CFU-Fs and investigate their potential role in IH pathogenesis, before and after propranolol therapy. The CFU-F phenotype (by flow cytometry), their differentiation capacity and ability to support angiogenesis (by in vitro cultures) and their gene expression (by RT-PCR) were evaluated. We found that CFU-Fs are actual circulating MSCs (cMSCs). In patients at T0, cMSCs had reduced adipogenic potential, supported the formation of tube-like structures in vitro and showed either inflammatory (IL1ß and ESM1) or angiogenic (F3) gene expression higher than that of cMSCs from CTRLs. In patients receiving one-year propranolol therapy, the cMSC differentiation in adipocytes improved, while their support in in vitro tube-like formation was lost; no difference was found between patient and CTRL cMSC gene expressions. In conclusion, in patients with IH at T0 the cMSC reduced adipogenic potential, their support in angiogenic activity and the inflammatory/angiogenic gene expression may fuel the tumor growth. One-year propranolol therapy modifies this picture, suggesting cMSCs as one of the drug targets.


Assuntos
Hemangioma , Células-Tronco Mesenquimais , Humanos , Propranolol/farmacologia , Propranolol/uso terapêutico , Propranolol/metabolismo , Transcriptoma , Células-Tronco Mesenquimais/metabolismo , Adipogenia/genética , Hemangioma/genética , Hemangioma/tratamento farmacológico , Hemangioma/metabolismo
8.
J Cardiothorac Surg ; 19(1): 78, 2024 Feb 09.
Artigo em Inglês | MEDLINE | ID: mdl-38336790

RESUMO

BACKGROUND: Pulmonary arteriovenous fistula (PAVF) is a rare disease, which can lead to the direct return of unoxidized venous blood to pulmonary veins and left heart, resulting in right-to-left shunt leading to hypoxia. Long term, the right-to-left shunt will cause severe pathophysiological changes in the patient's body and pulmonary circulation, and the prognosis will be poor if PAVF is not treated timely. CASE PRESENTATION: Here, we report the case of a 71-year-old man who presented with chest tightness and shortness of breath. After a series of examinations, PAVF and giant hemangioma were diagnosed, which are difficult to operate.Transcatheter interventional therapy was initiated. The patient recovered on the third day after operation and was discharged smoothly. During the long-term follow-up of nearly 4 years after discharge, the general condition and quality of life of the patient basically returned to normal. CONCLUSIONS: PAVF is rare but very important clinical problem. When the clinical manifestations of persistent unexplained hypoxia appear, it is necessary to fully consider the possibility of PAVF. Once the diagnosis of PAVF is clear, timely treatment is recommended to avoid deterioration of the disease and affecting the prognosis.


Assuntos
Fístula Arteriovenosa , Hemangioma , Artéria Pulmonar/anormalidades , Veias Pulmonares , Veias Pulmonares/anormalidades , Masculino , Humanos , Idoso , Veias Pulmonares/cirurgia , Qualidade de Vida , Artéria Pulmonar/diagnóstico por imagem , Artéria Pulmonar/cirurgia , Hipóxia/etiologia , Hemangioma/complicações , Hemangioma/diagnóstico , Hemangioma/cirurgia
9.
BMJ Case Rep ; 17(2)2024 Feb 21.
Artigo em Inglês | MEDLINE | ID: mdl-38383129

RESUMO

Intramuscular haemangiomas are benign tumours showing an extremely rare occurrence and making up less than 1% of all haemangiomas. The goal of this case report is to share our experience in treating a patient with an extremely rare intramuscular haemangioma, which occurred in the supraclavicular region VB level of the neck and extended towards the infraclavicular space, localised within and inferior to the trapezius muscle. The lesion was diagnosed by using multimodal imaging and a wide excision was performed with intraoperative feeding vessel ligation and a good postoperative result. The main difficulties regarding diagnosis and treatment include a lack of evidence-based guidelines, due to limited publications on this topic, the nature of the lesion frequently showing obscure anatomic localisation deep within muscles and unclear delineation, various anatomic origin requiring an individual treatment approach, inconclusive radiologic signs as well as non-specific symptoms.


Assuntos
Hemangioma , Humanos , Hemangioma/diagnóstico por imagem , Hemangioma/cirurgia , Músculos , Pescoço/patologia
10.
Zhonghua Yi Xue Za Zhi ; 104(8): 608-613, 2024 Feb 27.
Artigo em Chinês | MEDLINE | ID: mdl-38389238

RESUMO

Objective: The ultrasonography features of alveolar soft part sarcoma (ASPS) and intramuscular capillary-type hemangiomas (ICTH) were analyzed, and the diagnostic model of ASPS was established. Methods: A cross-sectional study was carried out. The clinical data of 52 patients [28 males and 24 females, aged (20.7±15.1) years] with pathologically confirmed ASPS and ICTH admitted to People's Hospital of Henan Province from January 2005 to February 2023 were included in the study. According to pathological types, the patients were divided into ASPS group and ICTH group. Clinical data of patients were retrospectively collected, and meaningful indicators in the univariate analysis were included in the regression analysis for screening. After comprehensive consideration of clinical significance and statistical significance, eligible indicators were selected for inclusion in the regression analysis. Binary logistic regression analysis was used to screen the factors that distinguished the pathological types of ASPS and ICTH, and the diagnostic model was established. The area under receiver operating characteristic (ROC) curve (AUC) was used to evaluate the diagnostic effectiveness of the diagnostic model in distinguishing ASPS from ICTH. Results: There were 20 patients in ASPS group, 10 males and 10 females, aged (26.9±13.5) years, and 32 patients in ICTH group, 18 males and 14 females, aged (16.8±15.0) years. The age difference between the ASPS group and the ICTH group was statistically significant (P<0.05), and there were statistically significant differences in the ultrasound imaging features of "clear boundary" "peripheral lobe" "thin blood vessels inside the lesion are straight and out of shape" "intra-lesion liquification" "peripheral thick blood vessels" and "peripheral muscle fiber disruption" between the two groups (all P<0.001).Variables with clinical and statistical significance were selected as independent variables. Binary logistic regression analysis showed that peripheral muscle fiber interruption (OR=97.358, 95%CI:6.833-1 387.249) and internal thin blood vessels were flat and out of shape (OR=0.052, 95%CI:0.003-0.921) was the correlation factor to distinguish the pathological types of ASPS and ICTH. Two ultrasonic image features of "peripheral muscle fiber interruption" and "internal thin blood vessels are straight and out of shape" were used to establish the diagnostic model. The sensitivity of "peripheral muscle fiber interruption" diagnostic model was 81.3%, and the specificity was 95.0%. The AUC was 0.811(95%CI: 0.761-0.954). The sensitivity, specificity and AUC of the diagnosis model of "internal thin vessels with flat misshape" were 90.0%, 96.9% and 0.934(95%CI: 0.830-0.984). The sensitivity, specificity and AUC of the combined diagnosis model of "peripheral muscle fiber interruption" and "internal thin blood vessel straight out of shape" were 96.9%, 90.0% and 0.974(95%CI:0.877-0.999). Conclusion: Ultrasonography can be used to distinguish ASPS from ICTH, and the combined diagnostic model based on the two ultrasonic imaging features of "peripheral muscle fiber interruption" and "internal thin blood vessel straight out of shape" can further improve the diagnostic efficiency.


Assuntos
Hemangioma , Sarcoma Alveolar de Partes Moles , Masculino , Feminino , Humanos , Sarcoma Alveolar de Partes Moles/diagnóstico por imagem , Sarcoma Alveolar de Partes Moles/patologia , Estudos Retrospectivos , Estudos Transversais , Ultrassonografia
11.
Medicine (Baltimore) ; 103(8): e37261, 2024 Feb 23.
Artigo em Inglês | MEDLINE | ID: mdl-38394533

RESUMO

RATIONALE: Intercostal hemangioma (IH) is an extremely rare disease, with only 18 cases reported in the past 30 years. Herein, we report the first case of IH coexisting with multiple hepatic hemangiomas, which recurred 32 months after surgery with rib erosion. IHs are invasive and difficult to distinguish from other intercostal tumors on imaging. To date, there have been no review articles on the imaging findings of IHs. We hope that this article will help clinicians improve their ability to diagnose and treat IH. PATIENTS CONCERNS: A 58-year-old male came to our hospital with gastrointestinal disease. Chest tumors were accidentally discovered on routine chest computed tomography (CT). The patient had no chest symptoms. The patient also had multiple liver tumors that had been present for 2 years but with no remarkable changes. DIAGNOSIS: Plain chest CT revealed 2 adjacent masses protruding from the left chest wall into the thoracic cavity. Neurogenic tumors or hamartomas were suspected on enhanced CT scans. Abdominal contrast-enhanced computed tomography scan indicated multiple liver tumors as MMHs, which was consistent with the 2 previous Doppler ultrasound findings. INTERVENTIONS: Surgeons removed the chest tumors by video-assisted thoracoscopic surgery. No treatment was provided for the MMHs. OUTCOMES: Two tumors of the chest wall were diagnosed as the IHs. There were no significant changes in the hepatic tumors after 32 months of follow-up. Unfortunately, the IH recurred, and the left 5th rib was slightly eroded. LESSONS: It is necessary to include IHs as a potential differential diagnosis for chest wall tumors because early clinical intervention can prevent tumor growth and damage to adjacent structures. The imaging findings of IH show special characteristics. Preoperative imaging evaluation and diagnosis of IH are helpful for safe and effective surgery. Because of the high recurrence rate, complete surgical resection of IH with a sufficient tumor-free margin is recommended. It should be noted that the ribs should also be removed when the surrounding ribs are suspected to have been violated.


Assuntos
Hemangioma , Neoplasias Hepáticas , Parede Torácica , Masculino , Humanos , Pessoa de Meia-Idade , Hemangioma/complicações , Hemangioma/diagnóstico por imagem , Hemangioma/cirurgia , Parede Torácica/patologia , Costelas/diagnóstico por imagem , Costelas/cirurgia , Costelas/patologia , Tomografia Computadorizada por Raios X , Neoplasias Hepáticas/complicações , Neoplasias Hepáticas/diagnóstico por imagem , Neoplasias Hepáticas/cirurgia
12.
BMC Pediatr ; 24(1): 138, 2024 Feb 23.
Artigo em Inglês | MEDLINE | ID: mdl-38395819

RESUMO

BACKGROUND: Hemangiomas, also called infantile hemangiomas (IH) or hemangiomas of infancy are the most frequently seen benign vascular tumors of infancy. Different types of hemangiomas are described in the literature. The current approach is to assess the risk and, if needed, first line treatment is to initiate systemic propranolol. CASE PRESENTATION: A 3-month-old Caucasian female patient was brought as an outpatient. The main complaint was an infantile hemangioma in the facial area, which as per the parents' story appeared within a week of birth like a small reddish line and it rapidly grew. Systemic propranolol was proposed as a first-line treatment and the adverse effects were explained. The parents, afraid of the side effects, wanted to explore other possibilities such as topical timolol, however, since it had no effect, propranolol was initiated in the end. Hemangioma was completely reduced in size; however, a skin defect was detected. As per the dermatologist's counsel, topical cream was initiated. The skin defect was reduced but not fully healed. The child is still being monitored periodically. CONCLUSION: After successful treatment of hemangioma, we identified a skin defect, which was very similar to steroid-induced skin atrophy. However, we cannot attribute this to a single factor. The only thing that can be concluded is that the subject needs a thorough studying, since rate of infantile hemangioma is high, and pediatricians need a clear management strategy of how to approach skin atrophy after successfully treating the hemangioma itself.


Assuntos
Hemangioma Capilar , Hemangioma , Neoplasias Cutâneas , Criança , Humanos , Feminino , Lactente , Propranolol/uso terapêutico , Antagonistas Adrenérgicos beta/uso terapêutico , Tratamento Conservador , Neoplasias Cutâneas/tratamento farmacológico , Neoplasias Cutâneas/complicações , Resultado do Tratamento , Hemangioma Capilar/complicações , Hemangioma Capilar/tratamento farmacológico , Hemangioma/complicações , Hemangioma/tratamento farmacológico
15.
Lasers Med Sci ; 39(1): 37, 2024 Jan 18.
Artigo em Inglês | MEDLINE | ID: mdl-38236327

RESUMO

Laser therapy has shown promising outcomes in treating infantile hemangiomas. However, the molecular mechanisms underlying laser treatment for IH remain incompletely elucidated. This study aimed to unravel the molecular mechanisms of laser therapy in IH treatment. We evaluated the inhibitory effects of laser treatment on the proliferation and promotion of apoptosis in human hemangioma endothelial cells (HemECs) through cell counting kit-8 (CCK-8) assay, Hoechst 33342 staining, and flow cytometric analysis. Transcriptome sequencing analysis of HemECs following laser treatment revealed a significant decrease in the expression level of the GSTM5 gene. The qRT-PCR and western blot analysis also showed that GSTM5 expression in HemECs was downregulated compared to human umbilical vein endothelial cells (HUVECs), and concomitantly, the p62-Nrf2 pathway was suppressed. Using siRNA to downregulate GSTM5 expression, we observed that inhibiting GSTM5 expression could restrain cell proliferation, elevate intracellular ROS levels, and induce apoptosis in HemECs. Furthermore, upon inhibition of the p62-Nrf2 pathway using p62-specific siRNA, a significant decrease in GSTM5 expression and an elevation in intracellular ROS levels were noted in laser-treated HemECs. These findings suggested that laser treatment may operate by inhibiting the p62-Nrf2 pathway, thereby downregulating GSTM5 expression, elevating ROS levels, and consequently inducing apoptosis in HemECs.


Assuntos
Hemangioma , Lasers de Estado Sólido , Humanos , Fator 2 Relacionado a NF-E2 , Espécies Reativas de Oxigênio , Transcriptoma , Hemangioma/genética , Hemangioma/radioterapia , Células Endoteliais da Veia Umbilical Humana , RNA Interferente Pequeno
16.
Int J Hyperthermia ; 41(1): 2285705, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-38269491

RESUMO

OBJECTIVE: The study aimed to compare the effectiveness and safety of ultrasound-guided microwave ablation (MWA) and percutaneous sclerotherapy (PS) for the treatment of large hepatic hemangioma (LHH). METHODS: This retrospective study included 96 patients who underwent MWA (n = 54) and PS (n = 42) as first-line treatment for LHH in three tertiary hospitals from January 2016 to December 2021. Primary outcomes were technique efficacy rate (volume reduction rate [VRR] > 50% at 12 months), symptom relief rate at 12 months and local tumor progression (LTP). Secondary outcomes included procedure time, major complications, treatment sessions, cost and one-, two-, three-year VRR. RESULTS: During a median follow-up of 36 months, the MWA group showed a higher technique efficacy rate (100% vs. 90.4%, p = .018) and symptom relief rate (100% vs. 80%, p = .123) than the PS group. The MWA group had fewer treatment sessions, higher one-, two- and three-year VRR, lower LTP rate (all p < .05), longer procedure time and higher treatment costs than the PS group (both p < .001). MWA shared a comparable major complications rate (1.8% vs. 2.4%, p = .432) with PS. After multivariate analysis, the lesion's heterogeneity and maximum diameter >8.1 cm were independent risk factors for LTP (all p < .05). In the PS group, lesions with a cumulative dose of bleomycin > 0.115 mg/cm3 had a lower risk of LTP (p = .006). CONCLUSIONS: Both MWA and PS treatments for large hepatic hemangioma are safe and effective, with MWA being superior in terms of efficacy.


Assuntos
Hemangioma , Neoplasias Hepáticas , Humanos , Escleroterapia , Micro-Ondas/uso terapêutico , Estudos Retrospectivos , Hemangioma/diagnóstico por imagem , Hemangioma/terapia , Neoplasias Hepáticas/terapia
17.
Cir. pediátr ; 37(1): 33-36, Ene. 2024. ilus
Artigo em Espanhol | IBECS | ID: ibc-228969

RESUMO

Introducción: La cutis marmorata telangiectásica congénita (CMTC) es una rara malformación capilar caracterizada por eritema reticular y violáceo persistente. Presentamos dos casos de CMTC. Observación clínica: Un varón de 13 meses presentaba una mácula violácea reticular en glúteo izquierdo y una pápula parduzca con signo de Darier en el maléolo interno del pie izquierdo, que fue biopsiada identificando > 15 mastocitos/campo, con lo cual se diagnosticó de CMTC y mastocitoma cutáneo solitario. El segundo caso, una recién nacida con una lesión característica de CMTC sin otras malformaciones al nacer, que durante el seguimiento desarrolló dos tumoraciones cutáneas compatibles con hemangiomas infantiles. Comentarios. La CMTC es una condición benigna, sin embargo, aproximadamente el 50% de los casos presentan anomalías asociadas. Ante la sospecha de CMTC se deben descartar malformaciones musculoesqueléticas, oftalmológicas y cutáneas. Hasta donde tenemos conocimiento, este es el primer reporte de CMTC asociada con mastocitoma y uno de los pocos con hemangioma infantil.(AU)


Introduction: Cutis marmorata telangiectatica congenita (CMTC) is a rare capillary malformation characterized by persistent reticular and violaceous erythema. We present two cases of CMTC. Clinical observation: The first case involved a 13-month-old male with a reticular violaceous macule on the left gluteal region and a brownish papule with Darier’s sign on the inner malleolus of the left foot, which was biopsied, revealing > 15 mast cells per field, leading to a diagnosis of CMTC and solitary cutaneous mastocytoma. The secondcase involved a newborn with a characteristic CMTC lesion without other malformations at birth, who subsequently developed two cutaneous tumors consistent with infantile hemangiomas during follow-up. Discussion. CMTC is a benign condition. However, approximately 50% of cases exhibit associated anomalies. When CMTC is suspected, musculoskeletal, ophthalmological, and cutaneous malformations should be ruled out. To the best of our knowledge, this is the first report of CMTC associated with mastocytoma and one of the few cases associated with infantile hemangioma.(AU)


Assuntos
Humanos , Masculino , Feminino , Recém-Nascido , Lactente , Livedo Reticular/diagnóstico por imagem , Nádegas/lesões , Pele/lesões , Eritema/diagnóstico por imagem , Hemangioma/diagnóstico , Mastocitoma , Pediatria , Pacientes Internados , Exame Físico , Avaliação de Sintomas
18.
BMC Pediatr ; 24(1): 8, 2024 01 03.
Artigo em Inglês | MEDLINE | ID: mdl-38172842

RESUMO

OBJECTIVES: To explore the clinical characteristics, postnatal treatment and prognosis of giant fetal hepatic hemangioma (GFHH). METHOD: Retrospective analysis was performed on children with giant fetal hepatic hemangioma (maximum tumor diameter > 40 mm) diagnosed by prenatal ultrasound and MRI from December 2016 to December 2020. These patients were observed and treated at the Children's Hospital of Fudan University after birth. The clinical data were collected to analyze the clinical characteristics, treatment, and prognosis of GFHH using independent sample t tests or Fisher's exact tests. RESULTS: Twenty-nine patients who were detected by routine ultrasound in the second and third trimester of pregnancy with giant fetal hepatic hemangiomas were included. The first prenatal ultrasound diagnosis of gestational age was 34.0 ± 4.3 weeks, ranging from 22 to 39 weeks. Of the patients, 28 had focal GFHHs and 1 had multifocal GFHHs. Surgery was performed, and the diagnosis was confirmed histopathologically in two patients. There were 8 cases with echocardiography-based evidence of pulmonary hypertension, 11 cases had a cardiothoracic ratio > 0.6, and 4 cases had hepatic arteriovenous fistula (AVF). The median follow-up time was 37 months (range: 14-70 months). During the follow-up, 12 patients received medical treatment with propranolol as the first-line therapy. The treatment group had a higher ratio of cardiothoracic ratio > 0.6 (P = 0.022) and lower albumin levels (P = 0.018). Four (14.8%) lesions showed postnatal growth before involuting. Complete response was observed in 13 (13/29) patients, and partial response was observed in 16 (16/29) patients. CONCLUSIONS: Fetal giant hepatic hemangioma is mainly localized, and its clinical outcome conforms to RICH (rapidly involuting) and PICH (partially involuting), but some fetal giant hepatic hemangiomas will continue to grow after birth and then gradually decrease. For uncomplicated giant fetal hepatic hemangioma, postnatal follow-up is the main concern, while those with complications require aggressive medical treatment. Propranolol may have no effect on the volume change of GFHH.


Assuntos
Hemangioma , Doenças do Recém-Nascido , Neoplasias Hepáticas , Gravidez , Recém-Nascido , Criança , Feminino , Humanos , Lactente , Propranolol/uso terapêutico , Estudos Retrospectivos , Hemangioma/diagnóstico por imagem , Hemangioma/terapia , Neoplasias Hepáticas/diagnóstico por imagem , Neoplasias Hepáticas/terapia , Neoplasias Hepáticas/patologia
19.
BMC Vet Res ; 20(1): 31, 2024 Jan 24.
Artigo em Inglês | MEDLINE | ID: mdl-38267947

RESUMO

BACKGROUND: Hemangiomas are a relatively common type of tumor in humans and animals. Various subtypes of hemangiomas have been described in the literature. The classification methods for hemangiomas differ between human and veterinary medicine, and the basis for tumor classification can be found in the literature. CASE PRESENTATION: This study describes a tumor in the subcutaneous tissue of the right dorsum of an artificially rescued juvenile Chinese pangolin. Computed tomography (CT) examination yielded the preliminary diagnosis of a vascular malformation, and surgery was performed to resect the tumor. Histopathological examination showed that the tumor mainly was consisted of adipose tissue, capillaries, and spindle cells in the fibrous stroma. Immunohistochemistry showed the positive expression of CD31, CD34, α-SMA, GLUT1 and WT-1 in the tumor tissue, and the tumor was eventually diagnosed as an infantile haemangioma. CONCLUSION: The final diagnosis of infantile hemangioma was depended on the histopathological immunohistochemical and CT examination of the neoplastic tissue. This is the first report of infantile hemangioma in a critically endangered species Chinese pangolin.


Assuntos
Hemangioma , Pangolins , Animais , Humanos , Hemangioma/diagnóstico por imagem , Hemangioma/veterinária , Tecido Adiposo , Espécies em Perigo de Extinção
20.
Hepatol Commun ; 8(1)2024 Jan 01.
Artigo em Inglês | MEDLINE | ID: mdl-38206210

RESUMO

BACKGROUND: The missing requirement for resection for the majority of hepatic hemangiomas (HH) and tissue scarcity for rare diseases such as hepatic epithelioid hemangioendotheliomas (HEHE) complicate the characterization of the spatial immunovascular niche of these benign and malignant vascular neoplastic diseases. METHODS: Two tissue cohorts containing 98 HHs and 13 HEHEs were used to study entity-specific and disease stage-specific endothelial cell (EC) phenotype and immune cell abundance. Using semiquantitative assessment, annotation-based cell classifiers, digital cell detection on whole slides, and tissue microarrays, we quantified 23 immunologic and vascular niche-associated markers and correlated this with clinicopathologic data. RESULTS: Both HH and HEHE ECs were characterized by a CD31high, CD34high, FVIII-related antigenhigh expression phenotype with entity-specific expression differences of sinusoidal EC markers Stabilin1, Stabilin2, CD32, and Lymphatic Vessel Endothelial Hyaluronan Receptor 1 (LYVE-1). Cell detection identified an HH margin-prevailing immunologic response dominated by Myeloperoxidase+ (MPO+) macrophages, CD3+ and CD8+ T cell subsets, and B cells (CD20+, CD79A+). In HEHE, increased CD68+ and CD20+ cell demarcation of lesion margins was observed, while CD3+ and CD8+ T cells were equally detectable both marginally and intralesionally. Stage-specific pairwise correlation analysis of HH and HEHE revealed disease entity-specific immunologic infiltration patterns as seen by high CD117+ cell numbers in HH, while HEHE samples showed increased CD3+ T cell infiltration. CONCLUSIONS: ECs in HH and HEHE share a continuous EC expression phenotype, while the expression of sinusoidal EC markers is more highly retained in HEHE. These phenotypic differences are associated with a unique and disease-specific immunovascular landscape.


Assuntos
Hemangioendotelioma Epitelioide , Hemangioma , Neoplasias Hepáticas , Humanos , Células Endoteliais , Linfócitos T CD8-Positivos
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