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Hennekam syndrome is a rare autosomal recessive disorder resulting from malformation of the lymphatic system. The characteristic signs of Hennekam syndrome are lymphangiectasia, lymph edema, facial anomalies, and mental retardation. This is a case in which a patient presented with left-arm lymphedema, facial-feature anomalies, and multiple organ lymphangiectasia consistent with symptoms of Hennekam syndrome. There is no curative therapy at this time, but rehabilitative treatments including complete decongestive therapy for edema control appeared to be beneficial.
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To predict the probability of lymphedema development in breast cancer patients in the early post-operation stage, we investigated the ability of quantitative lymphoscintigraphic assessment.
This retrospective study included 201 patients without lymphedema after unilateral breast cancer surgery. Lymphoscintigraphy was performed between 4 and 8 weeks after surgery to evaluate the lymphatic system in the early postoperative stage. Quantitative lymphoscintigraphy was performed using four methods: ratio of radiopharmaceutical clearance rate of the affected to normal hand; ratio of radioactivity of the affected to normal hand; ratio of radiopharmaceutical uptake rate of the affected to normal axilla (RUA); and ratio of radioactivity of the affected to normal axilla (RRA). During a 1-year follow-up, patients with a circumferential interlimb difference of 2 cm at any measurement location and a 200-mL interlimb volume difference were diagnosed with lymphedema. We investigated the difference in quantitative lymphoscintigraphic assessment between the non-lymphedema and lymphedema groups.
Quantitative lymphoscintigraphic assessment revealed that the RUA and RRA were significantly lower in the lymphedema group than in the non-lymphedema group. After adjusting the model for all significant variables (body mass index, N-stage, T-stage, type of surgery, and type of lymph node surgery), RRA was associated with lymphedema (odds ratio=0.14; 95% confidence interval, 0.04–0.46; p=0.001).
In patients in the early postoperative stage after unilateral breast cancer surgery, quantitative lymphoscintigraphic assessment can be used to predict the probability of developing lymphedema.
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We reported on a 60-year-old man presenting lymphedema of both lower extremities and scrotum for 3 years with unknown cause. We took a computed tomography scan of the lower extremities as a follow-up. There were diffuse subcutaneous edema in both lower extremities and multiple enlarged lymph nodes along the para-aortic and bilateral inguinal areas. For further evaluation, biopsy of an enlarged inguinal lymph node was taken, yielding a diagnosis of primary amyloidosis. A treatment of chemotherapy for amyloidosis was recommended for him. To our knowledge, this is the first report of lymphedema presenting with primary amyloidosis in Asia. This case suggests that primary amyloidosis could be one of the differential diagnoses in patients with lymphedema in the lower extremities.
To evaluate the prevalence and associated factors involved in cellulitis with lymphangitis among a group of Korean patients who were being treated for lymphedema. We present our epidemiologic research and we also report a systematic review of these types of cases.
This was a retrospective medical record study among 1,246 patients diagnosed with lymphedema. The study was carried out between January 2006 and December 2012 at the Kosin University Gospel Hospital and Seoul National University Bundang Hospital. Cases were examined for onset time, affected site, seasonal trend, and recurrence pattern of lymphedema, lymphangitis, and cellulitis. We also evaluated the history of blood-cell culture and antibiotic use.
Ninety-nine lymphedema patients experienced complications such as cellulitis with accompanying lymphangitis. Forty-nine patients had more than two recurrences of cellulitis with lymphangitis. The incidence and recurrence of cellulitis with lymphangitis were significantly higher in the patients with lower-extremity lymphedema. There was a significant trend toward higher cellulitis prevalence in the lower-extremity lymphedema group according to the time of lymphedema onset. Among the cellulitis with lymphangitis cases, 62 cases were diagnosed through blood-cell culture; 8 of these 62 cultures were positive for β-hemolytic streptococci.
The prevalence rate of cellulitis with lymphangitis in patients with lymphedema was 7.95%, and the prevalence of recurrent episodes was 3.93%. Especially, there was high risk of cellulitis with lymphangitis after occurrence of lower-extremity lymphedema with passage of time. Lymphedema patients should be fully briefed about the associated risks of cellulitis before treatment, and physicians should be prepared to provide appropriate preventive education.
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To evaluate the validity of quantitative lymphoscintigraphy as a useful lymphedema assessment tool for patients with breast cancer surgery including axillary lymph node dissection (ALND).
We recruited 72 patients with lymphedema after breast cancer surgery that included ALND. Circumferences in their upper limbs were measured in five areas: 15 cm proximal to the lateral epicondyle (LE), the elbow, 10 cm distal to the LE, the wrist, and the metacarpophalangeal joint. Then, maximal circumference difference (MCD) was calculated by subtracting the unaffected side from the affected side. Quantitative asymmetry indices (QAI) were defined as the radiopharmaceutical uptake ratios of the affected side to the unaffected side. Patients were divided into 3 groups by qualitative lymphoscintigraphic patterns: normal, decreased function, and obstruction.
The MCD was highest in the qualitative obstruction (2.76±2.48) pattern with significant differences from the normal (0.69±0.78) and decreased function (1.65±1.17) patterns. The QAIs of the axillary LNs showed significant differences among the normal (0.82±0.29), decreased function (0.42±0.41), and obstruction (0.18±0.16) patterns. As the QAI of the axillary LN increased, the MCD decreased. The QAIs of the upper limbs were significantly higher in the obstruction (3.12±3.07) pattern compared with the normal (1.15±0.10) and decreased function (0.79±0.30) patterns.
Quantitative lymphoscintigraphic analysis is well correlated with both commonly used qualitative lymphoscintigraphic analysis and circumference differences in the upper limbs of patients with breast cancer surgery with ALND. Quantitative lymphoscintigraphy may be a good alternative assessment tool for diagnosing lymphedema after breast cancer surgery with ALND.
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To support the establishment of lymphedema education plans and the actual practice of education by investigating the current lymphedema awareness status of Korean breast cancer patients.
cross-sectional population survey was conducted in 116 breast cancer patients in the Busan-Gyeongnam area. The survey included questions regarding demographic characteristics, breast cancer-related lymphedema (BCRL) risk factors, and characteristics and treatments of the disease. Some of the items were scored to determine the level of awareness. The items that affect the awareness of lymphedema were investigated by statistical analysis.
Eighty-one of the 116 patients answered that they had heard of lymphedema, and 30 of them (25.86%) had received explanations about the possibility of lymphedema before surgery. Only 20 patients (17.25%) knew that lymphedema is not a completely curable disease, 24 patients (20.68%) thought that lymphedema does not require any treatment, and only 56 patients (48.27%) knew that lymphedema is treated in the Department of Rehabilitation Medicine. The main factors that affected patients' awareness of lymphedema were their age, chemotherapy, duration of breast cancer, and lymphedema treatment history.
The majority of survey participants who were breast cancer patients either lacked awareness of BCRL or had false ideas about it, indicating the inadequate level of education provided for lymphedema. In the case of breast cancer diagnosis, early and continuous education for future management is essential, and the framework for the provision of education including education protocols related to age, disease duration, and lymphedema treatment is needed.
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Two patients with stage three secondary lymphedema of the upper extremities underwent treatment for breast cancer, including surgery, chemotherapy, and radiotherapy. They were examined with computed tomography (CT) before and after extracorporeal shock wave therapy (ESWT). We used a manual tracing method using PiViewSTAR software to calculate the volume of the upper extremities. There was a decrease in the volume of the subcutaneous compartment measured by CT before and after ESWT. CT may be helpful in determining the treatment target area of ESWT and to monitor the effect of treatment by measuring the changes in volume before and after ESWT in patients with lymphedema. Therefore, CT may have good clinical potential for treatment and follow-up in the management of lymphedema.
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To investigate the changing patterns of edema, quality of life (QOL), and patient-satisfaction after complex decongestive therapy (CDT) in three trajectories: arm lymphedema (AL), secondary leg lymphedema (LL) and primary leg lymphedema (PL).
Candidates for AL (n=35), LL (n=35) and PL (n=14) were identified from prospective databases. The patients were treated with CDT for 2 weeks, and lymphedema volume was measured before and immediately following the therapy. Patients then self-administered home therapy for 3 months and presented for a follow-up visit. The Korean version of Short Form-36 (SF-36) was used to assess QOL, and we administered a study-specific satisfaction survey.
There was no significant difference in the volume reductions between the 3 groups. There were no significant differences in all of the measures between PL and LL. Overall initial QOL was significantly lower in patients with LL than in patients with AL. SF-36 scores post-CDT did not differ significantly between AL and LL. Clinically significant differences were noted between AL and LL in the mean values of the satisfaction survey.
AL, LL, and PL may have different longitudinal courses. We suggest that lower extremity lymphedema patients present more favorable outcomes after CDT with respect to QOL and satisfaction than upper extremity lymphedema patients. Clinicians should approach patients with different therapeutic considerations specific to each type or region of lymphedema before using CDT in clinical practice.
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To investigate the long-term effects of complex decongestive therapy (CDT) on edema reduction in breast cancer-related lymphedema patients after axillary dissection, according to the initial volume of edema.
A retrospective review of 57 patients with unilateral arm after an axillary dissection for breast cancer was performed. The patients, treated with two weeks of CDT and self-administered home therapy, were followed for 24 months. Arm volume was serially measured by using an optoelectronic volumeter prior to and immediately after CDT; and there were follow-up visits at 3, 6, 12, and 24 months. Patients were divided into two groups according to the percent excess volume (PEV) prior to CDT: group 1, PEV<20% and group 2, PEV≥20%.
In group 1, mean PEV before CDT was 11.4±5.0% and 14.1±10.6% at 24 months after CDT with no significant difference. At the end of CDT, PEV was 28.8±15.7% in group 2, which was significantly lower than the baseline (41.9±19.6%). The reduction of PEV was maintained for 24 months in group 2.
The long-term effects of CDT were well-maintained for 24 months, but there was a difference in progression of PEV between the two groups. The patients with more initial PEV showed significant volume-reducing effects of CDT. In patients with less initial PEV, the severity of lymphedema did not progress to higher grades.
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To evaluate the usefulness of ultrasonography as a follow-up tool for evaluating the effects of complex decongestive physiotherapy (CDPT) in breast cancer-related lymphedema (BCRL).
Twenty patients with BCRL were enrolled in this study. All patients had undergone therapy in the CDPT program for 2 weeks. Soft tissue thickness of both the affected and unaffected upper limb was measured before and after CDPT. The measurements were taken at 3 points (the mid-point between the medial and lateral epicondyles at the elbow level, 10 cm proximal and 10 cm distal to the elbow) with and without pressure. We then calculated the compliance of soft tissue before and after CDPT. Circumferences of both the affected and unaffected upper limb were also measured before and after CDPT at the 3 defined points.
After 2 weeks of the CDPT program, the circumference and soft tissue thickness of the unaffected upper limb did not significantly change. In the affected upper limb, the circumference was significantly reduced in the 3 point, when compared with measurements taken prior to treatment. Additionally, soft tissue thickness was significantly reduced at the elbow and 10 cm proximal to the elbow. After CDPT, compliance at each of the 3 points had increased, but this trend was not significantly different.
Our results showed that arm circumference and ultrasonography-derived soft tissue thickness was useful as a way of assessing therapeutic effects of CDPT.
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To evaluate the effect of complex decongestive physiotherapy (CDPT) on malignant lymphedema patients.
Patients (n=22) with malignant lymphedema of the upper or the lower limb were assigned to this study. CDPT without manual lymphatic drainage (MLD) was used five times per week for two weeks. The main outcome measurements included measurement of the circumference of the limb (proximal, distal, and total) to assess volume changes. We also employed the visual analog scale (VAS) to evaluate pain, and the short form-36 version 2 questionnaire (SF-36) to assess quality of life (QOL). All items were assessed pre and post-treatment for each patient.
There was a statistically significant difference in the volume change of the upper limbs (3.7%, p=0.001) and the lower limbs (10.9%, p=0.001). A 1.5 point reduction on the ten-point VAS was noted after CDPT. The scores on the physical and the mental components of the SF-36 showed statistical improvement after treatment (p=0.006, p=0.001, respectively).
These results suggest that all components of the CDPT program except MLD are helpful in treating malignant lymphedema in terms of pain reduction and reduction of the volume of the affected upper or lower limb. This treatment regimen also has positive effects on QOL.
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To investigate the clinical effect of extracorporeal shock wave therapy (ESWT) in patients with secondary lymphedema after breast cancer treatment.
In a prospective clinical trial, ESWT was performed consecutively 4 times over two weeks in 7 patients who were diagnosed with stage 3 secondary lymphedema after breast cancer treatment. Each patient was treated with four sessions of ESWT (0.056-0.068 mJ/mm2, 2,000 impulses). The parameters were the circumference of the arm, thickness of the skin and volume of the arm. We measured these parameters with baseline values before ESWT and repeated the evaluation after each ESWT treatment. Subjective data on skin thickness, edema and sensory impairment were obtained using a visual analogue scale (VAS).
The mean volume of the affected arm after four consecutive ESWT was significantly reduced from 2,332 to 2,144 mL (p<0.05). The circumference and thickness of the skin fold of the affected arm were significantly decreased after the fourth ESWT (p<0.05). The three VAS scores were significantly improved after the fourth ESWT. Almost all patients were satisfied with this treatment and felt softer texture in their affected arm after treatment.
ESWT is an effective modality in the treatment of stage 3 lymphedema after breast cancer treatment. ESWT reduced the circumference and the thickness of arms with lymphedema and satisfied almost all patients with lymphedema. Therefore, this treatment provides clinically favorable outcome to patients with breast cancer-related lymphedema.
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To investigate the effect of repetitive magnetic stimulation (rMS) of the spinal cord on limb angiogenesis in healthy rats and explore its implication for the treatment of lymphedema.
Twelve adult male Sprague-Dawley rats were divided into four groups as follows: sham rMS followed by tissue harvest 5 minutes later (group 1, n=2), 1 Hz rMS and tissue harvest 5 minutes later (group 2, n=3), 20 Hz rMS and tissue harvest 5 minutes later (group 3, n=3), 20 Hz rMS and tissue harvest 30 minutes later (group 4, n=4). Animals were treated with 20-minute rMS with 120% of the motor threshold on their left side of upper lumbar spinal cord. Expression of angiogenic factors, that is, Akt, phospho-Akt (pAkt), endothelial nitric oxide synthase (eNOS), phospho-eNOS (p-eNOS) were measured by western blot. Bilateral hindlimb muscles (quadriceps and gastrocnemius) were harvested.
Expression of Akt in left quadriceps increased in group 4 compared with group 2 and 3 (3.4 and 5.3-fold each, p=0.026). Expression of eNOS in left plus right quadriceps markedly increased in group 3 and 4 compared with group 1 and 2 (p=0.007). Expressions of eNOS, Akt and p-eNOS, pAkt in gastrocnemius were not comparable between four groups (p>0.05).
Repetitive magnetic stimulation of the spinal cord may exert an angiogenic effect closely linked to lymphangiogenesis. It has clinical implication for the possible therapy of lymphedema caused by breast, cervical or endometrial cancer operation. Future studies with the specific lymphatic endothelial cell markers are required to confirm the effect of rMS on lymphangiogenesis.
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To evaluate quality of life (QOL), upper extremity function and the effect of lymphedema treatment in patients with breast cancer related lymphedema.
The basic data comprised medical records (detailing age, sex, dominant side, location of tumor, cancer stage, operation record, cancer treatment and limb circumferences) and questionnaires (lymphedema duration, satisfaction, self-massage). Further to this, we measured upper extremity function and QOL, administered the DASH (Disabilities of Arm Shoulder and Hand outcome measure) and used the EORTC (European Organization for Research and Treatment of Cancer)-QLQ-C30 and the EORTC-QLQ-Br23. Results of these were calculated as main outcome variables.
The questionnaire responses and arm circumferences of 59 patients with breast cancer related lymphedema were analyzed. In the DASH questionnaire, it was found that the older the lymphedema patient was, the lower their upper extremity function. On the EORTC-QLQ, patients with metastasis had significantly lower scores in physical functioning and role functioning. In terms of upper extremity circumference, there was a significant upper extremity size reduction after lymphedema treatment.
There were several dissociations between some subscales of quality of life questionnaires and those of upper extremity functions. Upper extremity function was correlated with the age of breast cancer patients and QOL was influenced by M-stage. Lymphedema treatment was found to be effective in reducing edema in patients with breast cancer related lymphedema.
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Lipedema is a chronic disease of lipid metabolism that results in the symmetrical impairment of fatty tissue distribution and storage combined with the hyperplasia of individual fat cells. Lipedema occurs almost exclusively in women and is usually associated with a family history and characteristic features. It can be diagnosed based on clinical history and physical examination. Lipedema is usually symmetrical, but spares the feet, is often painful to palpation, and is negative for Stemmer's sign. Additionally, lipedema patients can present with microangiopathies and lipomas. The well-known therapies for lipedema include complex decongestive therapy, pneumatic compression, and diet modifications. However, whether these treatments help reduce swelling is debatable. We encountered a case of lipedema that was initially misdiagnosed as lymphedema. The patient's clinical features and history were different from those typical of lymphedema, prompting a diagnosis of lipedema and she was treated with a complex decongestive therapy program.
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To investigate the usefulness of bioimpedance measurement for predicting the treatment outcome in breast cancer related lymphedema (BCRL) patients.
Unilateral BCRL patients who received complex decongestive therapy (CDT) for 2 weeks (5 days per week) were enrolled in this study. We measured the ratio of extracellular fluid (ECF) volume by using bioelectrical impedance spectroscopy (BIS), and single frequency bioimpedance analysis (SFBIA) at a 5 kHz frequency before treatment. Arm circumferences were measured at 10 cm above and below the elbow before and after treatment. We also investigated whether there is correlation between ECF ratio and SFBIA ratio with the change of arm circumference after CDT.
A total of 73 patients were enrolled in this study. The higher ECF ratio was significantly correlated with higher reduction of arm circumference at both above and below the elbow after treatment, but the higher SFBIA ratio was correlated only with the higher reduction of arm circumference below the elbow.
These results show that ECF volume measurements and SFBIA before treatment are useful tools for predicting the outcome of patients with lymphedema. We concluded that ECF volume measure can be used as a screening tool for predicting treatment outcome of BCRL patients.
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