J. Nihon

CASE

Univ.

Sch.

Dent.,

Vol.

34.

123-130.

1992

REPORT

A Case of Eosinophilic Granuloma Arising in the Mandible Teruhiko ISHII 1, Yasuhiro ISHIZAKI 1, Yuichi NAKADA 1, Mayumi KOIZUMI 1, Hiroyuki TATSUHARA 1, Mitsuhiko MATSUMOTO 1, Hiroshi TANAKA 1, Kazuo KOMIYAMA 2 and Itaru MORO 2 (Received9 Septemberand accepted22 October 1991) Key words:

eosinophilic granuloma, histiocytosis X, mandibular tumor Abstract

Eosinophilic granuloma is a disease of the reticuloendothelial system and its pathogenesis is not yet completely clear. The histopathological findings are granulomatous proliferation of histiocytes accompanied by diffuse infiltration of eosinophils. We recently experienced a case of eosinophilic granuloma arising monostotically in the body of the mandible in the 76 region. There has been no local recurrence 17 months after surgical removal of the lesion. Introduction Eosinophilic granuloma (EG) is a disease of the reticuloendothelial system, and histopathologically it is characterized by granulomatous proliferation of histiocytes accompanied by diffuse infiltration of eosinophils. The pathogenesis of EG is not yet completely clear. There are few Japanese case reports of EG arising in the maxilla and mandible. Here, therefore, we describe a recently experienced case of EG arising in the mandible. Case Report A 15-year-old Japanese girl was referred to the Department of Oral Surgery, Nihon University Dental Hospital, in October 1989 for evaluation of a swelling in the right mandibular molar region. The patient had become aware of slight pain in this region about one month before, but the pain was soon controlled by the use of analgesics. However, as slight swelling with spontaneous pain continued for two weeks, she was advised to undergo a further examination at our hospital. Extraoral examination revealed slight swelling of the right buccal area, but there was no abnormal appearance of the overlying skin (Fig. 1). One right submandibular lymph node about the size of the little finger tip was palpable, 石 井 輝 彦1,石 茂 呂 周2

崎 泰 広1,仲

田 雄 一1,小

泉 麻 由 美1,田

津 原 広 行1,松

本 光 彦1,田



博1,小

宮 山 一 雄2,

1 Department of Oral Surgery, Nihon University School of Dentistry 2 Department of Pathology, Nihon University School of Dentistry To whom all correspondence should be addressed: Dr. Hiroshi TANAKA, Department of Oral Surgery, Nihon University School of Dentistry, 1-8-13 Kanda-Surugadai, Chiyoda-Ku, Tokyo 101, JAPAN.

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being mobile and non-tender. No cervical lymphadenopathy was evident. Intraoral examination revealed a well demarcated tumor, which occupied the mucobuccal fold in the 761region. The tumor was hemispherical and its consistency was elastic and soft. Slight tenderness was evident, but there was no abnormal appearance of the mucous membrane in this region (Fig. 2). A test for pulp vitality of 761indicated a positive reaction.

Fig.

Fig.

1

Facial

2

Intra-oral

appearance

view

region

Fig.

3

Pantomograph

of

125

Pantomography and tomoradiography disclosed a radiolucent lesion covering an area from the apices of 76 to the mandibular basis. The lesion was surrounded by an irregular margin, and the inner surface of the cortex at the mandibular basis was resorbed and had an uneven appearance (Figs. 3 and 4). Skeletal and chest X-rays revealed no abnormal findings.

Fig.

Fig.

Fig.

6

4

5

Incisional

Tomograph

CT

scan

biopsy

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specimens

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A CT scan revealed a bone defect with disappearance of the buccal cortex. A periosteal reaction, or so-called Codman's triangle, was noted at the mesial and distal surfaces beside the bone defect (Fig. 5). Laboratory data including serum levels of tumor markers such as CEA, SCC, alpha-fetoprotein and CA 19-9 were within the normal ranges, although ASO and LAP were slightly increased. An incisional biopsy was performed, but this resulted in removal of most of the tumor (Fig. 6). The histopathological diagnosis was EG. The residual lesion was subsequently removed by curettage. At the time of the biopsy, the tumor did not adhere tightly to the periosteum, which remained intact. The tumor was easily detached from the bony wall using a raspatorium. The surface of the bony wall was rough in texture. Histopathological examination showed that the biopsy material was composed of a sheet-like proliferation of histiocytes, which had abundant cytoplasm with clear nuclei containing sparse chromatin (Figs. 7 and 8). The eosinophilic and lymphatic cells formed a granuloma in which multinuclear giant cells were scattered. These cells had invaded the bony structures and destroyed them in the peripheral area of the lesion.

Fig.

Fig.

8

Abundant

7

Sheet-like

cytoplasm

with

proliferation

clear

nuclei

of histiocytes

containing

sparse

chromatin

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Discussion JAFFEand LICHTENSTEIN[1] examined EG of the bone histopathologically, and discussed the interrelationship between EG and Letterer-Siwe disease (LS) and Hand-SchUller-Christian disease (HSC). They concluded that these three diseases were essentially based on the same pathogenesis, and that the difference between them depended on the clinical stage, clinical course, location of the lesion and other factors. From this standpoint. LICHTENSTEIN[2] grouped these three lesions within the broad category of histiocytosis X, and classified them as follows: (1) Histiocytosis X localized in bone (eosinophilic granuloma of bone), (2) Chronic disseminated histiocytosis X (Hand-SchUller-Christian disease), (3) Acute (or subacute) disseminated histiocytosis X (Letterer-Siwe disease). LICHTENSTEIN'S opinion is generally acceptee'41, but several reports15-71have criticized the grouping of these three conditions into a single entity of histiocytosis X, because their pathogeneses are not yet completely resolved. Some authors believe that LS is a distinctly independent entity, even if the identical nature of EG and HSC is accepted[8-10]. EG of the bone is usually divided into 2 types: monostotic and polyostotic. The monostotic type has a higher rate of occurrence, a better prognosis, and occurs in patients older than those affected by the polyostotic type111-141. The case reported here was diagnosed as monostotic-type EG arising in the mandible, because there were no other abnormal X-ray findings in the skeletal system except for the mandible. EG may arise in any bone of the body except for the carpal and tarsal bones[15],although its usual site is the skull, femoral bone, pelvis, rib or vertebral bone[16]. KommA[17] reported that the mandible was affected in only 4 of 128 cases of EG. SLEEPER[15] also reported that cases of EG in the jaw region numbered 12 out of 68. HARTMAN[14] described that about three quarters of all cases of EG in the jawbones arose in the mandible. Histiocytosis X occurs principally in younger individuals and especially with regard to LS, the large majority of cases arise in infants[13].In comparison with LS, the usual age of patients with EG is somewhat higher1131.Of the 17 Japanese cases of EG in the jawbone reported in the field of oral surgery[7,11,12,16,18-28], the average age of the patients was 16.9 years, and 10 of the 17 patients were male. The main clinical symptom of EG arising within the jawbone is prominence accompanied by tenderness[7,11,16,20,26,27]. The symptoms of lesions occurring in or extending into the alveolar bone are loosening or shedding of teeth due to resorption of the alveolar bone, and swelling or ulceration of the gingiva with bleeding or pus discharge. These resemble the symptoms of chronic marginal gingivitis[1,2,7,11,12,16,18-28]. The case reported here arose within the body of the mandible in the 76 region. The only clinical symptom was swelling with slight tenderness. With ragard to X-ray findings, a punch-out defect is the most typical[21].When the lesion advances into the alveolar bone, the affected teeth appear to be "floating"[13]. Unusual cases of cortical rarefaction and cortical defect have been reported by ENDO[16],and a

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case showing periosteal reaction has been reported by NAKAMURAr121. In our present case, a radiolucent defect with an irregular border was observed. It was conspicuous in CT images that the buccal cortical bone corresponding to the area of radiolucency was resorbed, and a periosteal reaction (so-called Codman's triangle) was apparent. It is generally accepted that there are no characteristic laboratory data in cases of EG[16,18].CRP-positive reactions[11,16,19] or increased ESR[16,18]were reported in 4 out of the 17 Japanese cases, and increased alkaline phosphatase[21,23,28] or increased WBC count[16,23,25] was reported in 3 of the 17. However, these laboratory data are not peculiar to EG of the jawbone because the data for the majority of the reported cases were within the normal ranges. Cyst, periodontitis, osteomyelitis, giant cell tumor, fibrous dysplasia of bone, myeloma, malignant lymphoma, osteogenic sarcoma, and metastatic tumor can all be included in the differential diagnosis for EG[16,18].However, many reported cases occurring in the jawbone have been diagnosed preoperatively as bone tumors[12,16,19-24,26,27]. The case reported here was diagnosed preoperatively as osteogenic sarcoma because of the young age of the patient, the short duration of her symptoms, resorption of the buccal cortex and the special periosteal reaction. Surgery or radiation therapy is the treatment of choice for EG. With regard to surgery, simple curettage is adequate[16], because spontaneous healing may sometimes occur[19,25].As to radiation therapy, it is generally accepted that small doses of 1000 to 2000 rad are effective[13], but it has been pointed out that irradiation is better for cases which recur after curettage, as the patients with this condition are usually young children[12,16]. In our present case, most of the lesion was removed at the time of biopsy. However, curettage was subsequently performed for the residual lesion. There has been no evidence of local recurrence for 17 months after surgery. Conclusion A case of EG arising monostotically within the body of the mandible in the 76

region

was

reported.

surgical removal

There

has

been

no

local

recurrence

for

17 months

after

of the lesion. References

[1] JAFFE,H. L. and LICHTENSTEIN, L.: Eosinophilic granuloma of bone. A condition affecting one, several or many bones, but apparently limited to the skeleton, and representing the mildest clinical expression of the peculiar inflammatory histiocytosis also underlying Letterer-Siwe disease and Schuller-Christian disease, Arch. Pathol., 37, 99-118, 1944 [2] LICHTENSTEIN, L.: Integration of eosinophilic granuloma of bone, "Letterer-Siwe disease" and "Schaller-Christian disease" as related manifestations of a single pathologic entity, AMA Arch. Pathol., 56, 84-102, 1953 [3] TSUNEMATSU, Y., KOIDE,R., TAKAHASHI, H., SASAKI, M. and SHIMIZU, K.: Histiocytosis X, Pathology and Clinical Medicine, 2, 35-45, 1984 (in Japanese) [4] WATANABE, S., NAKASHIMA, T., SHIMOSATO, Y., SHIMAMURA, K. and SAKUMA, N.: T-zone histiocytes with S100 protein, development and distribution in human fetuses, Acta Pathol. Jpn., 33, 15-20, 1983 [5] AKAZAKI, K.: Concept of reticulo-endothelial system, Saishin Igaku., 17, 168, 1962 (in

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Japanese) [6] HIRASAWA, K.: Pathological studies of eosinophilic granuloma, Hand-Schaller-Christian disease and Letterer-Siwe disease, with reference to the differences of these diseases, J. Jpn. Reticulo-endothelial System Soc., 13, 163-192, 1973 (in Japanese) [7] SHIMURA, H., MIYATA,G., KOMAGAMINE, M., NAKAJIMA, T., MIYAKE,K., IWAMOTO, K., MIKUNI,S. and IDE, F.: Three cases of eosinophilic granuloma, and an autopsy case of Letterer-Siwe disease, J. Nihon Univ. Sch. Dent., 56, 357-362, 1982, (in Japanese) [8] IIJIMA,S.: On the unusual reticulosis such as Letterer-Siwe disease, Hand-Schaller-Christian disease, mycosis fungoides and reticulosis cutis, Saishin Igaku., 19, 1774-1784, 1964 (in Japanese) [9] NYHOLAM, K.: Eosinophilic xanthomatous granulomatosis and Letterer-Siwe disease, Acta Pathol. Microb. Scand. Section A. Suppl., 216, 21-36, 1971 [10] LIEBERMAN, P. N., JANES,C. R., DARGEON, H. W. K. and BEGG,C. F.: A reappraisal of eosinophilic granuloma of bone, Hand-Schaller-Christian syndrome, Medicine, 45, 375-400, 1969 [11] YANAGISAWA, T., ONOUE,Y., HORIIKE,S., NATSUMI, Y., ISHIKAWA, T., MAEDA,N. and YOSHIOKA, W.: Eosinophilic granuloma of the mandible associated with abnormal chest radiographic findings: Report of a case, Jpn. J. Oral Maxillofac. Surg., 33, 2126-2130,1987 (in Japanese) [12] NAKAMURA, N., TAKENOSHITA, Y., HIGUCHI,Y., NAGANO, T. and OKA, M.: Eosinophilic granuloma of the mandibular condyle; Report of a case, Jpn. J. Oral Maxillofac. Surg., 30, 891-896, 1984. (in Japanese) [13] TAKAHASHI, M. and KAMOI, I.: Roentgenographic diagnosis of histiocytosis X, Jpn. J. Clin. Radiol., 15, 641-654, 1970, (in Japanese) [14] HARTMAN, K. S. and COLONAL, L.: Histiocytosis X: A review of 114 cases with oral involvement, Oral Surg., 49, 38-54, 1980 [15] SLEEPER, E. L.: Eosinophilic granuloma of bone; its relationship to Hand-ShUller-Christian and Letterer-Siwe disease with emphasis upon oral symptoms and findings, Oral Surg. Oral Med. and Oral Pathol., 4, 896-918, 1951 [16] ENDO,M., ISHIKAWA, Y., TAKAHASHI, N., MASUDA, G., SAITO, S., FUJITA,K., MASUDA, M. and OHTANI, T.: Eosinophilic granuloma of bone; Report of three cases, Jpn. J. Oral Maxillofac. Surg., 28, 1777-1787, 1982 (in Japanese) [17] KOJIMA, M. and TAKAHASHI, S.: Congenital Diseases of Reticuloendothelial System, Bunkodo, Tokyo, 561-684, 1974 (in Japanese) [18] KUROKAWA, H., AKAMA, I., TSURU, S., TANAKA, T., YANO,S., ETO,H., OHTSUKA, Y., MAKI,S. and KAJIYAMA, M.: Eosinophilic granuloma of the maxilla: Report of a case, Jpn. J. Oral Maxillofac. Surg., 35, 585-590, 1987 (in Japanese) [19] UCHIYAMA, T., SATO,T., HORIKAWA, H., SHIGEMATSU, S., KITAMURA, N., TAKANO,N., TSURUKI, T., SHIMONO, M. and TANAKA, Y.: A long-term follow-up report of eosinophilic granuloma of the mandible, Jpn. J. Oral Maxillofac. Surg., 35, 693-699, 1989(in Japanese) [20] OKUTOMI, T., WATANABE, F., HATANO,Y., OKUDA,T., NISHIWAKI, T., ITO, M., ITO, A., TATEMATSU, N. and OKA,N.: Eosinophilic granuloma of the mandible: Report of a case and findings of the electron microscope, Jpn. J. Oral Maxillofac. Surg., 34, 366-370, 1988 (in Japanese) [21] HAYATSU, Y., ADACHI, H., FUKUDA, O., FURUTA, I. and KOHAMA, G.: Eosinophilic granuloma of the mandible: Report of two cases, Jpn. J. Oral Maxillofac. Surg., 28, 1254-1259,1982, (in Japanese) [22] HISHIDA, I., MIMURA, T., OHEDA,N., WATATANI, K., YAKUSHIJI, N., SAITO, Y. and MIYAZAKI, T.: Eosinophilic granuloma of the maxilla: Report of a case, Jpn. J. Oral Maxillofac. Surg., 28, 1173-1179, 1982 (in Japanese) [231 SAKAMOTO, H., ASAKURA, A., OTOZURA, N., SUZUKI, K., HAYASHI, K. and KAMIYAMA, T.: Histiocytosis X (eosinophilic granuloma of bone) of the mandible in a 9-month infant girl: Report of a case, Jpn. J. Oral Maxillofac. Surg., 28, 143-148, 1982 (in Japanese) [24] ASADA,K., MASUO, K., NAKAI,Y., SHIMIZU, M., HORIUCHI, J., SUNAGAWA, H., MICHI,K. and

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[25] [26] [27] [28]

UENO,T.: Eosinophilic granuloma of the mandibular condyle: Report of a case, Jpn. J. Oral Maxillofac. Surg., 23, 689-691, 1977 (in Japanese) KITAYAMA, S., NAGANO, H., HAGA,Y., YAMAMOTO, T. and EGUCHI,T.: A case report of suspected eosinophilic granuloma on the maxilla and mandible, Jpn. J. Oral Maxillofac. Surg., 17, 147-151, 1971 (in Japanese) ADACHI,K., SHIMADA, K., NAKANISHI, K., TERANOBU, 0., NAKAO,K., ISHIDA,T. and SHIGETA, Y.: A case of histiocytosis X and review of the literature, J. Jpn. Stomatol. Soc., 29, 134-143, 1980 (in Japanese) SHIMOSATO, T., SUGIMURA, M., HIROSE, I., NOBEFUJI, N. and HASEGAWA, K.: Histiocytosis X with main symptoms in the mandible: Report of two cases, J. Jpn. Stomatol. Soc., 19, 924-934, 1970 (in Japanese) INouE, Y., KAMINO,T., MIZUNO,H., SHIBATA, M., KAMIYA, H., ISHIKAWA, K., YANO,S., YOKOI,M., ISHIGURO, H. and KONDO, N.: Eosinophilic granuloma of the mandible: Report of a case, J. Jpn. Stomatol. Soc., 28, 477-482, 1979 (in Japanese)

A case of eosinophilic granuloma arising in the mandible.

Eosinophilic granuloma is a disease of the reticuloendothelial system and its pathogenesis is not yet completely clear. The histopathological findings...
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