The Journal of Dermatology Vol. 6: 2] 1217.1979
STATISTICAL SURVEY OF 100 CASES OF SPOROTRICHOSIS HIDEO KARIYA
AND
TOKIO IWATSU
ABSTRACT
A statistical survey of 100 cases of sporotrichosis at the Dermatological Clinic of Chiba University Hospital from May, 1965 to June, 1975 was reported. The proportion of sporotrichosis cases to the total number of outpatients and to the total dermatomycoses was 0.14% and 1.8%. It has increased gradually since 1969. Greater percentage of cases fell into the less than 11 year old or more than 40 year old age groups. Females were more affected than males. Half of the cases occured in farmers. Geographic distribution was remarkable. 44 cases had a history of injury. 62 cases showed the cutaneous lymphatic type and 38 cases showed the localized cutaneous type. The face and upper extremities were the most affected. The sporotrichin test was positive in 41 of 42 cases. The causative organism was demonstrated in tissue sections in 97 of 99 cases.
INTRODUCTION
In May of 1965, the first case of sporotrichosis was seen at the Dermatology Clinic of Chiba University Hospital. Thereafter, with the cooperation of related hospitals, many more cases of sporotrichosis have been collected, chiefly in Chiba and Ibaraki prefectures. The number of cases of sporotrichosis has remarkably increased since 1969; it reached 50 by May of 1972 and 100 by June of 1975. Though a statistical study on the former 50 cases has already been reported (1), we now wish to report a statistical survey of all 100 cases, in view of the fact that there a report from one institution on 100 cases of this disease has never appeared in the Japanese literature. STATISTICAL OBSERVATIONS
Materials. Those cases of sporotrichosis were made the materials of study which were diagnosed as such at the Dermatology Clinic of Chiba University Hospital and were ascertained by isolating a culture By Invitation: Received October ]6, 1978; accepted for publication May I], 1979. From the Department of Dermatology (Director: Prof. Shoji Okamoto), School of Medicine, Chiba University, Chiba 280. Japan.
of Sporothrix schenckii from the lesion. Annual incidence. Table 1 shows the annual trend in the number of cases. The ratios of the number of sporotrichosis cases to the total number of outpatients and to that of dermatomycosis patients seen at the Dermatology Clinic of Chiba University Hospital were 0.14% and 1.8% respectively, for the average of the 10 year period from 1965 to 1974. These ratios were 0.056% and 0.81 % during the period from 1965 to 1968; however, the ratios have rapidly increased since 1969 and were calculated as 0.20% and 2.4% during the period from 1969 to 1974. Though these percentage are
Table 1.
Annual incidence male
1965 1966 1967 1968 1969 1970 1971 1972 1973 1974 1975 (to June) total
female
total
2 6 5 6
0 3 2 3 7 6 9 10 10 8 9
10 12 16 13 15
33
67
100
3 I I
1 2 5 I
3 4 3 4 9 11
212
KARIYA AND IWATSLJ
high in comparison with those from other institutions in Japan, they are far lower than the data of Sampaio & Lacaz (2) who reported that this disease comprised 0.5% of the total cases of all dermatoses in St. Paul in South America. Although, according to Fukushiro (3), the first case of sporotrichosis reported in Japan by Nishizawa & Tanabe in 1920 seems to be an uncertain case, no more than 13 certain cases were reported during the 20 year period thereafter until the end of the Second World War. However, the number of reported cases has gradually increased since the war, especially markedly since 1965. As mentioned above, the first case of this disease reported by the Dermatology Clinic of Chiba University Hospital was seen in 1965, which seems fairly late as compared with the first reported cases of other institutions, but the number of our reported cases has remarkably increased subsequently. This is due on one hand, to the fact that we have actively collected cases with the co-operation of allied hospitals; but, on the other hand, one of the factors which may have increased the number of reported cases is the suggestion that sporotrichosis is comparatively frequently seen in Kanto district. In addition, patients have been enabled to go to hospitals more easily owing to the diffusion of the nation-wide health insurance system, the adjustment of road traffic networks in Chiba and Ibaraki prefectures, the popularization of owner drivers, and so forth. Age and sex. Table 2 shows the age and sex distribution of sporotrichosis cases. The youngest case
Table 2.
Age and sex incidence male
female
total
0-11 years 12-19 20-29 30-39 40-49 50-59 60-69 70-
10 1 4 1 2 7 2 6
10 1 3 4 16 13 15 5
20 2 7 5 18 20 17 11
total
33
67
100
youngest: 87 days (female) oldest: 84 years (female)
was a girl 87 days old, and the oldest was a 84-yearold woman. The age distribution reveals that the largest number were seen in the age groups under 12 and in the sixth decade, the next number in the fifth and seventh decades, and the fewest in the age groups between 10 and 40. The 87 day old girl seems to be the youngest reported case in Japan. The male to female ratio of our cases was 1:2 and accorded well with Fukushiro's report (male 39.5%, female 60.5%). However, in the age group between the fifth and seventh decades, females were far more frequently affected; the ratio became 1:4. Occupation. Forty-eight patients, the majority of cases, were farmers. There were 35 farmers' wives, which formed one third of the total cases. Fourteen cases were company employees, 12 were ordinary housewives, and 6 were tradespeople. According to Sampaio & Lacaz's (2) and Nakajima's (4) reports, occupation has nothing to do with the incidence of infection, but in our cases, farmers were most common, as we previously reported, and comprised one half of the total cases. In the cases of children under 10 y, no corelation was found with agriculture. Geographical distribution. The sites of residence of the patients at the time of onset were Chiba Prefecture in 77 cases, Ibaraki Prefecture in 19 cases, Saitama Prefecture in 2 cases, Kanagawa Prefecture in I case and Brazil in 1 case. The Brazilian case was our second one. This patient was an emigrant who developed sporotrichosis while he was engaged in mushroom gathering in Brazil and happened to appear for consultation at the Dermatology Clinic of Chiba University Hospital when he came home. The distribution of the cases in Chiba and Ibaraki Prefectures was found to be dense in the plateaus from Yachiyo City to Narashino City, in and around Narita City and Yachimata, in and around Yokaichiba City and Togane City, and from the Tonegawa River to the area around Ryugasaki City of Ibaraki Prefecture; but sparse in the southern parts of Chiba Prefecture and in the Boso Peninsula (Fig. 1). This pattern of distribution of cases is closely related to climate. When the monthly average temperature and the amount of rainfall in the cold season are observed as
213
STATiSTICAL SURVEY OF !flO CASES OE SPOROTRICHOSIS
Table 3.
(1)Chiba (2J Narashino @Yachiyo @Narita @Yachimata @Sawara CVYokaichiba ®Togane ®Ryugasaki
Month of onset
8
Sept. Oct. Nov. Dec. Jan. Feb.
13 12 13
7 7
Mar.
7 6
Apr. May Jun. Jcl. Aug.
3 3
2 2
total unknown
'6"C The monthly average temperature Geographical Distribution and the amount of rainfall in January
Fig. I.
Geographical distribution and climate.
in Fig. 1, those areas with an average temperature of 0°_4°C and with rainfall of less than 50mm correspond to the areas of frequent development of sporotrichosis. Mackinnon's (5) and Abe's (6) reports also tell that sporotrichosis frequently developed in areas with a small amount of rainfall during cold seasons. Month of onset. Monthly distribution of onset of the disease is shown in Table 3. Frequent onset was found during cold seasons from December to April. Month of onset was not always certain in the cases of aged persons or those which had begun a long period before the first examination. Period from onset to first examination. The time period before the first examination was 10 days at the shortest and about 7 years at the longest; half of the cases consulted doctors within 4 months (Table 4). It should be noted that, during these periods, most of the cases had been treated as bacterial infections with antibiotics or with surgical procedures at various medical facilities. Episode of trauma. Forty-four cases had distinct episodes of trauma. Chief traumas were bruises, abnmons, insect bites, and stab wounds caused by wire or bamboo (Table 5).
Table 4.
83 17
Period from onset to first examination type L.
typeF.
total
within one month 2monrhs 3 4 5 6 7 8- 9 10-11 I year 1.5 2 3 beyond 5 years
5 11 11 8 3 5
5 3 1 2 2
I 2 2 2 5 2
8 17 15 10 5 7 4 6 2 7 5 3 7 4
total
62
38
100
I
4 I
3 6 4 2 2 2 3 2
shortest: 10 days longest: 7 years Table 5.
Incidence of trauma
yes type: bruise and abrasion insect bite stab wound (wire, iron fragment. prickle. etc.) scratch wound (dog, cat) cut wound (glass fragment, knife, etc.) miscellaneous no obscure
44 12 10 10 4 , 2 , 6
52 4
214
KARIYA AND IWATSt'
Table 6.
Interrelation between type of disease, localization and age (I) cutaneolyrnphangitic type - 62 cases
under II years 12-19 face rt , upper limb It. upper limb neck chest rt. lower limb It. lower limb
5
total
6
20-29
30-39
4049
50-59
4 5 2 2
2 6 6
I
2
60-69 4 9
over 70
total
3 4
17 24 16 2
I
I
0 2 4
Table 7.
2
14
15
13
62
Interrelation between type of disease, localization and age (2) localized cutaneous type - 38 cases
under II years
12-19
20-29
verrucous type: face
30-39
40-49
50-59
60-69
over 70
2
total
3
ulcerative type: face rt , upper limb It. upper limb It. lower limb nodulo-pustular type: face rt. upper limb It. upper limb rt. lower limb
7
I
2
3 I
4 5 3 I
12
14 4 2
I
2
I
fistular type: face total
14
3
Types of disease and sites of affection. According to Sampaio & Lacaz's classification, our cases can be divided as follows; 62 cases of cutaneolymphangitic type (type L), 38 cases of localized cutaneous type (type F) and no cases of any other type. Tables 6 and 7 show the interrelationships between type of disease, site of affection and age. Type F was frequent in children under the age of II, and type L was dominant in patients older than 19 (there were no cases between 12 and 18 years of age). According to Fukushiro's statistics, the ratio of type L: type F was 47.4: 50.8. But contrary to his statistics, our data showed type L dominant, Sampaio and Lacaz
3
3
6
4
4
38
also reported type L dominant case (76: 22.5). According to S. Watanabe's (9) and S. Watanabe's (7) statements, recent reports (4,7-11) in Japan indicate that type F is increasing and its cause is the shortening of the period from the onset of the disease to the first examination, due to elevation in patients' recognition of the disease, advance of doctors' diagnostic techniques, and improvement in medical conditions. In our cases, however, no significant differences were found between the two types related to the periods from the onset to the first examination (Table 4). Those data reported by S. Watanabe and S. Watanabe might indicate an in-
215
STATISTICAL SURVf.Y OF 100 CASES OF SPOROTRICHOSIS
crease in all cases of this disease rather than an increase in type F. Cutaneous sporotrichosis is usually classified into 3 types, type L, type F and a disseminated type, but as there are no distinct definitions of the types Land F, though most cases belong to these two types, we are at times embarassed at the classification of some cases. Kariya (12), one of the present authors, advocated a new type called "intermediate type" to include borderline cases in order not to forcibly classify them into type L or type F and to ease classification. According to this classification, 'our cases are composed of 52 cases of type L, 34 cases of type F, and 14 cases of intermediate type (Table 8: type L-+l0 cases of intermediate type; type F-+4 cases of intermediate type). The sites of affection were the face in 39 cases, neck in 2 cases, chest in 1 case, upper limbs in 53 cases and lower limbs in 5 cases. On the face, the periocular region and cheeks were predominantly affected, and on the arms, the right side was slightly more frequently involved. Lesions of the arms were mostly found on the hand to forearm and in only 7 cases were found to extend to the upper arm. In the cases of children, 19 of 20 developed lesions on the
Table 8.
face. The above data accord well with those of previous reports that the predilection sites were the face and arms, and especially, in the cases of children, the face (Table 9). Intracutaneous sporotrichin reaction. Sporotrichin tests were carried out in 42 cases, and positive reactions occurred in 41 cases (97.6%) and a doubtful reaction in one case (Table 10). According to Fukushiro, 528 cases of 579 cases showed positive reactions (91. 7 %), 9 cases doubtful reactions (1.6%) and 39 cases negative reactions (6.8%), and established the diagnostic utility of this reaction. Recently, differences between Sporothrix schenckii .and the genus Ceratocystis have been discussed. Though S. schenckii Hektoen et Perkins belongs to fungi imperfecti taxonomically, its generatio perfecto may be classified as Ceratocystis of the true ascomycetes. In order to make a comparative study of the relationship between the two fungi from the viewpoint of antigen, intracutaneous tests were carried out in 24 cases of this disease with antigen solution made from culture filtrates of 4 species of the genus Ceratocystis (G. stenoceras, C.
Table 9.
Age incidence of disease type
cutaneolymphangitic type
6
56
62
face: forehead eye-lid cheek nose mandible
localized cutaneous type: verrucous type ulcerative type nodule-pustular type fistular type
14 1 1 12 0
24 2 12 12 1
38
neck
total
20
80
100
Sampaio and Lacaz's classification under over 11 years 12 years
total
over 12 years
19 (95%) 1 8 8 1 1
20 (25%) 2 7 8 1 2
total
2
46 22 12
52 34 14
total
20
80
100
2
0 0 1
lower limb rt.
0 0
2 3
5
20
80
100
Table 10.
6 12 2
2
3
upper limb rt , It.
31 21 (65%)
total
cutaneolyrnphangitic type fixed type intermediate type
39 3 15 16
chest
total
under over 11 years 12 years
under 11 years
0
It. Kariya's classification
Site affected
Intracutaneous sporotrichin reaction
positive: doubtful: not yet done:
41 (97.6%) 1 ( 2.4%) 58
53
216
KARIYA AND IWATS\;
Table 11.
Fungus element in tissue
positive: free fungus element fungus element in giant cell asteroid body negative: not yet done:
97 (98.0%) 93 (93.9%) 41 (41.4%) 39(39.4%) 2 ( 2.0%) 1
minor, C. ulmi and C. IPS), together with the intracutaneous sporotrichin test. The results obtained are as follows: in reactivity, S. schenchii and genus Caratocystis (esp, C. stenoceras) showed similar tendencies, but significant differences were observed statistically. It was concluded that the two fungi are similar but not identical from the viewpoint of anti genicity. Fungus elements in tissue. Fungus elements in the tissue were studied with PAS staining. Twenty serial sections were prepared and examined initially, and, if no fungus elements were found, 20-40 more sections were additionally examined. The results were as follows: some fungus elements were found histologically in 97 of 99 cases (98.0%); free fungus elements in 93 cases (93.9%), fungus elements in giant cells in 41 cases (41.4 %) and asteroid bodies in 39 cases (39.4%) (Table 11). The remaining two cases in which no fungus elements were found seem to be due to the fact that the tissue specimens were insufficiently taken and therefore few serial sections could be made. A survey of studies on fungus elements in tissues reveals that some fungus elements were found by N akajima et al. (4) in 17 of 20 cases, by Morishita et al. (10) in 20 of26 cases, and by Kitamura et al. (11) in 21 of 28 cases. According to Fukushiro's statistics, fungus elements were observed in 626 (69.8%) of 896 cases of this disease; free fungus elements in 570 cases (63.7%), fungus elements in giant cells in 218 cases (24.3%), and asteroid bodies in 212 cases (23.7 %). In comparison with these reports, the positive rate of our fungus element research is fairly high. This result is thought to be due to our energetic research using 20-60 serial sections. Fukushiro et al. (16) has stated that the positive rate will approach 100% if many PAS-stained sections
are made for examination of fungus elements. Our data endorse this statement. Nishimura and Miyaji (18) observed many fungus elements in scales and crusts covering the surfaces of the lesions in all their 13 cases. The mycelial type was found in 9 of the 13 cases, and a peritheciurnlike organ was found in a crust of one case which seemed to have originated from S. schencku (17). Treatment. Oral treatment with potassium iodide was carried out in all cases, and local thermotherapy with Kairo (pocket heater) was used additionally in some cases, which led in all cases to a complete cure. Our mode of administration of potassium iodide was usually as follows. In adult cases, 0.6g per day was given initially, and the daily dose was then gradually increased to 1.8-2.1 g. In children's cases, 0.1 g was administered initially, and the dose was gradually increased to 0.3-0.6g according to age. The whole period of administration was 3 months in principle. Except in one case, no side effects of potassium iodide were observed. Recurrences occurred in two cases. In one of the two cases, as the lesions rapidly improved one week after commencement of treatment (0.6 g per day), the treatment was discontinued, and the lesion recurred 3 months later, but was completely cured by readministration of KI. In the one other case, although the lesion seemed to be completely cured by the above-mentioned formula of administration, it recurred 6 months later in the fall, and was treated with local thermotherapy. The potassium iodide therapy could not be continued because of gastrointestinal symptoms. Another patient who also suffered diabetes had not been cured by oral treatment with potassium iodide, and was cured by combination oflocal thermotherapy. CONCLUSIONS
A statistical study was made on 100 cases of sporotrichosis which have been observed at the Dermatology Clinic of Chiba University Hospital during the 10 year period from May 1965 to June 1975, and the results observed have been reported in this paper. The authors give their deepest thanks to Prof. Shoji Okamoto.
STATISTICAL SURVEY OF !OO CASES OF SPOROTRICHOSIS
The gist of this report was given orally by Dr. Iwatsu at the 550th meeting of the Tokyo Society of Japanese Dermatology Congress.
REFERENCES 1) Kariya, H. and Okamoto. S.: 50 cases of Sporotrichosis,]apan.f. Med. Mycol., 13: 125. 1972. 2) Sampaio, S.A.P. u. Lacaz, C.S.: Klinische u. statistiche Untersuchungen iiber Sporotrichose in Sao Paulo (Brasilien), Hautarzt, 10: 490,1959. 3) Fukushiro, R.: The 18th annual meeting of Japanese Medical Mycology Congress. 1974. . 4) Nakajima, H .. Katakura, H., Yanagida. H. and Uchiyama, M.: Statistic observation on twenty-three cases of Sporotrichosis. Dermat. et Urolog., 20: 1137, 1966_ 5) Mackinnon. J.E.: The dependence on weather of the incidence of Sporotrichosis. Mycopathologia, 4: 365, 1947-1949. 6) Abe. Y., Gibo, M. and Zaima, S.: 3 cases of Sporotrichosis (with statistical observation of 134 hitherto published Japanese cases of the disease). Dermat. et Urolog., 17: 545. 1963. 7) Watanabe. S.. Morita. Y., Sudo, N., Soh, Y., Doi, A. and Matsuda. Y.: Sporotrichosis in Kansai district of Japan, Acta Dermat. (Kyoto), 63: 49, 1968. 8) Takiuchi, 1.: 50 cases of Sporotrichosis observed at the Dermatology Clinic of Nippon University Hospital, rinsho derma, 13: 283. 1971. 9) Watanabe. S.: Sporotrichosis in Chugoku district of Japan. rinsho derma, 27: 313.1973. 10) Morishita, R., Okada, T. and Tashiro. M.: Sporotrichosis in Kagoshima prefecture, Japan. I Med.
217
Mycol., 15: 143, 1974. ll) Kitamura, K., Harada, T., Nishikawa, T. and Hatano, H.: A clinico-histopathological investigation of cutaneous Sporotrichosis,]ap. I Clin. Dermat., 29: 853, 1975. 12) Kariya , H.: A classification of Sporotrichosis .japan. I Med. Mycol., 16: 87, 1975. 13) Iwatsu, T., Kariya, H., Ishizaki, H. and Nakamura. Y.: Studies on the relationship between Sporothrix schenckii and Ceratocystis species (I) Skin test of Ceratocystis antigen in the patients with cutaneous Sporotrichosis.jap.f. Derm., 86: 544, 1976. 14) Ishizaki, H .. Nakamura. Y.. Kariya, H .. Iwatsu, T. and Wheat, R.: Delayed hypersensitivity crossreaction between Sporothrix schenckii and Ceratocystis species in Sporotrichotic patients. I Clin. Microbiol., 3: 545. 1976. 15) Iwatsu, T. and Miyaji, M.: A comparison of antigenicity between Sporothrix schenckii and some Ceratocystis species-Studies on the cell-mediated immune response, Japan. I Med. Mycol., 17: 136, 1976. 16) Fukushiro, R.. Kagawa. S., Nishiyama, S., Takahashi, H. und Ishikawa. H.: Die Plizelement im Gewebe der Hautsporotrichose des Menschen Hautarzt, 16: 18. 1965. 17) Nishimura, K.. Miyaji, M. and Kariya, H.: Studies on the parasitic form of Sporothrix schenckii in scales and crusts. (II) Perithecium like organs found in crusts. Japan.f. Med. Mycol., 15: 117, 1974. 18) Nishimura. K. and Miyaji, M.: Studies on the parasitic form of Sporothrix schenckii in scales and crusts. (III) Clinical study of thirteen cases, Japan. I Med. Mycol., 16: 57.1975.
STATISTICAL SURVEY OF 100 CASES OF SPOROTRICHOSIS HIDEO KARIYA
AND
TOKIO IWATSU
ABSTRACT
A statistical survey of 100 cases of sporotrichosis at the Dermatological Clinic of Chiba University Hospital from May, 1965 to June, 1975 was reported. The proportion of sporotrichosis cases to the total number of outpatients and to the total dermatomycoses was 0.14% and 1.8%. It has increased gradually since 1969. Greater percentage of cases fell into the less than 11 year old or more than 40 year old age groups. Females were more affected than males. Half of the cases occured in farmers. Geographic distribution was remarkable. 44 cases had a history of injury. 62 cases showed the cutaneous lymphatic type and 38 cases showed the localized cutaneous type. The face and upper extremities were the most affected. The sporotrichin test was positive in 41 of 42 cases. The causative organism was demonstrated in tissue sections in 97 of 99 cases.
INTRODUCTION
In May of 1965, the first case of sporotrichosis was seen at the Dermatology Clinic of Chiba University Hospital. Thereafter, with the cooperation of related hospitals, many more cases of sporotrichosis have been collected, chiefly in Chiba and Ibaraki prefectures. The number of cases of sporotrichosis has remarkably increased since 1969; it reached 50 by May of 1972 and 100 by June of 1975. Though a statistical study on the former 50 cases has already been reported (1), we now wish to report a statistical survey of all 100 cases, in view of the fact that there a report from one institution on 100 cases of this disease has never appeared in the Japanese literature. STATISTICAL OBSERVATIONS
Materials. Those cases of sporotrichosis were made the materials of study which were diagnosed as such at the Dermatology Clinic of Chiba University Hospital and were ascertained by isolating a culture By Invitation: Received October ]6, 1978; accepted for publication May I], 1979. From the Department of Dermatology (Director: Prof. Shoji Okamoto), School of Medicine, Chiba University, Chiba 280. Japan.
of Sporothrix schenckii from the lesion. Annual incidence. Table 1 shows the annual trend in the number of cases. The ratios of the number of sporotrichosis cases to the total number of outpatients and to that of dermatomycosis patients seen at the Dermatology Clinic of Chiba University Hospital were 0.14% and 1.8% respectively, for the average of the 10 year period from 1965 to 1974. These ratios were 0.056% and 0.81 % during the period from 1965 to 1968; however, the ratios have rapidly increased since 1969 and were calculated as 0.20% and 2.4% during the period from 1969 to 1974. Though these percentage are
Table 1.
Annual incidence male
1965 1966 1967 1968 1969 1970 1971 1972 1973 1974 1975 (to June) total
female
total
2 6 5 6
0 3 2 3 7 6 9 10 10 8 9
10 12 16 13 15
33
67
100
3 I I
1 2 5 I
3 4 3 4 9 11
212
KARIYA AND IWATSLJ
high in comparison with those from other institutions in Japan, they are far lower than the data of Sampaio & Lacaz (2) who reported that this disease comprised 0.5% of the total cases of all dermatoses in St. Paul in South America. Although, according to Fukushiro (3), the first case of sporotrichosis reported in Japan by Nishizawa & Tanabe in 1920 seems to be an uncertain case, no more than 13 certain cases were reported during the 20 year period thereafter until the end of the Second World War. However, the number of reported cases has gradually increased since the war, especially markedly since 1965. As mentioned above, the first case of this disease reported by the Dermatology Clinic of Chiba University Hospital was seen in 1965, which seems fairly late as compared with the first reported cases of other institutions, but the number of our reported cases has remarkably increased subsequently. This is due on one hand, to the fact that we have actively collected cases with the co-operation of allied hospitals; but, on the other hand, one of the factors which may have increased the number of reported cases is the suggestion that sporotrichosis is comparatively frequently seen in Kanto district. In addition, patients have been enabled to go to hospitals more easily owing to the diffusion of the nation-wide health insurance system, the adjustment of road traffic networks in Chiba and Ibaraki prefectures, the popularization of owner drivers, and so forth. Age and sex. Table 2 shows the age and sex distribution of sporotrichosis cases. The youngest case
Table 2.
Age and sex incidence male
female
total
0-11 years 12-19 20-29 30-39 40-49 50-59 60-69 70-
10 1 4 1 2 7 2 6
10 1 3 4 16 13 15 5
20 2 7 5 18 20 17 11
total
33
67
100
youngest: 87 days (female) oldest: 84 years (female)
was a girl 87 days old, and the oldest was a 84-yearold woman. The age distribution reveals that the largest number were seen in the age groups under 12 and in the sixth decade, the next number in the fifth and seventh decades, and the fewest in the age groups between 10 and 40. The 87 day old girl seems to be the youngest reported case in Japan. The male to female ratio of our cases was 1:2 and accorded well with Fukushiro's report (male 39.5%, female 60.5%). However, in the age group between the fifth and seventh decades, females were far more frequently affected; the ratio became 1:4. Occupation. Forty-eight patients, the majority of cases, were farmers. There were 35 farmers' wives, which formed one third of the total cases. Fourteen cases were company employees, 12 were ordinary housewives, and 6 were tradespeople. According to Sampaio & Lacaz's (2) and Nakajima's (4) reports, occupation has nothing to do with the incidence of infection, but in our cases, farmers were most common, as we previously reported, and comprised one half of the total cases. In the cases of children under 10 y, no corelation was found with agriculture. Geographical distribution. The sites of residence of the patients at the time of onset were Chiba Prefecture in 77 cases, Ibaraki Prefecture in 19 cases, Saitama Prefecture in 2 cases, Kanagawa Prefecture in I case and Brazil in 1 case. The Brazilian case was our second one. This patient was an emigrant who developed sporotrichosis while he was engaged in mushroom gathering in Brazil and happened to appear for consultation at the Dermatology Clinic of Chiba University Hospital when he came home. The distribution of the cases in Chiba and Ibaraki Prefectures was found to be dense in the plateaus from Yachiyo City to Narashino City, in and around Narita City and Yachimata, in and around Yokaichiba City and Togane City, and from the Tonegawa River to the area around Ryugasaki City of Ibaraki Prefecture; but sparse in the southern parts of Chiba Prefecture and in the Boso Peninsula (Fig. 1). This pattern of distribution of cases is closely related to climate. When the monthly average temperature and the amount of rainfall in the cold season are observed as
213
STATiSTICAL SURVEY OF !flO CASES OE SPOROTRICHOSIS
Table 3.
(1)Chiba (2J Narashino @Yachiyo @Narita @Yachimata @Sawara CVYokaichiba ®Togane ®Ryugasaki
Month of onset
8
Sept. Oct. Nov. Dec. Jan. Feb.
13 12 13
7 7
Mar.
7 6
Apr. May Jun. Jcl. Aug.
3 3
2 2
total unknown
'6"C The monthly average temperature Geographical Distribution and the amount of rainfall in January
Fig. I.
Geographical distribution and climate.
in Fig. 1, those areas with an average temperature of 0°_4°C and with rainfall of less than 50mm correspond to the areas of frequent development of sporotrichosis. Mackinnon's (5) and Abe's (6) reports also tell that sporotrichosis frequently developed in areas with a small amount of rainfall during cold seasons. Month of onset. Monthly distribution of onset of the disease is shown in Table 3. Frequent onset was found during cold seasons from December to April. Month of onset was not always certain in the cases of aged persons or those which had begun a long period before the first examination. Period from onset to first examination. The time period before the first examination was 10 days at the shortest and about 7 years at the longest; half of the cases consulted doctors within 4 months (Table 4). It should be noted that, during these periods, most of the cases had been treated as bacterial infections with antibiotics or with surgical procedures at various medical facilities. Episode of trauma. Forty-four cases had distinct episodes of trauma. Chief traumas were bruises, abnmons, insect bites, and stab wounds caused by wire or bamboo (Table 5).
Table 4.
83 17
Period from onset to first examination type L.
typeF.
total
within one month 2monrhs 3 4 5 6 7 8- 9 10-11 I year 1.5 2 3 beyond 5 years
5 11 11 8 3 5
5 3 1 2 2
I 2 2 2 5 2
8 17 15 10 5 7 4 6 2 7 5 3 7 4
total
62
38
100
I
4 I
3 6 4 2 2 2 3 2
shortest: 10 days longest: 7 years Table 5.
Incidence of trauma
yes type: bruise and abrasion insect bite stab wound (wire, iron fragment. prickle. etc.) scratch wound (dog, cat) cut wound (glass fragment, knife, etc.) miscellaneous no obscure
44 12 10 10 4 , 2 , 6
52 4
214
KARIYA AND IWATSt'
Table 6.
Interrelation between type of disease, localization and age (I) cutaneolyrnphangitic type - 62 cases
under II years 12-19 face rt , upper limb It. upper limb neck chest rt. lower limb It. lower limb
5
total
6
20-29
30-39
4049
50-59
4 5 2 2
2 6 6
I
2
60-69 4 9
over 70
total
3 4
17 24 16 2
I
I
0 2 4
Table 7.
2
14
15
13
62
Interrelation between type of disease, localization and age (2) localized cutaneous type - 38 cases
under II years
12-19
20-29
verrucous type: face
30-39
40-49
50-59
60-69
over 70
2
total
3
ulcerative type: face rt , upper limb It. upper limb It. lower limb nodulo-pustular type: face rt. upper limb It. upper limb rt. lower limb
7
I
2
3 I
4 5 3 I
12
14 4 2
I
2
I
fistular type: face total
14
3
Types of disease and sites of affection. According to Sampaio & Lacaz's classification, our cases can be divided as follows; 62 cases of cutaneolymphangitic type (type L), 38 cases of localized cutaneous type (type F) and no cases of any other type. Tables 6 and 7 show the interrelationships between type of disease, site of affection and age. Type F was frequent in children under the age of II, and type L was dominant in patients older than 19 (there were no cases between 12 and 18 years of age). According to Fukushiro's statistics, the ratio of type L: type F was 47.4: 50.8. But contrary to his statistics, our data showed type L dominant, Sampaio and Lacaz
3
3
6
4
4
38
also reported type L dominant case (76: 22.5). According to S. Watanabe's (9) and S. Watanabe's (7) statements, recent reports (4,7-11) in Japan indicate that type F is increasing and its cause is the shortening of the period from the onset of the disease to the first examination, due to elevation in patients' recognition of the disease, advance of doctors' diagnostic techniques, and improvement in medical conditions. In our cases, however, no significant differences were found between the two types related to the periods from the onset to the first examination (Table 4). Those data reported by S. Watanabe and S. Watanabe might indicate an in-
215
STATISTICAL SURVf.Y OF 100 CASES OF SPOROTRICHOSIS
crease in all cases of this disease rather than an increase in type F. Cutaneous sporotrichosis is usually classified into 3 types, type L, type F and a disseminated type, but as there are no distinct definitions of the types Land F, though most cases belong to these two types, we are at times embarassed at the classification of some cases. Kariya (12), one of the present authors, advocated a new type called "intermediate type" to include borderline cases in order not to forcibly classify them into type L or type F and to ease classification. According to this classification, 'our cases are composed of 52 cases of type L, 34 cases of type F, and 14 cases of intermediate type (Table 8: type L-+l0 cases of intermediate type; type F-+4 cases of intermediate type). The sites of affection were the face in 39 cases, neck in 2 cases, chest in 1 case, upper limbs in 53 cases and lower limbs in 5 cases. On the face, the periocular region and cheeks were predominantly affected, and on the arms, the right side was slightly more frequently involved. Lesions of the arms were mostly found on the hand to forearm and in only 7 cases were found to extend to the upper arm. In the cases of children, 19 of 20 developed lesions on the
Table 8.
face. The above data accord well with those of previous reports that the predilection sites were the face and arms, and especially, in the cases of children, the face (Table 9). Intracutaneous sporotrichin reaction. Sporotrichin tests were carried out in 42 cases, and positive reactions occurred in 41 cases (97.6%) and a doubtful reaction in one case (Table 10). According to Fukushiro, 528 cases of 579 cases showed positive reactions (91. 7 %), 9 cases doubtful reactions (1.6%) and 39 cases negative reactions (6.8%), and established the diagnostic utility of this reaction. Recently, differences between Sporothrix schenckii .and the genus Ceratocystis have been discussed. Though S. schenckii Hektoen et Perkins belongs to fungi imperfecti taxonomically, its generatio perfecto may be classified as Ceratocystis of the true ascomycetes. In order to make a comparative study of the relationship between the two fungi from the viewpoint of antigen, intracutaneous tests were carried out in 24 cases of this disease with antigen solution made from culture filtrates of 4 species of the genus Ceratocystis (G. stenoceras, C.
Table 9.
Age incidence of disease type
cutaneolymphangitic type
6
56
62
face: forehead eye-lid cheek nose mandible
localized cutaneous type: verrucous type ulcerative type nodule-pustular type fistular type
14 1 1 12 0
24 2 12 12 1
38
neck
total
20
80
100
Sampaio and Lacaz's classification under over 11 years 12 years
total
over 12 years
19 (95%) 1 8 8 1 1
20 (25%) 2 7 8 1 2
total
2
46 22 12
52 34 14
total
20
80
100
2
0 0 1
lower limb rt.
0 0
2 3
5
20
80
100
Table 10.
6 12 2
2
3
upper limb rt , It.
31 21 (65%)
total
cutaneolyrnphangitic type fixed type intermediate type
39 3 15 16
chest
total
under over 11 years 12 years
under 11 years
0
It. Kariya's classification
Site affected
Intracutaneous sporotrichin reaction
positive: doubtful: not yet done:
41 (97.6%) 1 ( 2.4%) 58
53
216
KARIYA AND IWATS\;
Table 11.
Fungus element in tissue
positive: free fungus element fungus element in giant cell asteroid body negative: not yet done:
97 (98.0%) 93 (93.9%) 41 (41.4%) 39(39.4%) 2 ( 2.0%) 1
minor, C. ulmi and C. IPS), together with the intracutaneous sporotrichin test. The results obtained are as follows: in reactivity, S. schenchii and genus Caratocystis (esp, C. stenoceras) showed similar tendencies, but significant differences were observed statistically. It was concluded that the two fungi are similar but not identical from the viewpoint of anti genicity. Fungus elements in tissue. Fungus elements in the tissue were studied with PAS staining. Twenty serial sections were prepared and examined initially, and, if no fungus elements were found, 20-40 more sections were additionally examined. The results were as follows: some fungus elements were found histologically in 97 of 99 cases (98.0%); free fungus elements in 93 cases (93.9%), fungus elements in giant cells in 41 cases (41.4 %) and asteroid bodies in 39 cases (39.4%) (Table 11). The remaining two cases in which no fungus elements were found seem to be due to the fact that the tissue specimens were insufficiently taken and therefore few serial sections could be made. A survey of studies on fungus elements in tissues reveals that some fungus elements were found by N akajima et al. (4) in 17 of 20 cases, by Morishita et al. (10) in 20 of26 cases, and by Kitamura et al. (11) in 21 of 28 cases. According to Fukushiro's statistics, fungus elements were observed in 626 (69.8%) of 896 cases of this disease; free fungus elements in 570 cases (63.7%), fungus elements in giant cells in 218 cases (24.3%), and asteroid bodies in 212 cases (23.7 %). In comparison with these reports, the positive rate of our fungus element research is fairly high. This result is thought to be due to our energetic research using 20-60 serial sections. Fukushiro et al. (16) has stated that the positive rate will approach 100% if many PAS-stained sections
are made for examination of fungus elements. Our data endorse this statement. Nishimura and Miyaji (18) observed many fungus elements in scales and crusts covering the surfaces of the lesions in all their 13 cases. The mycelial type was found in 9 of the 13 cases, and a peritheciurnlike organ was found in a crust of one case which seemed to have originated from S. schencku (17). Treatment. Oral treatment with potassium iodide was carried out in all cases, and local thermotherapy with Kairo (pocket heater) was used additionally in some cases, which led in all cases to a complete cure. Our mode of administration of potassium iodide was usually as follows. In adult cases, 0.6g per day was given initially, and the daily dose was then gradually increased to 1.8-2.1 g. In children's cases, 0.1 g was administered initially, and the dose was gradually increased to 0.3-0.6g according to age. The whole period of administration was 3 months in principle. Except in one case, no side effects of potassium iodide were observed. Recurrences occurred in two cases. In one of the two cases, as the lesions rapidly improved one week after commencement of treatment (0.6 g per day), the treatment was discontinued, and the lesion recurred 3 months later, but was completely cured by readministration of KI. In the one other case, although the lesion seemed to be completely cured by the above-mentioned formula of administration, it recurred 6 months later in the fall, and was treated with local thermotherapy. The potassium iodide therapy could not be continued because of gastrointestinal symptoms. Another patient who also suffered diabetes had not been cured by oral treatment with potassium iodide, and was cured by combination oflocal thermotherapy. CONCLUSIONS
A statistical study was made on 100 cases of sporotrichosis which have been observed at the Dermatology Clinic of Chiba University Hospital during the 10 year period from May 1965 to June 1975, and the results observed have been reported in this paper. The authors give their deepest thanks to Prof. Shoji Okamoto.
STATISTICAL SURVEY OF !OO CASES OF SPOROTRICHOSIS
The gist of this report was given orally by Dr. Iwatsu at the 550th meeting of the Tokyo Society of Japanese Dermatology Congress.
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217
Mycol., 15: 143, 1974. ll) Kitamura, K., Harada, T., Nishikawa, T. and Hatano, H.: A clinico-histopathological investigation of cutaneous Sporotrichosis,]ap. I Clin. Dermat., 29: 853, 1975. 12) Kariya , H.: A classification of Sporotrichosis .japan. I Med. Mycol., 16: 87, 1975. 13) Iwatsu, T., Kariya, H., Ishizaki, H. and Nakamura. Y.: Studies on the relationship between Sporothrix schenckii and Ceratocystis species (I) Skin test of Ceratocystis antigen in the patients with cutaneous Sporotrichosis.jap.f. Derm., 86: 544, 1976. 14) Ishizaki, H .. Nakamura. Y.. Kariya, H .. Iwatsu, T. and Wheat, R.: Delayed hypersensitivity crossreaction between Sporothrix schenckii and Ceratocystis species in Sporotrichotic patients. I Clin. Microbiol., 3: 545. 1976. 15) Iwatsu, T. and Miyaji, M.: A comparison of antigenicity between Sporothrix schenckii and some Ceratocystis species-Studies on the cell-mediated immune response, Japan. I Med. Mycol., 17: 136, 1976. 16) Fukushiro, R.. Kagawa. S., Nishiyama, S., Takahashi, H. und Ishikawa. H.: Die Plizelement im Gewebe der Hautsporotrichose des Menschen Hautarzt, 16: 18. 1965. 17) Nishimura, K.. Miyaji, M. and Kariya, H.: Studies on the parasitic form of Sporothrix schenckii in scales and crusts. (II) Perithecium like organs found in crusts. Japan.f. Med. Mycol., 15: 117, 1974. 18) Nishimura. K. and Miyaji, M.: Studies on the parasitic form of Sporothrix schenckii in scales and crusts. (III) Clinical study of thirteen cases, Japan. I Med. Mycol., 16: 57.1975.
