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PubMed2001

Lymphoscintigraphy in the diagnosis of lymphatic leak after surgical repair of femoral artery injury.

PURPOSE: Technetium-99m-labeled sulfur colloid lymphoscintigraphy is useful to evaluate lower extremity lymphatic circulation in cases of possible lymphedema and to reveal abnormal lymphatic collections. Groin lymphatic fistulas and lymphoceles are known complications of peripheral vascular surgical procedures. The authors describe a patient with ascites that developed into right lower extremity swelling after surgical repair of a femoral artery injury. Even after surgical ligation of multiple lymphatic channels, the patient continued to have lymphorrhea. It was unclear whether this was attributable to a persistent lymphatic leak or an ascitic leak from a postsurgical defect resulting in an abnormal connection with the peritoneal cavity. METHODS: Lymphoscintigraphy of the lower extremities was performed using Tc-99m sulfur colloid. Images were obtained at several intervals after injection of the radiotracer. Images were also acquired after the wound packing was removed. RESULTS: The images revealed an accumulation of radiotracer in the right groin, confirming the lower extremity lymphatic origin of the collection. CONCLUSIONS: Lymphoscintigraphy is useful to evaluate the origin of serous collections in the groin, a region in which lymphatic complications of vascular surgery are not uncommon.

Femoral Artery↗
PubMedDate not supplied

Abdominal manifestations of sarcoidosis in CT studies.

OBJECTIVE: The purpose of this study is to demonstrate the spectrum of abdominal CT findings in patients with tissue proved sarcoidosis, estimate their prevalence, and correlate abdominal findings with stage of thoracic disease. MATERIALS AND METHODS: Between 1987 and 1993, 140 patients with tissue proved sarcoidosis underwent abdominal or chest CT at our institution. After exclusions, 49 patients remained. Abdominal CT was retrospectively reviewed and the findings arrived at by consensus. Chest radiography within 1 month of CT was available for 46 patients. Chest radiography was retrospectively reviewed and stage of thoracic disease arrived at by consensus. RESULTS: Splenic abnormalities consisted of splenomegaly, low density lesions, and punctate calcifications. Hepatic sarcoidosis manifested as hypovascular lesions, hepatomegaly, and punctate calcifications. Lymphatic abnormalities consisted of lymphadenopathy, most commonly between 1 and 2 cm, and an increased number of normal sized lymph nodes. CONCLUSION: Sarcoidosis produces splenic abnormalities in 53% of patients, liver abnormalities in 16%, and lymphatic findings in 43%. No correlation was demonstrated between the abdominal findings and stage of thoracic sarcoidosis.

Female↗
PubMed1992

Lymphatic imaging in experimental filariasis using magnetic resonance.

RATIONALE AND OBJECTIVES: To evaluate acquired lymphatic abnormalities caused by filariasis, the authors examined the peripheral lymphatic system in normal ferrets and those chronically infected with Brugia malayi using magnetic resonance imaging (MRI). The findings were compared with previously obtained lymphangioscintigraphic (LAS) images in ferrets both with and without experimental filariasis. METHODS: Fifteen ferrets (11 infected with B. malayi and four noninfected controls) underwent whole body coronal MRI using a quadrature transmission-receive head coil at 0.5 Tesla operating at a resonant frequency of 21.5 mHz for protons with a 25-cm field of view. RESULTS: In contrast to normal animals, infected ferrets showed dilated hindlimb dermal lymphatic collaterals, enlarged high-signal intensity groin lymph nodes with punctate low-signal intensity centers and separate low-signal intensity spots with irregular thin channels, suggestive of nests of viable adult nematodes within tortuous lymphatics and nodes. MRI correlated with the LAS findings, and the interpretations were supported by light, scanning electron, and video microscopy. CONCLUSIONS: T2-weighted MRI in conjunction with LAS accurately depicts the peripheral lymphatic system in filarial-infected ferrets. These two modalities are useful complementary techniques to examine disorders characterized by lymphatic insufficiency.

Animals↗
PubMed2006

Temporal relationship between increased nuchal translucency and enlarged jugular lymphatic sac.

OBJECTIVE: To study the relationship between the volume of the jugular lymphatic sacs and nuchal translucency thickness in fetuses with increased nuchal translucency with advancing gestation. METHODS: Seventy-four fetuses with a nuchal translucency greater than the 95th percentile were examined weekly between 11 and 17 weeks of gestational age. The fetal neck region was studied by ultrasonography, followed by measurement of nuchal translucency and jugular lymphatic sacs. The measurements were analyzed using multilevel analysis. In case of termination of pregnancy postmortem examination was performed. RESULTS: In 40 euploid fetuses and 34 aneuploid fetuses, 159 measurements of jugular lymphatic sac volume and nuchal translucency thickness were analyzed. The volume of the jugular lymphatic sacs and gestational age showed a quadratic relation, which differed between euploid and aneuploid fetuses (P < .01). The maximum volumes were larger and present longer in fetuses with aneuploidy than in euploid fetuses (P < .01). In case of a cardiac anomaly, jugular lymphatic sac volume was larger than in cardiac normal fetuses (nonsignificant). Furthermore, the development of jugular lymphatic sac volume and increased nuchal translucency were related, whereby an increase of the nuchal translucency preceded enlargement of the jugular lymphatic sacs (P < .001). In each fetus an increase in jugular lymphatic sac volume was followed by a decrease with advancing gestation. The gestational age at maximum jugular lymphatic sac volume differed between fetuses, indicating a fetus-specific pattern. Nuchal translucency development showed a similar pattern. Postmortem examination confirmed distension of the jugular lymphatic sacs in all cases. CONCLUSION: Increased nuchal translucency is associated with abnormal lymphatic development, in which nuchal translucency enlargement precedes enlargement of jugular lymphatic sacs on ultrasound examination. Aneuploid fetuses have a more disturbed lymph angiogenesis. LEVEL OF EVIDENCE: III.

Abortion, Eugenic↗
PubMed2005

Clinical and molecular insights into lymphangioleiomyomatosis.

Lymphangioleiomyomatosis (LAM) is a rare disease of women that is characterized by a proliferation of abnormal smooth muscle-like cells (LAM cells), which leads to cystic lung lesions, lymphatic abnormalities, and abdominal tumors (e.g., angiomyolipomas). LAM occurs sporadically or in association with tuberous sclerosis complex, an autosomal dominant syndrome characterized by hamartoma-like tumor growths. The tumor suppressor genes TSC1 and TSC2 have been implicated in the etiology of LAM, as mutations and loss of heterozygosity (LOH) in TSC2 have been detected in LAM cells. TSC1 encodes hamartin, with a postulated role in actin cytoskeleton reorganization. TSC2 encodes tuberin, a protein with roles in cell growth and proliferation, transcriptional activation, and endocytosis. LAM cells, as defined by TSC2 LOH, have been detected in blood and body fluids, and can metastasize. LAM presents insidiously with progressive breathlessness, or dramatically with recurrent pneumothorax, chylothorax, or sudden abdominal hemorrhage. CT scans show numerous thin-walled cysts throughout the lungs, abdominal angiomyolipomas, and lymphangioleiomyomas. Pulmonary function tests show reduced flow rates (FEV1) and diffusion capacity (DL(CO)). Twenty per cent of patients have positive bronchodilator responses. Exercise testing shows gas-exchange abnormalities, ventilatory limitation, and hypoxemia that may occur with near-normal lung function. Progression of disease is best assessed by measurements of DL(CO) and FEV1. In the proper clinical setting, LAM may be diagnosed by a thoraco-abdominal CT scan. Tissue biopsy with special stains (HMB-45) should be reserved for cases with atypical presentations. There is no effective treatment for LAM, but on-going therapeutic trials with rapamycin appear promising.

Biopsy, Needle↗
PubMed2005

Lymphatic malformation of the lingual base and oral floor.

BACKGROUND: Lymphatic malformation of the tongue and floor of the mouth is associated with chronic airway problems, recurrent infection, and functional issues related to speech, oral hygiene, and malocclusion. There are no accepted anatomic guidelines or treatment protocols. METHODS: This retrospective review focused on anatomic extent, treatment, complications, and airway management in 31 patients with lymphatic malformation of the lingual base and oral floor. RESULTS: Involved adjacent structures included the neck (77 percent), mandible (41 percent), face (42 percent), lips (10 percent), pharynx (45 percent), and larynx (26 percent). Fifty-eight percent of patients required tracheostomy during infancy; decannulation was possible in two-thirds of these patients. Management included resection alone (42 percent), resection and sclerotherapy (26 percent), resection and laser coagulation (16 percent), sclerotherapy and laser coagulation (16 percent), and resection and radiofrequency ablation (3 percent). Resection involved the neck (58 percent), floor of the mouth (52 percent), and tongue (42 percent); there were often multiple procedures. Aspiration was tried with little success in 10 percent of patients. Virtually all patients had residual abnormal lymphatic tissue. Complications and posttherapeutic problems included infection (81 percent), neural damage (27 percent), difficulty in speech (23 percent), feeding problems (10 percent), and seroma or hematoma (6 percent). Associated dental/orthognathic conditions, particularly prognathism and anterior open bite, were documented in one-third of patients. CONCLUSIONS: The initial step in the protocol is control of the neonatal airway. Staged cervical resection is undertaken in late infancy to early childhood; resection should also include abnormal tissue in the oral floor. Sclerotherapy is primarily for macrocystic disease or secondarily for recurrent cysts following partial extirpation. Vesicles of the mucous membranes and dorsal tongue are treated either by sclerotherapy, laser (carbon dioxide, yttrium-aluminum-garnet, or potassium-titanyl-phosphate), or radiofrequency ablation. Reduction for macroglossia is indicated for persistent protrusion or to allow correction of malocclusion. Embolization controls lingual bleeding. Orthognathic procedures are undertaken at the appropriate age, only after lingual size and position are acceptable.

Catheter Ablation↗
PubMedDate not supplied

Progressive vertebral collapse in diffuse angiomatosis.

A case of vanishing bone disease is presented, with radiologic, endoscopic, and peritoneoscopic evidence of angiomatous involvement of the bones, surrounding soft tissues, and gastrointestinal tract. The diagnosis was made by a combination of radiologic techniques and endoscopy. Areas of absent bone were shown to opacify after intralymphatic injections of contrast material, providing in vivo demonstrations that these areas of vanishing bone are occupied by abnormal lymphatics. The etiology remains obscure, but our review of the literature suggests that this condition may be one of a large spectrum of disorders due to a basic underlying endothelial dysplasia. The lymphatic system, vascular system, or both may be involved, and the condition may manifest itself as an abnormality of the bones, soft tissues, viscera, or a combination of these.

Adult↗
PubMed2003

Cerebro-oculo-facial-lymphatic syndrome.

We describe a boy whose prime features are severe-to-profound mental retardation, intractable complex seizures, lissencephaly, facial dysmorphism, and lymphatic abnormalities. To our knowledge, this is the fourth reported case of this syndrome. We propose the syndromic appellation of cerebro-oculo-facial-lymphatic syndrome, suggest cardinal diagnostic features, and discuss several possible overlapping syndromic diagnoses.

Abnormalities, Multiple↗
PubMed1990

Congenital abnormalities of the lymphatic system: a new clinical classification.

The numerous clinical presentations of congenital abnormalities of the lymphatic system in children and the confusing terminology used to describe their pathologic diagnoses impede the physician's understanding of the condition. The clinical classification based on the actual symptoms of the congenital problems we have presented here should help the physician identify the specific abnormality and a potential treatment. Future research should concentrate on the specific causes and the treatment of these congenital abnormalities.

Adolescent↗
PubMed2002

Value of isotope lymphography in the diagnosis of lymphoedema of the leg.

BACKGROUND: Isotope lymphography has largely replaced contrast lymphography in the diagnosis of lymphoedema. Its accuracy has only been assessed in small studies and it is not known if it can identify patients with a proximal lymphatic obstruction who may be suitable for lymphatic bypass surgery. METHODS: Three hundred and ninety-five patients suspected to have lymphoedema were investigated by isotope lymphography between 1985 and 1995. Contrast lymphography was also carried out in 29 of these patients because the isotope results were thought to be misleading, or because lymphatic bypass surgery was being considered. RESULTS: In the 29 patients who had both investigations isotope lymphography detected 20 of 24 abnormal lymphatic systems. Four legs with obstructed groin lymphatics were reported as normal. Two legs with normal contrast lymphograms were erroneously diagnosed as having lymphoedema in the isotope study. Detectable groin nodes on the scintigrams were indicative of either normal lymphatics or proximal lymphatic obstruction. An increase in isotope uptake over 30-60 min of less than 50 per cent, or a total absence of isotope within groin nodes, was a sensitive indicator that patients were unsuitable for lymphatic bypass surgery. CONCLUSION: Isotope lymphography is a moderately sensitive test for lymphoedema, which will mistakenly classify some normal legs as lymphoedematous. It will usually correctly identify patients who are suitable for lymphatic bypass surgery.

Contrast Media↗
PubMed2005

Lymphangiogenesis-mediated shedding of LAM cell clusters as a mechanism for dissemination in lymphangioleiomyomatosis.

Lymphangioleiomyomatosis (LAM) affects exclusively women of reproductive age, involves the lungs and axial lymphatic system, and is frequently complicated with renal angiomyolipomas. LAM lesions are generated by the proliferation of LAM cells with mutations of one of the tuberous sclerosis complex (TSC) genes. Recent studies indicate that LAM cells can migrate or metastasize to form new lesions in multiple organs, although they show a morphologically benign appearance. In the previous study, we reported LAM-associated lymphangiogenesis and implicated its role in the progression of LAM. In this study, we further focused on the lymphatic abnormalities in LAM: LAM-associated chylous fluid (5 pleural effusion and 2 ascites), surgically resected diaphragm (1 patient), and axial lymphatic system including the thoracic duct, lymph nodes at various regions, and diaphragmatic lymphatic system (5 autopsy cases). We demonstrated that LAM cell clusters enveloped by lymphatic endothelial cells (LCC) in all chylous fluid examined. We identified LAM lesion in the diaphragm (2 of 5 autopy cases and one surgical specimen), thoracic duct (5 of 5), and lymph nodes (retroperitoneal (5 of 5), mediastinal (4 of 5), left venous angle (5 of 5) with total positive rate of 68% to 88% at each region of the lymph node, but less frequent or none at remote lymph nodes located away from the axial lymph trunk (cervical [1 of 5] and axillary [0 of 5]). LCCs were identified in intra-LAM lesional lymphatic channels where LAM cells proliferate along lymphatic system. In in vitro culture system, LCC can fragment into each proliferating LAM cell. These findings suggest that LAM-associated lymphangiogenesis demarcates LAM lesion into bundle- or fascicle-like structure and eventually shed LCC into the lymphatic circulation and that LCCs play a central role in the dissemination of LAM lesion.

Adult↗
PubMed2002

The newborn examination: part I. Emergencies and common abnormalities involving the skin, head, neck, chest, and respiratory and cardiovascular systems.

The routine newborn assessment should include an examination for size, macrocephaly or microcephaly, changes in skin color, signs of birth trauma, malformations, evidence of respiratory distress, level of arousal, posture, tone, presence of spontaneous movements, and symmetry of movements. A newborn with one anatomic malformation should be evaluated for associated anomalies. Total and direct bilirubin levels should be measured in newborns with jaundice, and a complete blood count should be obtained in those with pallor or a ruddy complexion. Neurosurgical consultation is necessary in infants with craniosynostosis accompanied by restricted brain growth or hydrocephalus, cephaloceles, or exophytic scalp nodules. Neck masses can be identified by their location and include vascular malformations, abnormal lymphatic tissue, teratomas, and dermoid cysts. Most facial nerve palsies resolve spontaneously. Conjunctivitis is relatively common in newborns. Infants with chest abnormalities may need to be evaluated for Poland's syndrome or Turner's syndrome. Murmurs in the immediate newborn period are usually innocent and represent a transition from fetal to neonatal circulation. Because cyanosis is primarily secondary to respiratory or cardiac causes, affected newborns should be evaluated expeditiously, with the involvement of a cardiologist or neonatologist.

Birth Injuries↗
PubMed1984

Pretibial myxoedema.

This paper reviews the clinical, endocrine and lymphatic status of 6 patients with pretibial myxoedema. The lymphatics were studied to determine whether they were involved in the pathogenesis of the disease. The main lymph trunks were normal although there was occasionally some collateral flow and some of the lymph nodes had small irregular filling defects. These changes suggest compression of the lymphatics by the myxomatous material and the deposition of mucinous material in the lymph nodes. There was no evidence of a primary lymphatic abnormality.

Female↗
PubMedDate not supplied

Pathophysiology of increased nuchal translucency: a review of the literature.

First-trimester nuchal translucency (NT) measurement is a widely used technique to determine the risk of fetal aneuploidy. The aim of this review was to explore the different hypotheses on the aetiology of increased NT. Using a computerized database (PubMed), articles on the aetiology of first-trimester NT were retrieved. Furthermore, the cited references of the studied articles were used to find additional articles. Based on ultrasonographic and post-mortem morphological studies, the findings in increased NT fetuses are described and classified into three categories. First, an association between increased NT and cardiac abnormalities, combined with abnormal ductus venosus flow velocities, is found, leading to the theory that cardiac failure causes NT enlargement. Second, various types of abnormalities are found in the extracellular matrix of the nuchal skin of fetuses with increased NT. Third, abnormal lymphatic development is demonstrated in fetuses with increased NT. In conclusion, many hypotheses on NT enlargement are based on associations and speculations. This report provides an overview of the different theories and an attempt to fit the different associations and findings into a single hypothesis of abnormal embryological development.

Aneuploidy↗
PubMed2006

Videocapsule endoscopy is useful for the diagnosis of intestinal lymphangiectasia.

We study two authentic cases of protein-losing enteropathy, the diagnosis of which was facilitated using Given M2A videocapsule endoscopy. The first case corresponded to a primary intestinal lymphangiectasia confirmed by jejunum biopsies and the second one to a protein-losing enteropathy with lymphatic abnormalities secondary to a chronic constrictive pericarditis. In the first case, the mucosa of jejunum presented with a diffuse oedematous aspect, whitish villi, white curved lines probably related to submucosal dilated lymphatics and lacteal juice. In the second case, capsule endoscopy showed oedematous aspect of jejunum mucosa associated with white curved lines similar to those observed in the first case. Videocapsule endoscopy is useful in cases of protein-losing enteropathy to identify presence of intestinal lymphangiectasia and to specify their localisation after ruling out other disorders liable to induce protein-losing gastrointestinal syndrome.

Adult↗
PubMed1988

Lymphedema: evaluation of qualitative and quantitative lymphoscintigraphy in 238 patients.

Lymphoscintigraphy was performed in 128 patients with primary lymphedema, in 91 patients with secondary lymphedema, and in 19 healthy volunteers. A total of 457 extremities were examined. Technetium-99m-labeled human serum albumin was injected subcutaneously, and passive muscular exercise was standardized to reduce the variability of lymphatic function. The scintigrams were analyzed for visualization of lymph vessels and lymph nodes, dilatation of lymphatic vessels, existence of collateral vessels, and dermal backflow. With this qualitative interpretation alone, the diagnosis of lymphedema was established in 216 of 308 extremities (70.1%). Quantitative parameters derived from clearance data showed abnormal lymphatic function in all 308 extremities. Whereas qualitative lymphoscintigraphy allows the characterization of lymphatic morphology, quantitative lymphoscintigraphy is very accurate in detection of incipient lymphedema.

Adolescent↗
PubMed1997

Lymphatic dysplasia in a neonate with Noonan's syndrome.

We present the case of a patient with Noonan's syndrome and massive nonimmune hydrops fetalis in whom lymphangiography was performed in the neonatal period. This demonstrated generalised lymphatic dysplasia, and we suggest a mechanism by which the lymphatic abnormalities in Noonan's syndrome may arise.

Contrast Media↗
PubMed1995

Use of mesenteric lymphangiography in a calf with chylothorax and chyloperitoneum.

Lymphatic abnormalities resulting in chylous effusion into a body cavity are uncommon in domestic animals. In a 6-day-old calf admitted to our hospital because of failure to suckle and abdominal distention, however, mesenteric lymphangiography revealed an obstruction of lymphatic flow. Laparoscopic examination of the abdomen was unsuccessful. Fluid accumulation was resolved in this calf by drainage. In cattle with chylothorax and concurrent chyloperitoneum in which a traumatic lesion of the thoracic duct is possible, conservative management, with drainage and supportive treatment, should be attempted prior to considering surgical intervention.

Animals↗