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3.
J Contemp Dent Pract ; 22(6): 713-720, 2021 Jun 01.
Artigo em Inglês | MEDLINE | ID: mdl-34393132

RESUMO

AIM: To report a treatment case of mandibular deviation caused by congenital cervical lymphangioma with traditional orthodontic techniques, following-up by 10-year retention. BACKGROUND: Lymphangiomas, developmental anomalies, can induce various disturbances of swallowing, mastication, speech, breathing, and skeletal deformities as well as psychological stress and anxiety for the patient and their family. Lymphangiomas are benign with virtually no possibility of turning into a malignant lesion, so clinical management aims to treat the patient functionally. CASE DESCRIPTION: A girl, aged 6 years and 4 months, complained about facial asymmetry and anterior crossbite caused by congenital cervical lymphangioma. Her facial profile was the straight type with an adequate lip position. Anterior and right-side posterior crossbites were observed. On the frontal cephalogram, the menton shifted 3.0 mm to the right. A functional appliance with an expander was placed to correct her dental midline deviation and posterior crossbite. After 2-year treatment, the anterior and right-side posterior crossbites were improved. Multibracket treatment began after the growth spurt. After 44-month active treatment, a functional occlusion, including a Class I molar relationship with a proper interincisal relationship, was achieved. A functional occlusion was maintained during a 10-year retention period, while a mandibular downward growth was observed through the retention period. CONCLUSION: Conventional orthodontic techniques enable functional and stable occlusion even in patients with mandibular deviation caused by congenital cervical lymphangioma, although only using early orthodontic management by itself may have some limitations. CLINICAL SIGNIFICANCE: The hybrid technique combining functional appliance and intermaxillary elastics proves to be an effective therapy for correcting occlusal cant and mandibular deviation caused by cervical lymphangioma.


Assuntos
Linfangioma , Má Oclusão , Cefalometria , Assimetria Facial , Feminino , Seguimentos , Humanos , Linfangioma/complicações , Linfangioma/terapia , Mandíbula
4.
J Med Case Rep ; 15(1): 398, 2021 Aug 10.
Artigo em Inglês | MEDLINE | ID: mdl-34372919

RESUMO

BACKGROUND: Generalized lymphatic anomaly previously known as diffuse systemic lymphangiomatosis is a rare multisystem congenital disease arising from the lymphatic system, and it is characterized by abnormal proliferation of the lymphatic channels in osseous and extraosseous tissues. It typically affects children or young adults. Although it is benign, it can be misdiagnosed as malignancy because of its diffuse and debilitating nature depending on the site of involvement. Due to its rarity, diagnosis is often delayed, leading to potential significant morbidity or mortality if vital organs are involved. Furthermore, its potential for multiorgan involvement with no curative treatment makes its management challenging. CASE PRESENTATION: We describe a case of a 35-year-old Caucasian female, who presented with epigastric pain and was subsequently extensively investigated at multiple tertiary centers by numerous specialists for query malignancy and metabolic bone disorder following incidental computed tomography imaging findings of multiple osteolytic lesions in the axial skeleton, and low-attenuating lesions in the axilla, spleen, and mediastinum. The diagnosis was confirmed with an axillary excisional biopsy. She was clinically stable with no end organ damage. She was monitored conservatively. CONCLUSIONS: The case illustrates the importance of increased awareness among clinicians for this rare congenital disease to enable earlier diagnosis and to avoid unnecessary invasive investigations. Furthermore, this case highlights the potential need for multiple biopsies of affected sites to confirm diagnosis. We also discuss the emergence of interferon therapy, chemotherapy, immunosuppression, and immunotherapy as medical management for this condition.


Assuntos
Linfangioma , Anormalidades Linfáticas , Adulto , Biópsia , Criança , Feminino , Humanos , Anormalidades Linfáticas/diagnóstico por imagem , Anormalidades Linfáticas/terapia , Baço , Tomografia Computadorizada por Raios X
5.
Clin J Gastroenterol ; 14(6): 1791-1797, 2021 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-34386941

RESUMO

Lymphangiomas are classified as lymphatic malformations, which are more common in children and rare in adults. It frequently occurs in the cervical and axillary regions and uncommonly in the retroperitoneum. A 39-year-old woman presented to our department for the investigation for a 55 mm asymptomatic mass in the right anterior adrenal cavity. Abdominal ultrasound showed a tumor containing cysts in the right anterior adrenal cavity. Contrast-enhanced computed tomography showed that the tumor was poorly contrasted and ill-defined. Magnetic resonance imaging suggested that the tumor contained a small amount of fat. The tumor tended to grow, and the possibility of malignant diseases such as liposarcoma could not be excluded. Therefore, surgical resection was performed. Since intraoperative findings showed that the tumor tightly invaded to the duodenum and pancreatic head, a pancreaticoduodenectomy was selected. The entire tumor was removed without exposing the tumor. Macroscopic findings indicated that the specimen was 55 mm in size, indistinctly demarcated, yellow-white in color, and polycystic. Histologically, lymphovascular proliferation was observed with infiltration of the pancreatic head and the duodenal muscle layer. The diagnosis of lymphangioma was finally made. There was no recurrence 2 years after surgery.


Assuntos
Linfangioma , Neoplasias Retroperitoneais , Adulto , Criança , Feminino , Humanos , Linfangioma/diagnóstico por imagem , Linfangioma/cirurgia , Recidiva Local de Neoplasia , Pâncreas , Pancreatectomia , Pancreaticoduodenectomia , Neoplasias Retroperitoneais/diagnóstico por imagem , Neoplasias Retroperitoneais/cirurgia
6.
BMC Gastroenterol ; 21(1): 273, 2021 Jul 03.
Artigo em Inglês | MEDLINE | ID: mdl-34217200

RESUMO

BACKGROUND: Haemolymphangioma arising from the small bowel and its mesentery is extremely rare in the clinical setting. To date, only 8 cases of small bowel haemolymphangioma have been reported, and there have been no previously reported cases of haemolymphangioma in the small bowel mesentery (PubMed). The formation of this tumour is mostly congenital, but the exact mechanism is still unclear. As a benign tumour, the presentation of the disease may vary from a simple well-defined cystic lesion to an aggressive ill-defined lesion mimicking malignancy. However, there are no typical symptoms, and preoperative diagnosis is difficult. CASE PRESENTATION: We present two cases of haemolymphangioma in the small bowel mesentery in a 54-year-old man and a 52-year-old woman. Both of them came to the hospital due to an abdominal mass. In the first case, a cystic teratoma in the left abdominal area was considered after abdominal plain computed tomography (CT) and magnetic resonance imaging (MRI) scans. After taking an enhanced CT scan, a lipoma was considered based on the images. In the second case, cystic masses of the left upper and middle abdomen were observed on abdominal ultrasonography. An abdominal plain CT scan showed an irregular low-density mass in the left upper and middle abdomen. With an enhanced CT scan, haemolymphangioma was considered based on the images. After complete surgical removal, the masses were found to originate from the small bowel mesentery and had not invaded into the peripheral lymphatic tissue. In case 1 in this study, the routine pathology diagnosis was lymphangioma, while in case 2, the diagnosis was haemangioma. The final diagnosis was confirmed to be haemolymphangioma by immunohistochemistry in both cases. No recurrence was evident during 4 months of follow-up. We review the previous case reports of haemolymphangioma in the abdominal cavity and discuss their clinical features, diagnosis, treatment and prognosis. CONCLUSIONS: The clinical manifestations of abdominal haemolymphangiomas can vary for both location and size. Abdominal CT examination has important clinical value for haemolymphangioma in the abdominal cavity. The final diagnosis of haemolymphangioma depends on a postoperative pathological examination. In addition, postoperative regular follow-up is necessary.


Assuntos
Hemangioma , Linfangioma , Feminino , Humanos , Masculino , Mesentério , Pessoa de Meia-Idade , Recidiva Local de Neoplasia , Tomografia Computadorizada por Raios X
9.
Chest ; 160(1): e63-e67, 2021 07.
Artigo em Inglês | MEDLINE | ID: mdl-34246391

RESUMO

CASE PRESENTATION: A 57-year-old woman was admitted to our hospital for an abnormal chest shadow found during routine chest radiography. She had no respiratory symptoms. Her medical history included dyslipidemia, and her surgical history included conization for cervical cancer at age 38 years. She was a social drinker and ex-smoker of approximately 10 cigarettes per day (from ages 20 to 30 years); she denied recreational drug use.


Assuntos
Neoplasias Pulmonares/diagnóstico , Pulmão/diagnóstico por imagem , Linfangioma/diagnóstico , Doenças Raras , Biópsia , Diagnóstico Diferencial , Endossonografia , Feminino , Humanos , Pulmão/cirurgia , Neoplasias Pulmonares/cirurgia , Linfangioma/cirurgia , Pessoa de Meia-Idade , Pneumonectomia , Tomografia por Emissão de Pósitrons combinada à Tomografia Computadorizada
10.
Vnitr Lek ; 67(E-4): 9-12, 2021.
Artigo em Inglês | MEDLINE | ID: mdl-34275313

RESUMO

Lymphangiomatosis is rare disease, we can find this entity in differential diagnosis of osteolytic leasions of bones of unknown origin. Typical sign for lymphangiomatosis is proliferation of lymphatic tissue with production of lymphangiomas in various organs and systems. Clinical manifestation of disease is variable, involvement of lungs and bone is typical. In our article we present recent classification of lymphatic tissue neoplasias, their clinical symptoms and treatment possibilities.


Assuntos
Linfangioma , Vasos Linfáticos , Osso e Ossos , Diagnóstico Diferencial , Humanos , Linfangioma/diagnóstico por imagem , Vasos Linfáticos/diagnóstico por imagem , Doenças Raras
11.
Nepal J Ophthalmol ; 13(25): 157-161, 2021 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-33981112

RESUMO

INTRODUCTION: Management of orbital lymphangioma is challenging. Complete surgical excision is often impossible due to its infiltrative nature. Sclerosing agents have been used in its management with variable outcomes. We report a case of recurrent orbital lymphangioma managed with intralesional bleomycin. CASE: A 14-year-old female presented with proptosis of the right eye for two weeks. She had a similar history at five years of age for which she underwent surgical excision. We performed negative pressure aspiration using a 20-gauge angiocatheter, injected bleomycin, and left the cannula in situ for repeat aspiration to maintain cyst collapse. OBSERVATION: The lymphangioma regressed, and there was no recurrence at six months of follow-up. CONCLUSION: This report highlights the use of negative pressure aspiration and intralesional bleomycin injection by minimal intervention using angiocatheter in the successful management of orbital lymphangioma.


Assuntos
Linfangioma , Neoplasias Orbitárias , Adolescente , Bleomicina/uso terapêutico , Feminino , Humanos , Injeções Intralesionais , Linfangioma/diagnóstico , Linfangioma/tratamento farmacológico , Recidiva Local de Neoplasia/diagnóstico , Recidiva Local de Neoplasia/tratamento farmacológico , Neoplasias Orbitárias/diagnóstico , Neoplasias Orbitárias/tratamento farmacológico
12.
Artigo em Chinês | MEDLINE | ID: mdl-34010999

RESUMO

Objective: To investigate the clinical features, diagnoses and treatments of head and neck occupying lesions in newborns. Methods: All newborns with head and neck occupying lesions admitted to Neonatel Intensive Care Unit of the First Affiliated Hospital of Zhengzhou University form January 2014 to November 2019 were included. There were 23 males and 17 females, admission age was from 2 d-28 d, and the clinical manifestations, examinations, treatments and outcomes were evaluated. Results: Among 40 newborns with head and neck occupying lesions, 22 cases were admitted with dyspnea, 15 cases with masses in oral cavity or head and neck, 2 cases with fever as the first symptom, and 1 case with hoarseness as the first symptom. There were 5 cases with local infection. All cases were examined with local ultrasound and CT or MRI. Nine cases with severe dyspnea were treated with invasive ventilationm, of them 6 cases underwent invasive ventilation for more than 48 hours, 4 cases received tracheal intubation and artificial nose. Diagnostic punctures were performed in 2 cases. Seven cases received conservative treatments. Surgeries were performed in 31 cases, and 25 cases obtained pathologic diagnoses, including 3 cases of soft palate mature teratomas, 1 case of hard palate teratoma, 1 case of granulosa cell tumor, 1 case of lobulated spindle cell tumor in tongue base, 1 case of polyp in right glottis, 1 case of polyp at esophageal entrance, 4 cases of lingual root cysts, 1 case of laryngeal cyst, 2 cases of thyroglossal duct cysts, 2 cases of lymphangiomas, 1 case of lymphangioma with hibernoma, 1 case of tracheal cyst, 1 case of esophageal cyst, 3 cases of left neck abscesses, 1 case of occipital hemangioma, and 1 case of left temporoparietal abscess. Conclusions: The head and neck occupying lesions in the newborn is prone to upper airway obstruction. Imaging examination can assist the diagnosis. Different treatments can be selected according to the natures of occupying lesions.


Assuntos
Cisto Dermoide , Linfangioma , Teratoma , Cisto Tireoglosso , Feminino , Humanos , Recém-Nascido , Masculino , Pescoço , Teratoma/diagnóstico , Teratoma/terapia
13.
BMC Gastroenterol ; 21(1): 151, 2021 Apr 06.
Artigo em Inglês | MEDLINE | ID: mdl-33823798

RESUMO

BACKGROUND: Ectopic pancreas (EP) is defined as pancreatic tissue that lacks anatomical or vascular communication with the normal body of the pancreas. Despite improvements in diagnostic endoscopy and imaging studies, differentiating ectopic pancreatic tissue from gastric submucosal diseases remains a challenge. CASE PRESENTATION: Here, we present a case of a 44-year-old woman with severe epigastric pain. Initially, gastric lymphangioma was highly suspected due to a well-demarcated protruding mass with a large size that occurred in the submucosal layer of the gastric antrum and appeared as a cystic lesion. The final correct diagnosis of gastric EP was made during surgery. CONCLUSION: Gastric EP with serous oligocystic adenoma appearing as a giant gastric cyst is extremely rare. The difficulty of making an accurate diagnosis and differential diagnosis is highlighted, which may provide additional clinical experience for the diagnosis of EP with serous oligocystic adenoma in the stomach.


Assuntos
Coristoma , Cistos , Linfangioma , Gastropatias , Adulto , Feminino , Humanos , Pâncreas/diagnóstico por imagem , Gastropatias/diagnóstico
16.
Dermatol Surg ; 47(7): 948-952, 2021 07 01.
Artigo em Inglês | MEDLINE | ID: mdl-33625132

RESUMO

BACKGROUND: Bleomycin sclerotherapy became a popular nonsurgical option for the management of lymphangiomas. However, its efficacy has not been thoroughly evaluated. The purpose of this study was to assess the clinical outcomes and the effectiveness of bleomycin injection for the treatment of lymphangioma. METHODS: This retrospective study was conducted in 4 centers and included 47 infants and children. All patients had bleomycin sclerotherapy between November 2005 and September 2020. Men presented 53.2% of the study sample (n = 25), and the most common site was the head and neck (n = 29, 61.7%). RESULTS: Two injections were required in 11 patients (23.4%), and 7 patients (14.9%) required 3 or more injections. Excellent response was achieved in 63.8% (n = 30), 14 patients (29.8%) had a good response, and 4 had a poor response (8.5%). There was no difference in the response according to the site of the lesion (p = .75). The most frequent complication was recurrence (n = 11, 23.4%), and swelling occurred in 5 patients (10.6%). No patient had facial or phrenic nerve palsy or hoarseness. Two patients had persistent pain, and 2 had an infection (4.3%). CONCLUSION: Intralesional bleomycin injection could be an effective therapy for lymphangiomas. The procedure has a low complication profile, and long-term study is recommended to evaluate the systemic and late bleomycin injection complications.


Assuntos
Antibióticos Antineoplásicos/administração & dosagem , Bleomicina/administração & dosagem , Linfangioma/terapia , Escleroterapia , Pré-Escolar , Feminino , Humanos , Lactente , Injeções Intralesionais , Masculino , Estudos Retrospectivos , Resultado do Tratamento
17.
J Int Med Res ; 49(2): 300060520986677, 2021 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-33528281

RESUMO

We describe herein a 37-year-old woman with a 2-week history of melena who was eventually diagnosed with ileal haemolymphangioma, a rare benign tumour. Local mucosal congestion and swelling were found through single-balloon enteroscopy, which showed an irregular protuberance approximately 10 cm long, located 3.2 m from the Treitz ligament. We performed a laparoscopic-assisted partial resection of the small intestine combined with intestinal adhesiolysis. According to postoperative pathology, the final diagnosis was ileal haemolymphangioma with haemorrhage.


Assuntos
Hemangioma , Laparoscopia , Linfangioma , Adulto , Feminino , Hemorragia Gastrointestinal/etiologia , Hemorragia Gastrointestinal/cirurgia , Hemangioma/complicações , Hemangioma/diagnóstico por imagem , Hemangioma/cirurgia , Humanos , Intestino Delgado , Linfangioma/diagnóstico por imagem , Linfangioma/cirurgia
18.
Medicine (Baltimore) ; 100(4): e23866, 2021 Jan 29.
Artigo em Inglês | MEDLINE | ID: mdl-33530180

RESUMO

INTRODUCTION: Metastasis of a papillary thyroid microcarcinoma (PTMC) in the lateral neck is characterized primarily by solid lymphadenopathy, although some cases may rarely present with a cervical cystic mass. We report a case of lateral cervical lymph node metastases of PTMC that appeared as a cystic lymphangioma of the lateral neck. PATIENT CONCERNS: A 55-year-old man with a painless egg-sized mass in the right side of the neck that had been present for 1 month underwent physical examination, ultrasonography, computed tomography (CT), fine needle aspiration biopsy (FNAB), and intraoperative fast-frozen pathological examination, which indicated that the cystic masses in the neck were benign. However, the final pathology report identified the lateral neck masses as lymph node metastases of thyroid carcinoma. DIAGNOSIS: The patient was diagnosed with PTMC of the right lobe of the thyroid gland with lateral neck metastases. INTERVENTIONS: The patient underwent right cervical neck dissection together with a right thyroidectomy, followed by levothyroxine therapy and routine follow-up. OUTCOMES: No postoperative complications were reported, and the thyroid-stimulating hormone inhibition target was <0.1 mmol/L; there was no detectable tumor recurrence on routine clinical follow-up for up to 16 months. CONCLUSIONS: This case report emphasizes the need to consider cervical lymph node metastases of thyroid carcinoma in the differential diagnosis for patients with large, multiple, simple cystic neck masses.


Assuntos
Carcinoma Papilar/patologia , Neoplasias de Cabeça e Pescoço/secundário , Linfangioma/patologia , Metástase Linfática , Neoplasias da Glândula Tireoide/patologia , Carcinoma Papilar/tratamento farmacológico , Carcinoma Papilar/cirurgia , Neoplasias de Cabeça e Pescoço/cirurgia , Terapia de Reposição Hormonal , Humanos , Linfangioma/cirurgia , Masculino , Pessoa de Meia-Idade , Esvaziamento Cervical , Neoplasias da Glândula Tireoide/tratamento farmacológico , Neoplasias da Glândula Tireoide/cirurgia , Tireoidectomia , Tiroxina/uso terapêutico
20.
BMJ Case Rep ; 14(1)2021 Jan 18.
Artigo em Inglês | MEDLINE | ID: mdl-33462010

RESUMO

We describe a case of a woman diagnosed at the age of 35 years with a generalised mediastinal and abdominal lymphangiomatosis associated with a protein losing enteropathy, who successfully improved when treatment with sirolimus was initiated.


Assuntos
Neoplasias Abdominais/diagnóstico , Linfangioma/diagnóstico , Neoplasias do Mediastino/diagnóstico , Adulto , Feminino , Humanos
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