å¡ CASE REPORT å¡

Eosinophilic Pneumonia with Eosinophilic Gastroenteritis Takahito Miyazono, Masaharu Kawabata, Ikkou Higashimoto, Yoshifusa Koreeda, Yuuji Iwakiri, Kimiyoshi Arimura and Mitsuhiro Osame

A 48-year-old man was admitted to our hospital with cough, fever and dysphagia. He had a past history of bronchial asthma and surgery for nasal polyp. Chest radiograph and computed tomography showed atelectasis in the right lower field and infiltrative shadow in the left lower field and overall thickening of the esophageal wall. Transbronchial lung biopsy (TBLB) speci- mensrevealed infiltration of eosinophils and lymphocytes under the bronchial mucosa. Gastrointes- tinal tract biopsy specimens showed submucosal infiltration of eosinophils. These findings led to a definite diagnosis of associated with eosinophilic gastroenteritis, a dis- ease whichhas been rarely reported. (Internal Medicine 38: 450-453, 1999) Key words: hypereosinophilic syndrome (HES), transbronchial lung biopsy (TBLB), esophageal wall

Introduction findings. Wheezeon expiration wasaudible in the wholelung field with fine crackles in the right lower field. Abdomenand Eosinophils play an important role in hypereosinophilic syn- extremities had no abnormal findings. Clubbedfinger was not drome(HES)as well as in other allergic diseases such as asthma, found. Neurological examination on admission revealed a de- parasite infection and atopic dermatitis ( 1-3). Wereport a case crease in olfactory sensation. Other sensations and deep re- of eosinophilic pneumonia and eosinophilic gastroenteritis in flexes were normal. Blood laboratory tests were performed on a patient with a history of bronchial asthma, and discuss the admission (Table 1 ). Neutrophilia and were present pertinent literature (4, 5). with elevated immunoglobulin E (IgE) and C-reactive protein (CRP). C-Antineutrophil cytoplasmic antibody (C-ANCA) was Case Report negative. Respiratory function test suggested obstructive ven- tilatory impairment with forced expiratory volume in one sec- A 48-year-old man was admitted to Kagoshima University ond of 1,330 ml and forced expiratory volume%in one second Hospital on December 18, 1995, because of cough and dysph- of 38.78% (<70%). An arterial blood gas analysis showed hy- agia. He began to complain of transient dysphagia and poxemia with the partial pressure of oxygen (PaO2) was 65.6 epigastralgia while swallowing solids in August in 1995. Due Torr in room air. Chest radiograph and chest CT showed to fever and cough on November 23, he visited a doctor. Since atelectasis in the right lower field and an infiltrative shadow in pneumoniawassuspected, he was admitted to a local hospital the left lower field (right S 9, 10). Chest CT demonstrated over- and treated with antibiotics for relief of symptoms. Because all thickening ofesophageal wall ( Fig. 1). Transbronchial lung findings did not improve on chest radiograph and chest com- biopsy (TBLB) specimens from B7 in the right lung under puted tomography (CT), he was referred to our department for fiberoptic bronchoscopy showed infiltration of eosinophils and further examination and was admitted on December18, 1995. lymphocytes without granuloma under the bronchial mucosal He suffered from bronchial asthma at age 14 and underwent after hematoxylin and eosin staining (Fig.2A). Biopsy speci- surgery for nasal polyp at age 42. His father had asthma. There mens of stomach and duodenumshowed submucosal infiltra- was a history of allergy to penicillin and aspirin-like drugs. He tion of eosinophils (Fig. 2B). Although scleroderma was con- had no smoking history. His height was 163 cm, his weight sidered as a differential diagnosis for digestive and respiratory was 74 kg, blood pressure was 132/84 mmHg,pulse was 70 symptoms with an elevated scleroderma antibody 70 (Scl-70) beats per minute (regular). His face and neck had no abnormal on admission, the above findings led to a definite diagnosis of

From the Third Department of Internal Medicine Faculty of Medicine, Kagoshima University, Kagoshima Received for publication February 12, 1998; Accepted for publication January 25, 1 999 Reprint requests should be addressed to Dr. Masaharu Kawabata, the Third Department of Internal Medicine Faculty of Medicine, Kagoshima University, 8-35- 1 Sakuragaoka, Kagoshima 890-8520

450 Internal Medicine Vol. 38, No. 5 (May 1999) Eosinophilic Pneumonia Table 1. Laboratory Findings on Admission

W B C 1 2 ,00 0/u l T o ta l p ro te in 8. 5 g /dl N e u 6 4 % a lb u m in 4 7. 1 % E o 6 % A /G 0 .8 9 B a so 1 % M o n o 7 % I g E ( R I S T ) 4 ,0 8 7 I U/ / L y m p h 2 2 % R B C 4 9 3 x l O V u l Ig G ,9 0 0 IU // H b 1 4.8 g/ dl Ig A 4 6 1 TU /Z H t 4 6 . 1 % Ig M 1 3 IU // Pl atelet 3 5 x l O V m l C 3 1 8 6 m g / d l A S T 20 I U /1 C 4 4 1 . 4 m g / d l A L T 22 I U /i C -A N C A < 10 E U y -G T P 47 T U /l L D H 4 2 2 I U // C R P 53 . 4 mg / d l B U N 1 4. 2 mg / d l H T L V - 1 (- ) C r 0 . 9 m g / dl S c l - 7 0 An t i b o d y 1 3. 1 In d e x U A 5 . 9 m g / dl N a 1 4 3 m E q / d l P u l m o n a r y F un c t i o n T e s t K 4. 0 m E q / d l v c 3 , 3 9 0 m l F B S 8 4 m g/ d l % v c 9 3 .4 T -C H O 1 7 1 m g / d l F E V l. 0 , 3 30 m l F R V l. 0 % 3 8 .7 8 % B l o o d G a s A n a l y s i s ( ro om ai r) p H 7 . 4 4 0 P a C O , 37 .5 To r r P a O 9 6 5 . 7 T o n H C O f 2 5 . 4 m m ol / /

WBC:white blood cell, Neu: neutrophil, Eo: eosinophil, Baso: basophil, Mono: monocyte, Lymph: lymphocyte, RBC:red blood cell, Hb: hemoglobin, Ht: hematocrit, AST: aspartate aminotrans- ferase, ALT: alanine aminotransferase, y-GTP: y-glutamyl transpeptidase, LDH:lactate dehy- drogenase, BUN:blood urea nitrogen, Cr: creatinine, UA: uric acid, FBS: fasting blood sugar, T- CHO:total cholesterol, PaCO2: partial pressure of carbon dioxide, PaO2: partial pressure of oxy- gen, A/G: albumin-globulin ratio, Ig: immunoglobulin, C-ANCA:circulating antineutrophil cy- toplasmic antibodies, CRP: C-reactive protein, HTLV-1: human T cell lymphotropic type 1 , VC: vital capacity, FEV1.0: forced expiratory volume in one second.

Figure 1. Chest radiograph and CTon admission. Chest radiograph showed atelecta- sis in the right lower field and infiltrative shadow in the left lower field. Chest CTshowed overall thickening of the esophageal wall. 451 Internal Medicine Vol. 38, No. 5 (May 1999) Miyazono et al

eosinophilic pneumonia associated with eosinophilic gastro- enteritis. Relief from symptomsand a decrease in CRPwere noted after the treatment with corticosteroids started on Janu- ary 20, 1996. Chest CT on March 14 showed improvement in atelectasis and esophageal wall thickening (Fig. 3). Discussion Table 2 shows a that are associated with pe- ripheral eosinophilia (3, 6). Concerning drug allergies, the pa- tient exhibited allergies to penicillins and aspirin-like drugs. However, he had not taken any drug at that time. Regarding malignancy, systemic radiological and hematological exami- nations revealed negative findings. Weexamined stool and he- matological findings to identify parasites but the results were negative. Theclinical symptomsof dysphagia and epigastralgia and elevated SCL-70 indicated scleroderma, but this case did not satisfy sufficient diagnostic criteria for collagen diseases. The patient demonstrated bronchial asthma at age 14. On ad- mission, hecomplained of cough, and wheeze was audible. His eosinophilia may have been induced by allergic diseases in- cluding bronchial asthma. Eosinophilic gastroenteritis is char- acterized by peripheral eosinophilia and infiltration of eosino- phils in the whole digestive tract, especially in the stomach,

Figure 2. A. Lung biopsy specimen (rt. B7). Infiltration of eosinophils (arrow) and lymphocytes was shown under the brochial mucosal surface. (Hematoxylin and Eosin staining, x 400 ) B. Stomach biopsy specimen. Submucosal infiltration of eosinophils was found (arrows) (HE stain, x 200).

Figure 3. Pre-therapy and Post-therapy chest radiograph and CT. Post- therapy chest radiograph and CTshowed improvement in atelectasis and esophagial wall thickening, compared with pre-therapy.

452 Internal Medicine Vol. 38, No. 5 (May 1999) Eosinophilic Pneumonia

Table 2. Diseases Associated with Eosinophilia Table 3. The Criteria for Hypereosinophilic Syndrome (HES) 1 Allergic diseases (Bronchial asthma, urticaria etc.) 1) A persistent eosinophilia of 1,500 eosinophils/mm3 for longer than 2 Drug allergies 6 months, or death before 6 months associated with the signs and 3 Malignancies (Eosinophilic leukemia, Hodgkin's disease etc.) symptomsof hypereosinophilic disease. 4 Dermatic diseases 2) A lack of evidence for parasitic, allergic or other knowncauses of 5 Parasites eosinophilia. 6 Collagen diseases duodenumand small intestine. This case was consistent with severe nor long-term peripheral eosinophilia was observed. eosinophilic gastroenteritis. Onthe other hand, patients with Weconsidered that this case did not satisfy the HEScriteria eosinophilic pneumoniahave symptomsof cough, sputa and and diagnosed this patient as having eosinophilic pneumonia fever, findings of infiltration on plain chest radiography and with eosinophilic gastroenteritis. CT, and eosinophilia on the blood cell count (6). The findings of this case also indicated eosinophilic pneumonia. The asso- References ciation of between eosinophilic pneumoniaand eosinophilic gastroenteritis in multiple organs, as found in the present case, 1) Hardy WR,Anderson RE. The hypereosinophilic syndromes. Ann Intern Med68: 1220-1229,1968. has rarely been reported. Wefound only 1 case report by 2) Chusid MJ, Dale DC, West BC, Wolff SM. The hypereosinophilic syn- Marnocha et al in English (4) and another case reported by drome: Analysis of fourteen cases with review of the literature. Medicine Tanoue which was presented at a local meeting in Japan (5). (Baltimore) 54: 1-27, 1975. Weexamined the possibility that the condition of simultaneous 3) Okubo Y, Sekiguchi M. Hypereosinophilic Syndrome (HES). in : infiltration of eosinophils in multiple organs is classified into Kousankyu (Eosinophil), Kokusai Igaku Shuppan, Tokyo: 222-235, 1991 the category of HES. (in Japanese). 4) Marnocha KE, Maglinte DD, Kelvin FM, McCune M, Weiser DC, Strate HESis defined as a condition where peripheral eosinophilia R. Eosinophilic Enteritis Associated with Chronic Eosinophilic Pneumo- persists for more than 6 months or a patient with symptoms of nia. Am J Gastroenterol 81: 1205-1208, 1986. eosinophilia whodies within 6 months without any other re- 5) Tanoue M. A case report of bronchial asthma patient complicated with sponsible disease associated with eosinophilia such as para- eosinophilic gastroenteritis and eosinophilic pneumonia. NipponNaika sitic infection or allergy (1-3). Gakkai Kantou Chihoukai 4: 1 17, 1993 (Abstract in Japanese). 6) Chihara J, Eosinophilic pneumonia, in: Kousankyu (Eosinophil), Kokusai The present case indicated eosinophilic infiltration to mul- Igaku Shuppan, Tokyo: 236-249, 1991 (in Japanese). tiple organs such as the lung and digestive organs, but neither

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