Manual lymphatic drainage in management of edema in a case with CRPS: why the(y) wait?

Complex regional pain syndrome (CRPS) is a disorder characterized by pain, edema, skin color changes and autonomic abnormalities. Its treatment is quite difficult and in most of the patients effective results cannot be reached. Manual lymphatic drainage is a very rare method for managing limb edema in CRPS. In this case report, the dramatic response of an excessive edema to lymphatic drainage was discussed in a CRPS patient.     

Decompression of the thoracic duct: A novel transcatheter approach

In patients with total cavopulmonary connections, elevated central venous pressures (CVP) have detrimental effects on the lymphatic system causing an imbalance in fluid production and drainage of the interstitium. This combination may result in life‐threatening lymphatic complications including plastic bronchitis (PB), protein losing enteropathy (PLE), chylothorax, and ascites. While embolization of the abnormal lymphatics has greatly improved outcomes from these complications, alternative treatment strategies have been proposed that would result in improved lymphatic drainage while leaving the lymphatic system intact. We report two novel transcatheter approaches for thoracic duct (TD) decompression in two patients who developed PLE after completion of the Fontan procedure as part of staged palliation for congenital heart disease. In addition, one patient had severe concurrent PB. In both patients, a connection was created between a left superior vena cava (LSVC) to the left atrium allowing for a nonsurgical method to decompress the TD. This procedure resulted in significant clinical and laboratory improvement of both patients' PLE and other symptoms of lymphatic dysfunction.     

Osteomyelitis of a long bone due to Fusobacterium nucleatum and Actinomyces meyeri in an immunocompetent adult: A case report and literature review

ABSTRACT: BACKGROUND: Fusobacterium species are uncommon causes of osteomyelitis. These organisms are normal flora of the oral cavity. Therefore, they mostly cause osteomyelitis of the head and neck. Hematogenous osteomyelitis at distant sites other than the head and neck has rarely been reported in pediatric or immunocompromised patients. Here, we report the first case of osteomyelitis of a long bone combined with a muscle abscess due to Fusobacterium nucleatum in an otherwise healthy adult. CASE PRESENTATION: A 59-year-old Korean man was admitted for pain and swelling of the right lower leg, which had been persistent for two weeks. Magnetic resonance imaging showed osteomyelitis of the right fibula with a surrounding muscle abscess of the right lower leg. Incision and drainage was performed, and repetitive tissue cultures grew F. nucleatum. In this patient, it was presumed that recurrent periodontitis caused hematogenous seeding of F. nucleatum to a distant site leading to osteomyelitis with a muscle abscess. The patient was successfully treated with intravenous ampicillin-sulbactam for three weeks and oral amoxicillinclavulanate for eight weeks. He also underwent repeated surgical drainage. He has no evidence of recurrence after seven months of follow-up. CONCLUSIONS: Clinicians should be aware that F. nucleatum could be the etiologic agent of hematogenous osteomyelitis of a long bone in an immunocompetent patient.     

Tularemia during pregnancy

Tularemia is a zoonotic infection caused by Francisella tularensis with a worldwide distribution and diverse clinical manifestations. Although F. tularensis has been recognized as a human pathogen for a century, there are few reports regarding the occurrence of tularemia in pregnant women and its effect on the fetus; only seven cases have been reported in the literature. In view of the sparse literature, it is not clear whether tularemia increases the risk of adverse pregnancy outcomes. In this paper we review tularemia infection during pregnancy, its complications and management. In addition, we present a case of tularemia that occurred in the first trimester of pregnancy and resulted in third-trimester intrauterine fetal death, highlighting the consequences of tularemia in pregnancy and the importance of early detection and treatment.     

23-jähriger Patient mit Halsschmerzen und progredienter Lymphknotenschwellung

We report a case of oropharyngeal tularemia – an uncommon manifestation of this disease. There is a low prevalence of tularemia in Germany. Therefore the diagnosis can be confirmed only by well directed laboratory diagnostics. Without correct antibiotic therapy mortality can reach 33% – depending on the subspecies of Francisella tularensis. For this reason tularemia should be included into the differential-diagnostic considerations in patients with unclear lymph node enlargement.     

Fieber und Lymphadenopathie

We report on 4 patients with different types of tularemia acquired in Switzerland or nearby countries. All patients presented with fever, moderate to highly elevated signs of inflammation, and local lymphadenopathy. Additionally, 3 patients did not respond to empirical antimicrobial therapy with aminopenicillins. A tick bite was identified as mode of transmission in 2 patients, while 1 patient showed a possible connection to a tick bite. The route of transmission for the fourth patient remained unknown. The diagnosis of tularemia was either based on positive serology, on a positive polymerase chain reaction (PCR) from the lymph node samples or on positive blood cultures. The treatment in adult patients was ciprofloxacin 500–750 mg twice daily orally for 3 weeks. The pediatric patient was treated with gentamicin 4 mg/kg i.v. once daily for 1 week and ciprofloxacin 15 mg/kg twice daily orally for another 2 weeks. All patients recovered completely. Due to the increasing incidence of tularemia in Switzerland, this infection should be considered in patients with fever and lymph node enlargement particularly after tick bite. We recommend treatment with ciprofloxacin orally for 14–12 days.     

Cervical necrotizing fasciitis associated with descending necrotizing mediastinitis

ABSTRACT: We report a case of potentially fatal cervical necrotizing fasciitis and descending necrotizing mediastinitis due to deep neck infection in a 66-year-old male patient with no history or evidence of immunocompromising disorders. On admission, he had painful neck movements and the skin over his neck was red, hot and tender. A computerized tomography (CT) scan of his neck and chest showed evidence of air collection in soft tissues. He was treated with broad-spectrum intravenous antibiotics and early massive cervical drainage. Prompt diagnosis by CT of the neck and chest enabled an early surgical treatment of cervical necrotizing fasciitis. Although acute mediastinitis is a fatal infection involving the connective tissues that fill the interpleural spaces and surround the median thoracic organs, an extensive cervicotomy combined with appropriate antibiotics can prevent the need for mediastinal drainage.     

Adénopathies prétragiennes liées à Bartonella henselae

IntroductionCat scratch disease is characterized by adenitis with usually positive outcome. We reported two cases of cat scratch disease with preauricular involvement occurring in immunocompetent patients.ObservationsObservation 1: a 28-year-old man had a recent onset of left cervical swelling, with a peripheral facial paralysis and liver cytolysis. Serologies for EBV, viral hepatitis, CMV, HIV and toxoplasma were negative. Node excision biopsy suggested granulomatous lymphadenitis and Bartonella henselae PCR on lymph node was positive. With doxycyclin for 3 months, associated with rifampicin for 15 days, abnormal liver function disappeared and facial paralysis improved. Observation 2: a 17-year-old man had parotid swelling associated with right posterior cervical lymphadenopathies associated with fever and profuse sweating. A large right preauricular lymphadenopathy with necrotic remodeling was visible on the CT-scan. Lymph fluid B. henselae PCR was positive. Positive outcome occurs after surgical drainage and short azithromycin treatment.ConclusionPhysicians should be aware of the rare preauricular localization of cat scratch disease and ask for contact with a cat. Parotid tumor localization, lymphoma or tuberculosis should be ruled out. Diagnosis is based on the B. henselae PCR. Outcome is often spontaneously positive but surgical treatment may be required.     

Tularemia in Central Anatolia

Background

Tularemia is a bacterial zoonosis with diverse clinical manifestations depending on bacterial subspecies and the route of the infection.

Methods

We collected data prospectively of cases diagnosed and treated for tularemia in our institution during the epidemics from December 2009 to August 2011. Specific antibodies were screened by a microagglutination test. Throat swab and lymph node aspirate cultures were obtained and polymerase chain reaction (PCR) was performed on these specimens. Lymph nodes were characterized on the basis of ultrasound reports.

Results

A total of 139 patients were confirmed with tularemia. The age range of the patients was 6–83 years (mean: 43) and 84 (60.4 %) of them were females. Patients had clinical presentations compatible with oropharyngeal (74 %), glandular (15.8 %), and oculoglandular (5.0 %) tularemia. Ultrasonography (US) was performed in 108 patients. Antibiotics (aminoglycosides, quinolones, and doxycycline) were used in 138 patients. Fine-needle aspiration (FNA) or surgical drainage of fluctuant lymph nodes were performed in 51 (39 %) patients. Therapeutic failure was observed in 43 (30.9 %) patients. Elevation of erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) levels were observed to be significantly higher in patients with therapeutic failures (p = 0.003 and 0.004, respectively). The success rate was significantly higher in patients with early treatment (p = 0.004). No difference was found between the effectiveness of aminoglycoside or quinolone treatments. The increase in the short and long axes, and the characteristics of lymph nodes detected on US were significantly associated with treatment failures (p < 0.001). Intranodal necrosis was found in 45 patients. The treatment success rate was 40 % in patients with intranodal necrosis.

Conclusion

To the best of our knowledge, this is the first study defining the US findings of patients with tularemia and its association with treatment success. Ciprofloxacin is an effective and convenient choice in epidemics of tularemia and early treatment is still the cornerstone of successful therapies.     

Isolated spread of hepatic metastatic disease to a mediastinal lymph node

The lymphatic system is an important route of spread of hepatic metastatic disease to extrahepatic sites. Although portal and celiac nodes are commonly evaluated both pre- and intraoperatively in patients considered for resection, cephalad sites of drainage of the liver represent a more occult pitfall. We report a case of colon cancer metastatic to the right lobe with an isolated extrahepatic deposit in a mediastinal lymph node. This preoperative diagnosis was confirmed at a subsequent operation, leading to a change in treatment plan. We believe that such occurrences may be unrecognized rather than rare. Careful evaluation of the mediastinum prior to proceeding with hepatic resection may improve patient selection, and hence the outcome, of this procedure.     

Chylothorax following myocardial revascularization with the internal mammary artery

Chylothorax is a rare but serious complication of coronary artery bypass grafting. We describe a case of double myocardial revascularization with the internal mammary artery developing the complication ten weeks after cardiac surgery. The reasons for late symptomatology of lymphatic injury are analyzed. Conservative treatment with low-fat diet, total parenteral nutrition and pleural drainage was attempted unsuccessfully; chyle leakage of around 500 ml/day and onset of nutritional deficiency made it advisable to seal the thoracic duct surgically.     

Unusual association of chylopericardium and aortic hypoplasia in a neonate

We report the case of a neonate with two very rare anomalies: primary chylopericardium and diffuse hypoplasia of the thoraco-abdominal aorta. The presentation on the 16th post-natal day was with dyspnoea and refusal to feed. The initial clinical examination revealed hepatomegaly and weak femoral pulses. 2D ultrasound gave a diagnosis of a large compressive pericardial effusion combined with moderate hypoplasia of the aortic isthmus. Emergency pericardial drainage removed 80ml of chylous liquid. Following prolonged parenteral nutrition, the pericardial effusion stabilised. A mechanism of diffuse vascular disease affecting the aorta and the lymphatic system is suggested.     

Mesenteric lymph node abscess due to Yersinia enterocolitica: case report and review of the literature

We describe the case of a 74-year-old female with a mesenteric lymph node abscess caused by a Yersinia enterocolitica infection. She had been administered an immunosuppressive drug and was admitted to the hospital due to a high fever, right lower abdominal pain and advanced leukocytosis. We initially diagnosed her with lymphadenitis based on the symptoms and the imaging studies. However, conservative treatment with antibiotics did not yield any improvement, and abscess formation was suspected. Surgical treatment was performed, and the culture from the drainage fluid grew Y. enterocolitica. The histological findings suggested that an ulcerative lesion of the terminal ileum was the entry port of Y. enterocolitica. The pathogen infected the mesenteric lymph nodes and spread along the ileocecal lymphatic vessels, resulting in the formation of an abscess. We also provide a review of the previously published literature on lymph node abscesses due to Y. enterocolitica infections.     

Elephantiasis nostras--a case report

Elephantiasis nostras, the result of chronic lymphedema, is characterized by marked edema of the affected extremity with a thickened, verrucous, pebbly appearance of the skin. The pathogenesis is thought to be related to fibroblast proliferation following impaired lymphatic drainage, leading to fibrosis and further restriction of lymph drainage with progressive edema. A case report of a patient with massive chronic lymphedema of her feet is presented.     

Primary amyloidosis with pulmonary involvement which presented exudative pleural effusion and high fever

We describe here an extremely rare case of primary amyloidosis which presented moderate pleural effusion and high fever. A 71-year-old man was admitted to our hospital because of exertional dyspnea, fatigue and fever. A chest X-ray showed right-sided moderate pleural effusion. A thoracocentesis revealed an exudative pleural effusion. Cytology and cultures of the effusion were negative. External drainage failed to control the effusion. To determine the etiology of the effusion and fever, bronchoscopy was performed. Biopsies of the tracheal wall showed amyloid deposition. The pleural effusion might have been due to the inflammation and the disturbed lymphatic drainage caused by the amyloid deposition. Treatment with melphalan (6 mg) and prednisolone (35 mg) for 4 days every 6 weeks decreased the fever and alleviated his symptoms.     

Tularemia--an unusual cause of a solitary pulmonary nodule in the post-transplant setting.

We report a case of tularemia presenting as a solitary pulmonary nodule following syngeneic PBSC transplant. Seven months after undergoing a syngeneic PBSC transplant for AML, our patient presented with fever without localizing signs. Chest X-ray revealed a solitary pulmonary nodule. Culture of a CT guided needle aspiration revealed Francisella tularensis. The patient was successfully treated with ciprofloxacin. His fever resolved and clearance of the nodule was documented on a CT scan 2 months after diagnosis and initiation of treatment. To our knowledge, this is the only reported case of tularemia occurring in the post-transplant setting. The possible relationship between transplant-induced immune dysfunction and the occurrence of this rare infection is discussed.     

Cervical lymphatic malformations amenable to transhairline robotic surgical excision in children: A case series

Lymphatic malformations are rare benign malformations that predominantly occur in the head and neck region. The advent of surgical robots in head and neck surgery may provide beneficial outcomes for pediatric patients. Here, we describe our experiences with transhairline incisions for robot-assisted surgical resection of cervical lymphatic malformations in pediatric patients.In this prospective longitudinal cohort study, we recruited consecutive patients under 18 years of age who were diagnosed with congenital cervical lymphatic malformations and scheduled for transhairline approach robotic surgery at a single medical center. We documented the docking times, console times, surgical results, complications, and postoperative follow-up outcomes.The studied patients included 2 with mixed-type lymphatic malformations and 2 with macrocystic-type lymphatic malformations. In all 4 patients, the incision was hidden in the hairline; the incision length was <5 cm in 3 patients but was extended to 6 cm in 1 patient. Elevating the skin flap and securely positioning it with Yang retractor took <1 hour in all cases. The mean docking time was 5.5 minutes, and the mean console time was 1 hour and 46 minutes. All 4 surgeries were completed endoscopically with the robot. The average total drainage volume in the postoperative period was 21.75 mL. No patients required tracheotomy or nasogastric feeding tubes. Neither were adverse surgery-associated neurovascular sequelae observed. All 4 patients were successfully treated for their lymphatic malformations, primarily with robotic surgical excisions.Cervical lymphatic malformations in pediatric patients could be accessed, properly visualized, and safely resected with transhairline-approach robotic surgery. Transhairline-approach robotic surgery is an innovative method for meeting clinical needs and addressing esthetic concerns.     

Bilateral chylothorax after transsternal total thymectomy: resolution with short period of fasting and total parenteral nutrition

Bilateral chylothorax after transsternal total thymectomy is very rare, but can be a serious complication. Disruption of minor lymphatic channels in the anterior mediastinum which is remote from thoracic duct is considered to be the cause of chylothorax. We report the case of bilateral chylothorax followed by thymectomy which was treated with pleural drainage, total parenteral nutrition, and fasting without octreotide injection.     

<Emphasis Type="Italic">Francisella tularensis</Emphasis> aortitis

Francisella tularensis, the agent of tularemia, is a Gram-negative coccobacillus primarily pathogen for animals and occasionally for humans. The clinical manifestations of tularemia include pneumonia, ulceroglandular, oropharyngeal, or typhoidal disease. Rare manifestations are also described, but to our knowledge, we describe here the first case of F. tularensis aortitis in a human. Diagnosis was confirmed by the presence of F. tularensis in blood culture, by the presence of F. tularensis DNA in the aortic biopsy and by specific IgG and IgM responses against the bacteria. The outcome was favorable after surgery and specific antimicrobial therapy.     

Ecoepidemiology of tularemia in the southcentral United States

We combined county-based data for tularemia incidence from 1990 to 2003 for a nine-state region (Arkansas, Illinois, Indiana, Kansas, Kentucky, Missouri, Nebraska, Oklahoma, and Tennessee) in the southcentral United States with Geographic Information System (GIS)-based environmental data to determine associations between coverage by different habitats (especially dry forest representing suitable tick habitat) and tularemia incidence. High-risk counties (> 1 case per 100,000 person-years) clustered in Arkansas-Missouri and far eastern Oklahoma and Kansas. County tularemia incidence was positively associated with coverage by dry forested habitat suitable for vector ticks for Oklahoma-Kansas-Nebraska and Arkansas-Missouri but not for Illinois-Indiana-Kentucky-Tennessee. A multivariate logistic regression model predicting presence of areas with risk of tularemia based on GIS-derived environmental data was developed for the Arkansas-Missouri tularemia focus. The study shows the potential for research on tularemia ecoepidemiology and highlights the need for further modeling efforts based on acarologic data and more fine-scale point or zip code/census tract epidemiologic data.