Kampo Extract of Shinbuto Improved Refractory Diarrhea in Milroy's Disease

Milroy's disease is a hereditary congenital lymphedema caused by lymphatic obstruction. The legs are most commonly affected, but impaired intestinal lymphatic flow can cause loose bowel movements. Here, we report the use of the Kampo extract of shinbuto for successful treatment of and abdominal pain in a patient with Milroy's disease. Milroy's disease was diagnosed because of left leg lymph-edema with onset at birth. Conservative therapy with a compression bandage was applied. However, when the patient moved to Manila at 35 years of age, she was exposed to drastic temperature changes between the air-conditioned cold environment in her room and the hot and humid environment outside. She developed a constitutional state of coldness as in hiesho (冷え症). Then sudden lower abdominal pain and diarrhea began to occur 3 times per week and lasted at least 1 hour, sometimes accompanied by vomiting. It happened particularly when she was exposed to the cold environment and was not related to meals. Conventional anti-cholinergic or antidiarrhetic drugs had no therapeutic effect. These attacks continued in the same frequency for 3 years, so the patient visited a Kampo (traditional Japanese medicine) clinic, where her diagnosis of Milroy's disease–associated diarrhea and abdominal pain was augmented by the Kampo diagnosis of hiesho, suitai (body fluid retention). She was prescribed 7.5 g of shinbuto extract per day (TJ-30; Tsumura Co, Tokyo, Japan). The shinbuto extract significantly reduced abdominal pain and refractory diarrhea to about 2 days per month, and it tapered off completely in 3 months. Shinbuto is usually used against cold-induced diarrhea. Rewarming and water movement by shinbuto resulted in significant improvement in symptoms induced by hiesho and suitai triggered by the cold environment, though the patient's leg swelling did not change.