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Nocturia: An Uncommon Presentation of Lower-Limb Lymphedema

      To the Editor:
      We report a case of nocturia as an uncommon presentation of lower-limb lymphedema in a 56-year-old man. To our knowledge, this is the first case reported in the literature.

      Case Report

      A 56-year-old man was admitted to the Department of Internal Medicine, University of Genova, on May 2008 for progressively worsening edema of the lower limbs. He had a history of nocturia (6-7 voids per night) since February 2007. Laboratory examination showed normal serum creatinine (0.7 mg/dL), urea nitrogen (13 mg/dL), and glucose (68 mg/dL). Urinalysis revealed normal urine pH (5.5), specific gravity (1.020), and osmolarity (750 mOsm/kg), and absence of proteinuria. Microalbuminuria, serum total protein, and albumin levels were in the normal range (20 mg/24 h, 7.2 g/dL, and 4.2 g/dL, respectively). Serum levels of protein C, protein S, and antithrombin III were in the normal range. Methylenetetrahydrofolate reductase C677T gene mutation, factor V Leiden, and prothrombin G20210A gene mutations were absent. Thoracic and abdominal computed tomography scans excluded thoracic duct obstruction and thrombosis of the superior and inferior venae cavae and the renal and iliac veins, and showed normal kidneys without perfusion defects. Lower-limb venous Doppler ultrasonography excluded venous insufficiency and thrombosis. Echocardiography showed only septal myocardial hypertrophy. Lower-limb 99mTc lymphoscintigraphy suggested dilated superficial lymphatic collectors and deep lymphatic trunks, delayed and asymmetric visualization of regional lymph nodes, and the presence of “dermal back-flow.” Clinical and imaging data were suggestive for the diagnosis of lower-limb lymphedema. Treatment with low-dose aspirin (100 mg/d), low-molecular-weight heparin (4.000 IU/d), and chlortalidone (25 mg/d) was started. Edematous enlargement was treated with massage and a compression stocking. This approach was helpful and allowed the complete remission of nocturia and lymphedema within 10 days. The use of an elastic stocking transmitted high-grade compression (60 mm Hg) and prevented fluid accumulation. At present, the patient feels good, always uses an elastic stocking, and is receiving treatment with low-dose aspirin (100 mg/d).

      Discussion

      Nocturia, defined as “waking at night to void,” is now being examined as a clinical entity in its own right, as opposed to being viewed as a symptom of another condition.
      • Appell R.A.
      • Sand P.K.
      Nocturia: etiology, diagnosis, and treatment.
      • Van Kerrebroeck P.
      • Abrams P.
      • Chaikin D.
      • et al.
      The standardization of terminology in nocturia: Report from the standardization subcommittee of the International Continence Society.
      Nocturia can be associated with the administration of drugs (ie, diuretics, β-blockers, xanthines), diabetes mellitus, diabetes insipidus, congestive heart failure, low blood albumin, venous stasis, high salt intake, renal insufficiency, and sleep apnea syndrome.
      • Weiss J.P.
      • Blaivas J.G.
      Nocturnal polyuria versus overactive bladder in nocturia.
      • Weiss J.P.
      • Blaivas J.G.
      Nocturia.
      Our patient developed nocturia as the first manifestation of an idiopathic form of lymphedema caused by lymphatic vessel insufficiency. A broad spectrum of inherited and acquired diseases are characterized by an impaired ability of the lymphatic vasculature to collect and transport fluid.
      • Rockson S.G.
      Diagnosis and management of lymphatic vascular disease.
      Primary lymphedema comprises a heterogeneous group of recessive and dominant transmitted disorders.
      • Rockson S.G.
      Diagnosis and management of lymphatic vascular disease.
      Secondary lymphedema is favored by surgical and radiotherapeutic interventions for cancer,
      • Szuba A.
      • Rockson S.
      Lymphedema: a review of diagnostic techniques and therapeutic options.
      pregnancy, contact dermatitis, and rheumatoid arthritis.
      • Rockson S.G.
      Diagnosis and management of lymphatic vascular disease.
      Although the diagnosis of lymphedema relies on physical examination (edema, peau d'orange, cutaneous fibrosis, and positive “Stemmer sign”), 99mTc lymphoscintigraphy plays a key role in confirming the suspicion. Our patient responded to treatment with lymphatic-specific massage, exercise, applied compression, and diuretics, in agreement with current literature.
      • Rockson S.G.
      Diagnosis and management of lymphatic vascular disease.
      To our present knowledge, this is the first reported case of nocturia as the first presentation of lower-limb lymphedema.

      Conclusions

      The present case suggests that nocturia may be the first symptom of lower-limb lymphedema and that lymphatic-specific massage, compression stocking, and diuretics might be an adequate therapeutic approach.

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