A 57-year-old Asian woman experienced an abnormal feeling on the left side of her neck and difficulty breathing 90 minutes after eating a Chinese noodle dish containing roasted pork, egg, spinach, seasoned bamboo shoots, laver, and garlic. She was advised to call an ambulance by our triage nurse after telephone consultation and was transported to our hospital by a physician-staffed helicopter due to the long distance from her house to the hospital. She had a history of removal of a left sphenoid ridge meningioma one year earlier. She had experienced limited rigidity of her left neck and limited peripheral cold sensation on her left side in winter since approximately 10 years of age. She had experienced limited peripheral swelling of her left side and limited lower back pain of unknown origin on her left side several times. She had been suffering from oral allergy syndrome since she was young. She sometimes experienced a tingling sensation on her lips and an unpleasant feeling in her throat after eating some types of fruit. Those fruits included mandarins, oranges, kiwis, bananas, and pineapples. She was married but did not have any children.\nOn arrival, 180 minutes after eating the noodles, she had clear consciousness, and her blood pressure was 130/90 mmHg, her heart rate was 80 beats per minute, her percutaneous oxygen saturation was 100% under room air, and her body temperature was 36.9°C. She had left neck and chest swelling without color change. Her breathing difficulty subsided spontaneously.\nThe results of a blood analysis were as follows: white blood cells, 6,000/mm3; hemoglobin, 14.8 g/dL; platelets, 38.3 × 104/mm3; aspartate aminotransferase, 15 IU/L; alanine aminotransferase, 11 IU/L; total bilirubin, 0.5 mg/dL; total protein, 6.7 g/dL; glucose, 118 mg/dL; blood urea nitrogen, 18.9 mg/dL; creatinine, 0.90 mg/dL; amylase, 63 IU/L; creatine phosphokinase, 89 IU/L; sodium, 142 mEq/L; potassium, 4.0 mEq/L; chloride, 106 mEq/L; C-reactive protein, 0.05 mg/dL; prothrombin time, 10.6 (control, 11.9) sec; activated partial thrombin time, 26.4 (control, 26.9) sec; fibrinogen, 310 mg/dL; fibrin degradation products, 0.8 μg/mL; troponin T, 0.06 (0-0.14) ng/mL; and immunoglobulin E (IgE), 429 (0-250) IU/mL. Chest roentgen, an electrocardiogram and cardiac echography findings were all negative. Neck computed tomography (CT) with contrast medium and magnetic resonance imaging (MRI) revealed left-side-limited edema in the subcutaneous area and surrounding esophagus and bronchus (Figures , ).\nThese radiological findings excluded hemorrhaging or pseudoaneurysm formation. She underwent observational admission. After her edema improved, she was discharged on the third hospital day. A follow-up examination one week later showed complete resolution of the neck and chest edema. The results of a blood allergen test analysis (0-1.39) showed an elevated immunoglobulin E (IgE) level as follows: Japanese cedar, 44.30; betula alba, 7.27; orchard grass, 3.42; Japanese cypress, 2.65; dermatophagoides farina, 3.03; house dust, 1.85; and mugwort, 1.70. The remaining allergens were within the normal ranges, as follows (0-1.39): alder 1.35, timothy 0.85, 0.33; cat skin, 0.25; and Candida, 0.22. The results for the following allergens were all zero: ragweed, Alternaria, dog skin, wheat, buckwheat, soybean, rice, tuna, salmon, lobster, crab, milk, beef, chicken, pork, albumen, ovomucoid, peanut, sesame, kiwi, banana and latex. We also obtained blood samples from the bilateral upper extremities at the same time. The examination items that showed ≥5% laterality were as follows: white blood cell count (6,500/mm3 vs. 6,100/mm3), platelets (40.8 × 104/mm3 vs. 38.0 × 104/mm3, aspartate aminotransferase (12 IU/L vs. 11 IU/L), lactate dehydrogenase (184 IU/L vs. 172 IU/L) and creatinine (0.63 mg/dL vs. 0.58 mg/dL). We could not obtain permission from the patient for further a genetic analysis by skin biopsy.
A 29-year-old female patient had complained of discomfort and mild pain in the left shoulder since she was 18-year-old. After childbirth, she had severe ongoing pain and contracture of the shoulder. She had no significant prior medical or surgical history. Axial fat-saturated T2-weighted MRI revealed high signal intensity and sagittal T1-weighted MRI revealed low signal intensity within the muscle of the shoulder. (Fig. c, d). Under clinical diagnosis of VM, the lesion was resected.