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REPORT OF EXPERIENCE WITH KIMURA'S DISEASE (기무라씨 질환, 5 예 보고)

  • Seel David J.;Park Yoon-Kyu;Lee Kwang-Min
    • Korean Journal of Head & Neck Oncology
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    • v.5 no.1
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    • pp.39-46
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    • 1989
  • Kimura's Disease is a chronic inflammatory and proliferative condition producing subcutaneous masses especially in the head and neck area. This report of our experience with 5 patients with this disease is the first in the Korean surgical literature. Kimura's Disease is thought to be part of the larger spectrum of the entity known as angiolymphoid hyperplasia with eosinophilia (ALHE). It is characterized pathologically by hyperplastic lymphoid follicles, eosinophilic infiltration, and vase 비 ar proliferation. It produces masses which are most common in the area of the parotid, submandibular gland and upper neck. These masses occupy the subcutaneous tissues but also extend into salivary tissue and into upper neck nodes. One of our patients had masses in the groin. The tumors are extremely vascular due to the presence of new proliferative vessels and sinusoids. The average age of our 5 patients was 35, but all but one case were younger than 38 years of age. The male: female ratio was 3 : 2, and the average duration of symptoms was 5,2years. All patients had peripheral blood eosinophilia. All had multiple masses, sometimes symmetrical. The management was surgery alone in one case, surgery and steroids in one case, surgery and radiotherapy in two cases, and all three modalities in one case. The relationship of this entity to ALHE and our experience in the management of this disease are presented. A clinicopathological discrepancy alerted us to the existence of Kimura's Disease. A nineteen-year old male presented with subcutaneous masses over both mastoid areas present for 3 years (Case III). When biopsy on each side was reported as 'eosinophilic granuloma' we submitted the slides to an internationally expert pathologist. Symmetrically occurring tumors in the peri-parotid subcutaneous areas did not fit any category of neoplasm or granuloma known to us. The diagnosis, made by Dr. Gist Fan at the Ochsner Clinic, was Kimura's Disease. We found two additional cases in a review of soft tissue eosinophilic granuloma previously reported at Presbyterian Medical Center, and since then have diagnosed two new cases. These five cases constitute the basis for this, the largest series to be reported in Korea. These vascular, tumor-like lesions of the skin, subcutaneous areas and subjacent structures of the head and neck have been a variety of names, such as angiolymphoid hyperplasia with eosinophilia, eosinophilic hyperplastic lymphogranuloma, angioblastic lymphoid hyperplasia with eosinophilia, histioid hemangioma, and epithelioid hemangioma. The history of this disease spectrum dates back to 1937 when Kimm and Szeto (1) reported 7 cases of 'eosinophilic hyperplastic lymphogranuloma' in the Proceedings of the Chinese Medical Journal. In 1948 Kimura and his associates(2) reported additional cases in Japan under the title 'On the unusual granulation combined with hyperplastic changes of lymphatic tissue.' From then until 1966 several hundred cases were reported in China and Japan. The first report from the West was by Wells and Whimster(3) in the British Journal of Dermatology, in 1969. These authors coined the term, angiolymphoid hyperplasia with eosinophilia (ALHE). Since that time a debate has ensued as to whether Kimura's Disease and ALHE are distinct entities, or whether Kimura's is part of the larger spectrum of ALHE, perhaps a later or advanced phase. From the clinical perspective, surgeons should be aware of the diagnosis of Kimura's Disease not only as part of the differential diagnosis of head and neck tumors but also because these lesions are indolent, and generally require conservative surgical removal as part of the management program. CASE I. A 37-year-old female company employee presented in August 1982 with submental swelling of 12 years' duration and with inguinal swelling of 7 years' duration. The submental mass measured 5x5cm. and the inguinal mass was 8x4cm. in size. Peripheral eosinophilia varying from 14% to 40% was found. On August 20, 1982, the submental mass was removed and a superficial groin dissection was done. In May 1983 an intraoral lesion of the palate was removed. The patient is free of disease. CASE II. A 23-year-old unemployed man visited this hospital for the first time in July, 1984, with swelling of the right cheek present for 6 years. The mass was soft and ill-defined but measured 10x20cm. and extended from the submandibular upper neck to the zygomatic arch, and from the mastoid to the cheek, over the parotid gland. Eosinophilia varying from 27% to 29% was noted in the peripheral blood. On March 21, 1986, the lesion was resected. The procedure comprised an extended superficial parotidectomy from the temporalis fascia to the upper neck. Post-operatively radiotherapy 3000 rad tissue dose was administered using the 6 MeV linear accelerator. The patient remains free of disease. CASE III. A 19-year-old student came to the clinic with masses over both mastoid areas, present 3 years. On the right there were two adjacent lesions, one over the mastoid, the other in the upper jugular level of the neck. On the left it was a single mass over the mastoid. Eosinophilia varied from 13 to 32% in the peripheral blood, and 11.6% in the bone marrow. Incisional biopsy revealed 'eosinophilic granuloma' and a trial of predisolone was employed. The mass increased in size so a small dose of radiation (600 rads) was used, with substantial regression,. The lesion on the left was excised and follwed by 1000 rads radiotherapy. Finally recurrent tumor on the right side was removed on November 5, 1985. The patient remains free of disease. CASE N. A 29-year-old local merchant had had swelling of both upper necks since childhood. At the time of his first visit on March 17, 1986, the right submandibular mass measured 5x3.5cm. and the ,right upper neck and parotid tail mass measured 2.5cm. On the left there were masses in the upper neck, the largest of which measured 2.5cm, and of the parotid tail, 2.0cm. in size.(See Fig. 1) Peripheral eosinophilia of 39% was recorded. Left side partial parotidectomy and resection of the upper neck and subdigstric mases was done on May 2, 1986. The mass involving the right parotid tail and upper neck nodes was removed on Angust 7,1986. Postoperatively the patient was placed on prednisolone 30 mg. per day. No definite masses are palpable. CASE V. A 66-year-old housewife informed us, at the time of her first visit in May, 1986, that she had had multiple neck masses since 10 years ago. On the right side there was a 2.5cm. subcutaneous mass of the upper neck, over the upper jugular chain. On the left there was a 9x4.5cm. mass involving the entire parotid, the post-auricular area and the upper neck. A third mass presented in the submental area and measured 3.5cm. (See Fig. 2) Eosinophilia of 51% was noted in the peripheral blood. partial excision of the left upper neck lesion and complete excision of the submental mass were performed on june 6, 1986. post-operatively she was placed on 20 mg. of prednisolone daily, but when the mass re-grew after two months she was referred to Radiation Therapy for a 2500 rad course of treatment. A barely palpable thickening remains.

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Current Status and Transition of the Prevalence of Intestinal Parasitic Infections in Korea (한국(韓國)에 있어서의 장내기생충감염(腸內寄生虫感染)의 현황(現況)과 추이(推移))

  • Kim, Dong-Chan
    • Journal of agricultural medicine and community health
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    • v.9 no.1
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    • pp.83-108
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    • 1984
  • Out of a total of 58 species of helminthic and protozoan parasitic infections in Korea, so far 38 species were reported as intestinal parasites of man. Quite a few species of the intestinal parasitic infections have long been prevalent throughout the country and this has been a significant public health problem. In this paper, current status and transition of the intestinal parasitic infections in the past years were presented. Chronological reviewing of data show background and prospects of change in the prevalence of infections. In the national prevalence survey on parasitic infections conducted once every five years since 1971, stool examinations were done by both cellophane thick smear and brine flotation techniques. Every egg positive case of Ascaris lumbricoides and Clonorchis sinensis was further examined by Stoll's dilution egg counting technique. In 1981, perianal swab using adhesive cellulose tape was added for Enterobius infection. For protozoan cyst examination conducted by province and city in '81, fecal specimens were fixed in SAF solution and examined by the formalin-ether concentration technique. High prevalence of parasitic infection in ana before the 1960s can be easily understood from the data given by the Ministry of Health and Social Affairs in 1967. From a parasite control point of view, the 1960s was the preparatory period particularly for control of soil-transmitted helminthiasis. Several organizations which have contributed to parasite control were founded in this period and the prevention law of parasitic infections was passed in '66. In the '70s, overall prevalence rates of the common intestinal parasitic infections, which were highly prevalent in the past were turned into reduction phase for the first time. The '80s can be said to be an active control period of parasitic infections. Intestinal helminths According to the reports of the national prevalence survey on parasitic infections, the prevalence of helminthic infections was 84.3%(number of person examined:24,887) in '71, 63.2%(27,178) in '76, and 41.1%(35,018) in '81. By area, the prevalence rate in '81 was 35.1%(20,569) in urban areas and 49.6%(14,447) in rural areas. Intestinal nematodes Ascaris lumbricoides The prevalence of Ascaris infection has decreased significantly in recent years. Among students, the prevalence was 55.4%, in '69 and decreased to 4.7% in '83. In the national prevalence surveys, the prevalence decreased to 13.0% in '81 from 54.9% in '71. By area, the prevalence decreased to 8.5% in '81 from 46.4% in '71 in urban area and 19.4% in '81 from 59.6% in '71 in rural area. By age, the prevalence has become in recent years relatively even in all age groups, although higher prevalence used to be seen in young age groups of around 10 years old, particulary in the highly prevalent rural areas. By sex, the prevalence was higher in the female than in the male. Unfertilized egg positive rates among the ascariasis cases increased gradually up to 55.4% on the average in '81. The intensity of the infection was also significantly decreased. Trichuris trichiura Trichuris infection had also decreased to 23.4% in '81 from 65.4% in '71. By area, the decreasing tendency of the prevalence became faster in urban areas than in the rural areas. The prevalence in urban and rural areas in '71 was 69.7%, and 63.1% respectively and decreased to 19.5% and 29.0% respectively in '81. By age, the prevalence reached a peak at the 10-14 age group and showed relatively even distribution throughout all age groups. By sex, the prevalence was close in young age groups, but in the 30s or over age group, especially in rural area, the prevalence was significantly higher in the female than in the male. The prevalence has much fluctuated depending in the area. The prevalence in rural areas surveyed in the '80s shows a range between 20.9% and 73.7% by locality. It is anticipated that the prevalence of Trichuris infection will drop more rapidly, when mass treatment is conducted. Hookworms Hookworm infection by mostly Ancylostoma duodenale and a few by Necator americanus has decreased to a negligible levels in recent years. In the national prevalence surveys, the prevalence was 10.7% in '71, 2.2% in '76, and 0.5% in '81. The prevalence was higher in rural areas than in urban areas. Wide application of multi-specific anthelminthics in the ascariasis control programmes conducted in the past decade appear to have been effective against hookworm infection. Trichostrongylus orientalis As in the case with hookworm infection, the prevalence of Trichostrongylus infection has reached a negligible levels. In the national prevalence surveys, the prevalence was 7.7% in '71, 1.0% in '76 and 0.2% in '81. Enterobius vermicularis In the national prevalence survey in '81, the egg positive rate was 12.0%. Higher prevalence is expected when examined repeatedly. The prevalence rate was 10.3% in urban area and 14.6% in rural area. In recent surveys conducted in rural areas among schoolchildren, the prevalence was 32.4% in Gimhae Gun in '82 and 64.1% in Yeongyang Gun in '83. By age, the egg positive rate was higher in young age groups of around 10 and sharply decreased in age groups of around 20 and then somewhat increased again in middle age groups. By sex, the prevalence was higher in the female than in the male. Strongyloides stercoralis Strongyloides stercoralis infection has rarely been found in Korea. Three cases were reported in 1914. And 0.1-0.5% were found infected out of 2,642 persons examined at the prisoner-of-war camp on Geojedo in 1956. One case was reported in '54 and '82, respectively. Anisakis spp. No systematic survey has been conducted for anisakiasis In Korea. So far, only several cases have been found 1 case in Seoul in '71, 5 cases in Busan in '81 and 1 case in Busan in '84. Intestinal trematodes Metagonimus yokogawai In the national prevalence survey conducted in 1981, the egg positive rate was 1.24% on the average. High endemic areas are located in the southwestern part of Korea. The prevalence in Hadong Gun was 29.1% on the average in '79. In a survey conducted in 76, the prevalence was 44.0% in Gwangyang, 55.0% in Gogseung and 29.0% in Gurye. The infection is closely correlated with raw sweetfish consumption in these areas. Other intestinal trematodes A human case of Heterophyes heterophyes was reported in 1914. Several species were reported in the '80s : 17 cases of Fibricola seoulensis, 9 cases of Pygidiopsis summa, 8 cases of Heterophyes heterophyes nocens, 1case of Heterophyopsis continua, 2 cases of Stellantchasmus falcatus, 1 case of Stictodora sp., 1 case of Echinostoma hortense, and 4 cases of Echinochasmus japonicus. As the intermediate hosts, snakes and frogs play a role for F.seoulensis and fish for the rest of the species. Intestinal cestodes Taenia saginata and T. solium Egg positive rates in the national prevalence survey were 0.7% in '76 and 1.1% in '81. The prevalence in '81 was 0.6% in urban area and 1.8% in rural area. The proglottid positive rate in Jeju Do was 19.2% on the average. On Udo, Jeju Do in 1983, the egg positive rate among the inhabitants was 2.9%. Hymenolepis nana In the national prevalence survey, egg positive rates were 0.6% in '76 and 0.4% in '81. No difference was seen in the prevalence by area and sex. Hymenolepis diminuta Infected cases were reported : 3 in '64 and I in '66. Egg positive rate in '81 was 0.01% in the national prevalence survey. Diphylobothrium latum So far, about 30 cases have been reported. The cases have been reported more frequently in recent surveys. Mesocestoides sp. A case was reported from a hospitalized patient in Seoul in '67. Spirometra erinacei Two cases were reported in '84 following reidentification of the adult worms collected in '74. Intestinal protozoa Out of a total of 23 species of human protozoan infections in Korea, 13 species were reported as intestinal protozoa : Entamoeba histolytica, E coli, Endolimax nana, Iodamoeba b$\ddot{u}$tschlii, Dientamoeba fragilis, Giardia lamblia, Chilomastix mesnilii, Embadomonas sp., Enteromonas hominis, Trichomonas hominis, Isospora belli, I. Hominis(Sarcocystis hominis), and Balantidium coli. Since the first report on intestinal protozoan infections in 1925, there have been quite a few survey data on the prevalence of the infection. It was found reviewing the data chronologically that up to the early '70s the infection was prevalent around a 30-50%. After that, the protozoan cyst positive rate has shown the tendency of gradual decrease throughout the country. Protozoan cyst survey conducted in Seoul and several provinces in 1981 revealed infection rates of 8.9%(1,310) in Gangweondo, 10.7%(1,703) in Gyenggi Do, 11.7%(1,032) in Jeonra Buk Do, 9.1%(4,116) in Jeonra Nam Do, and 1.4%(5,275) in Seoul. Entamoeba histolytica In the survey conducted by province in '81, the cyst positive rate was 0.8% in Gangweon-do, 0.3% in Gyeonggi Do, 1.4% in both Jeonra Buk Do and Jeonra Nam Do, and 0.2% in Seoul. Giardia lamblia In the survey by province in '81, cyst positive rates were 2.2% in both Gyeonggi Do and Jeonra Buk Do, 1.9% in Jeonra Nam Do, 0.5% in Gangweon Do, and 0.9% in Seoul. Balantidium coli Two cases were reported. One in 1930 and the other in '74. Isospora belli and I. Hominis(Sarcocystis hominis) Isospora belli was reported : 1 case in '56 and 3 cases in '66. I. Hominis, recently identified to be synonymous with Sarcocystis hominis, was reported : 3 cases in '66. Other intestinal protozoa The protozoan parasites other than the above mentioned are generally treated as commensal, although some of them are considered to be pathogenic. The data of '81 show that about 10% of the inhabitants are still infected with protozoa.

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