CHROMOSOME ABNORMALITY IN THREE CASES OF LYMPHATIC LEUKAEMIA IN CATTLE.
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Animals↗
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Neonatal teratoma presenting as hygroma colli.
We describe a neonate with a large tumour involving cranial, cervical and upper mediastinal regions, which presented clinically as hygroma colli. Radiological and pathological investigations showed characteristics of a mature teratoma and prominent cystic components within the tumour. These findings suggest that during early fetal development primary lymphatic sacs were obstructed by a teratoma leading to hygromatous dilatations of lymphatic vessels or that the abnormal proliferation of lymphatic vessels (hygroma) was part of the teratoma, developing from mesoderm as one of the three germinal layers from which teratomas originate. A third possibility is that the cystic part of the tumour originated from plexus chorioideus tissue, containing CSF. The last possibility is most probable in this patient.
Diagnosis, Differential↗
Gorham's disease: an autopsy report.
We present the case report of a 35-year-old man with Gorham's disease (disappearing bone disease, massive osteolysis) with initial clinical findings of small bowel lymphangiomatosis and multicentric osteolysis. The patient, who otherwise was healthy, had a chylothorax develop and he died 9 months later of thoracic and pulmonary complications. An autopsy revealed absence of the proximal thoracic duct and significant lymphangiectatic abnormalities of the pleural, peritoneal, diaphragmatic, splenic, and small bowel tissue with lymphangiomatous masses in the thoracic and mediastinal regions. The findings suggest a subtype of Gorham's disease characterized by a dysplastic lymphatic system. Osteolysis was correlated anatomically with lymphangiectatic tissue, suggesting mediation of osteoclastic resorption via local lymphatic tissue factors.
Adult↗
Lymphedema: anatomy, physiology and pathogenesis.
The authors review the current understanding of lymphatic anatomy and physiology, and the pathophysiology of lymphedema. The skin lymphatic system consists of the initial lymphatics, which converge into lymphatic precollectors, collectors and lymphatic ducts; these in turn convey the lymph to the regional lymph nodes. Interstitial fluid and particles enter the initial lymphatics through interendothelial openings and by vesicular transport. Lymphatic uptake is enhanced by external compression. Lymphatic transport depends greatly on contraction of lymphangions, which generate the suction force that promotes absorption of interstitial fluid and expels lymph to collecting ducts. In lymphedema, various types of congenital and acquired abnormalities of lymphatic vessels and lymph nodes have been observed. These often lead to lymphatic hypertension, valvular insufficiency and lymphostasis. Accumulation of interstitial and lymphatic fluid within the skin and subcutaneous tissue stimulates fibroblasts, keratinocytes and adipocytes eventuating in the deposition of collagen and glycosaminoglycans within the skin and subcutaneous tissue together with skin hypertrophy and destruction of elastic fibers.
Collagen↗
Lymphoscintigraphic assessment of leg oedema following arterial reconstruction using a load produced by standing.
Lymphoscintigraphy using a test involving standing from a supine position and performed following an intradermal injection of 99Tcm-human serum albumin (HSA) was developed to evaluate the function of the lymphatic system in the lower extremities of patients who developed lymphoedema following arterial reconstructive surgery. In normal subjects, the load produced by standing tended to increase lymphatic function as indicated by the frequent appearance of a large spiking wave and a rapid stepwise increase in tracer activity and, less often, a phase of decreasing tracer activity. However, there was either no or less activation of lymph flow following standing in the group which developed leg oedema postoperatively. These findings indicate that lymphatic disruption is responsible for the leg oedema seen in these patients. Performance of this test following an intradermal injection of 99Tcm-HSA is technically simple, requires no special apparatus, and can be completed in 30 min. Lymphoscintigraphy using our new method can provide useful information on abnormalities in lymphatic function.
Aged↗
Perinatal outcomes with the antepartum diagnosis of cystic hygroma.
OBJECTIVE: To describe antenatal diagnosis and clinical outcomes of fetal cystic hygroma. STUDY DESIGN: Retrospective case series derived from ultrasound database. Diagnoses were made from July 2000 through January 2003. RESULTS: Twenty-five fetuses were identified with cystic hygroma. Median gestational age at diagnosis was 17.3 weeks. Posterior nuchal cystic hygromas accounted for 21 (84%) cases, while two (8%) were axillary, and one each in the anterior neck and buttock. Additional abnormalities were noted in twenty (80%) fetuses; most common were non-immune hydrops (12/25, 48%) and cardiac defects (5/25, 20%). Eighteen patients (72%) underwent amniocentesis and ten were aneuploid. Six fetuses (25%) sustained intrauterine fetal demise and nine (38%) patients sought therapeutic termination. Nine infants (38%) were liveborn, but only six (25%) survived the neonatal period. CONCLUSIONS: Antenatal diagnosis of fetal cystic hygroma heralds an extremely poor perinatal prognosis. Associated abnormalities were noted in 80% of fetuses with a significant incidence of aneuploidy. Targeted ultrasonography and extensive counseling of parents is recommended.
Chromosome Aberrations↗
In vitro high-resolution helical CT of small axillary lymph nodes in patients with breast cancer: correlation of CT and histology.
OBJECTIVE: This study seeks to determine whether high-resolution in vitro helical CT can show the internal structure of small axillary nodes and to establish the CT characteristics of benign versus metastatic axillary nodes in patients with breast cancer. SUBJECTS AND METHODS: We obtained in vitro helical CT images of 212 nodes excised from 19 patients with breast cancer. The longest mean size was 5.9 mm, and the range was 0.5 to 26.5 mm. The hilar and cortical characteristics, the size, and the ratio of the longest axis to the shortest axis were evaluated. CT findings were correlated with histologic findings. RESULTS: Pathologic assessment of excised nodes with a central low-density hilum visualized on CT showed arteries, veins, lymphatic sinuses, and fatty tissue. A peripheral high-density cortex on CT contained mostly lymphatic tissue. Abnormal (eccentric, irregular) cortices were observed in malignant nodes (p<0.0001). Marked differences were observed among the proportions of benign and malignant nodes when the ratio of the longest axis to the shortest axis was less than 2 and an abnormal cortex was observed. CT could also detect extracapsular lymph node extension. CONCLUSION: In vitro high-resolution helical CT can detect the internal structure of small nodes. Morphologic changes detected on helical CT help distinguish benign from malignant nodes.
Adult↗
Effect of Brugia malayi on the growth and proliferation of endothelial cells in vitro.
Athymic mice (C3H/HeN) parasitized by Brugia malayi develop massively dilated lymphatics. The lymphatic endothelial lining is perturbed, and numerous mononuclear and giant cells are closely apposed to the endothelium. The hyperplastic endothelial cells and low opening pressure of the lymphatics suggest abnormal multiplication of these cells may be important in the dilation. We studied the in vitro growth rate of human umbilical vein endothelial cells cultured with adult worms and microfilariae of B. malayi. The tetrazolium salt reduction assays were used to quantify possible direct mitogenic or inhibitory effects. The growth factor-induced proliferation of endothelial cells was significantly suppressed by 44-51% on day 1, 46-81% on day 3, and 45-79% on day 5 in cultures containing adult female worms, which had greater suppressor activity on endothelial cell proliferation than male worms, microfilariae, or soluble adult worm extract. Culture supernatant containing female worm excretory-secretory products significantly inhibited the growth and multiplication of cells, suggesting that adult female worms release antigens or proteins that have inhibitory activity on growth factors necessary for endothelial cell proliferation in vitro. Excess human recombinant epidermal growth factor and bovine brain extract partly reversed the inhibitory activity of worms in culture and restored the endothelial cell proliferation when incubated with worm culture supernatant. Indomethacin and BW 775Hcl failed to restore normal endothelial proliferation in the presence of female worms, suggesting that parasite-derived prostanoids and cyclooxygenase products did not cause the inhibition. Lymph from dilated lymphatics, but not serum from infected mice, increased the proliferation of cells in vitro. Together, these data demonstrate that excretory-secretory products of B. malayi parasites suppress vascular endothelial proliferation in vitro. Furthermore, increases in the number of these cells in vitro in the presence of lymph suggest that parasite-induced host factors may be important in modulating the degree of proliferation.
Animals↗
Hemodynamic changes after resection of thoracic duct for en bloc resection of esophageal cancer.
An en bloc resection of esophageal cancer is one of the most radical forms of esophagectomy, and includes the resection of the thoracic duct, but a relatively high hospital mortality rate has been reported. There is very little knowledge on the pathophysiological changes after resection of the thoracic duct. We examined 24 patients who underwent en bloc resection. Some patients developed severe tachycardia or shock postoperatively which subsided after a massive infusion of plasma. Analysis of the fluid balance revealed that much more fluid was necessary during surgery and the postoperative 24 h than in patients treated by a standard esophagectomy. Postoperative lymphangiography or CT revealed abnormal collateral lymphatics around the kidneys or in the pelvic cavity. This suggests the development of the lymphaticovenous shunts, which differed depending on the anatomy of each patient. One patient with chronic hepatitis developed uncontrollable ascites. These are important findings which can hopefully reduce the high rate of hospital death after this operation.
Aged↗
[Surgical treatment of lymphatic malformations].
Lymphatic malformations remain a therapeutic challenge. Many treatments by the past led to poor success. The wide variety of clinical presentations makes it difficult to outline specific management programs. Often, diagnosis or complication circumstances as infection, bleeding, airway obstruction, or handicap force acting. Thus, a specific follow-up joined to a multidisciplinary analysis are the key of well-planned surgery. Through our experience and a review of literature, we will describe principles of lymphatic malformations surgery and specific technics for specific locations.
Face↗
[Indications of lasers treatment for lymphatic malformations].
Cutaneous dermatologic lesions of LM, in type of superficial, translucent, localized or diffuse vesicles, are accessible to laser treatment. The useful lasers are essentially laser CO(2), and rarely pulsed dye laser and the Nd: YAG. Treatment by laser allows to obtain the ablation of vesicles by limiting aftereffects (scare) and so to dry up the transcutaneous lymphatic liquid. However laser has no action on the deep constituent of the LM and it's why medium and long term recurrences are inevitable. Finally, the indication of the laser must be retained only as a supplement to the surgery and has to be done by experimented persons.
Aluminum Silicates↗
[Percutaneous treatment of cystic lymphangiomas].
Management of cystic lymphangioma necessitate for optimal diagnosis and treatment the expertise of a trained multidisciplinary team including dermatologists, radiologists, plastic and vascular surgeons. An initial imaging work-up of these lesions by ultrasound Doppler examination and MR imaging are necessary before treatment planning. Depending of the size, the location, the risk for the adjacent organs, a therapeutic decision may be mandatory. Percutaneous sclerotherapy is a safe and efficient treatment. It is the treatment of choice that must be proposed in first intention.
Age Factors↗
Haemangiomas and vascular malformations of the maxillofacial region--a review.
Congenital vascular anomalies have been and remain poorly understood. Since 1982 haemangiomas and vascular malformations have been recognised as distinct entities that exhibit unique characteristics and demand appropriately tailored treatment plans. However, "haemangioma" still continues to be used as a clinical and pathological description of many different types of vascular anomalies, which complicates both the care of patients and the interpretation of reports in journals. In this review we summarise the current classification, terminology, and the pathological basis of these lesions and discuss their management. The various therapeutic options available and their outcome will be discussed in addition to the recent advances in the psychosocial aspects of care, interventional radiology, laser and pharmacological therapy.
Angiography, Digital Subtraction↗
Pleural effusions of extravascular origin.
Most pleural effusions are caused by hydrostatic and oncotic pressure imbalance, inflammation or infection, or abnormalities in lymphatic drainage. A select number of effusions are caused by fluid of extravascular origin. Some of these effusions result from complications of treatment, whereas others are a ramification of the underlying disease. The incidence, pathogenesis, clinical presentation, chest radiographic manifestations, pleural fluid analysis, diagnosis, and management are discussed.
Catheterization, Central Venous↗
Lymphatic malformations of the head and neck: introduction of a disease score for children, Cologne Disease Score (CDS).
OBJECTIVE: A clinical staging system for children with lymphatic malformations that would allow for a standardized comparison of disease and treatment outcomes. METHODS: We developed an examination sheet for the determination of a disease score ranging from 0 (worst) points to 10 (best) points, Cologne Disease Score (CDS). Disfigurement, dysphagia, dysphonia, dyspnea and an observer statement towards progression were contributing to CDS. Each parameter yielded two, one or zero points. Two points were given when no limitation was seen in the patient concerning the respective item. One point was given at mild limitation and zero points were given when considerable limitation in the respective item could be observed. We evaluated 26 patients with lymphatic malformations of the head and neck retrospectively by completing one examination sheet for each patient-visit. RESULTS: Four patients had an initial CDS value of three or less points (severe disease-group). After therapy the mean value of their score increased only slightly to 3.9+/-2.6 points. Six patients had an initial CDS value of four, five or six points (advanced disease-group). After treatment their mean score value increased dramatically to 9.0+/-1.5 points (P<0.001). Sixteen patients initially had seven points or higher (moderate disease-group), they had 9.8+/-0.4 points after treatment. CONCLUSIONS: The evaluation of the CDS was easy and expeditious. The score itself was a good predictor in view of the outcome. This new disease score for paediatric patients might be appropriate to evaluate therapeutic trials in paediatric patients.
Adolescent↗
Use of the deep inferior epigastric perforator flap in a 15-month-old boy after resection of lymphatic malformation.
Lymphatic malformations are benign tumors that arise from congenital defects of the lymphatic system. Despite the fact that all lymphatic malformations develop embryologically, their presence may not be detected until the tumor enlarges from hemorrhage or infection. Typical of these tumors is a slow-growing, asymptomatic phase with the potential for rapid and massive expansion and a tendency to infiltrate into surrounding tissues. These tumors are associated with a high rate of recurrence, especially with incomplete excisions. To minimize the chance of recurrence, a complete resection of a groin microcystic lymphatic malformation was performed on this 15-month-old boy, creating a significant defect in the upper thigh. Immediate coverage was felt to be the best option for reducing any associated morbidity. We used a skin-soft tissue flap supplied by perforators of the deep inferior epigastric artery to repair the wound. Using this technique, the musculofascial structures in the abdominal wall were completely spared, and the skin and fat were transferred on a vascular leash to the desired location. The donor site was closed by approximating the fascial edges without tension. The defect on the thigh was filled with tissue of similar thickness and characteristics, avoiding potential contour irregularities and color mismatch. The deep inferior epigastric perforator (DIEP) flap is a useful option for groin defects and can be applied safely in the pediatric population.
Groin↗
Lymphatic malformations of the tongue base.
PURPOSE: In light of the paucity of literature on lymphatic malformations of the tongue base, our aim was to present our experience and long-term outcomes of patients with this rare and challenging pathologic entity. METHODS: Medical records of 25 patients treated by the 3 senior authors (RGA, MTC, and RTC) between 1974 and 2003 were retrospectively reviewed, and comprehensive clinical data were collected and analyzed. RESULTS: Twenty-one patients (13 female and 12 male infants) were diagnosed either prenatally or at birth. Of these patients, 18 required early airway stabilization; 17 required tracheotomy. Four patients were diagnosed after 1 year of age and had no airway problems. Follow-up ranged from 2 days (owing to death) to 28 years, with a mean of 10 years. In 21 patients, pathology was extensive, involving contiguous anatomical areas such as the anterior tongue, larynx, pharynx, and floor of mouth. Multiple resections and debulking procedures were performed to restore function and improve cosmesis. Four patients died, all with laryngeal involvement. Of the 14 survivors who had tracheotomies, only 5 are decannulated. Normal oral feeding has been achieved in 14 patients and normal speech, in 8 patients. Cosmesis has improved with time. Orthodontic and dental problems are common, and 9 patients have significant macrognathia. CONCLUSIONS: Although most patients with lymphatic malformations of the tongue base achieve normal oral feeding, airway, speech, and cosmesis issues remain problematic throughout life. Laryngeal involvement signifies extensive disease and is the most significant risk factor for serious complications and death.
Airway Obstruction↗
Classification, diagnosis, and interventional radiologic management of vascular malformations.
Vascular anomalies are comprised of either hemangiomas or vascular malformations.Low-flow vascular malformations can be divided into capillary, venous, and lymphatic types and are usually present at birth, undergo pari passu growth, and produce symptoms related to mass effect or stasis. High-flow malformations are comprised pre-dominantly of arteriovenous malformations that follow a more aggressive clinical course of hyperemia, adjacent mass effect, steal phenomenon, tissue destruction, and ultimately high output failure. Ultrasound, CT, nuclear medicine, angiography, and particularly MRI have greatly enhanced diagnostic accuracy and provide detailed information for percutaneous and surgical treatment planning and an objective means of following therapeutic efficacy. Interventional radiologic percutaneous sclerotherapy for low-flow lesions and embolosclerotherapy for high-flow lesions with or without adjunctive surgical intervention have become the mainstay of therapy.
Antimetabolites, Antineoplastic↗