―Case Reports― A Case of Metagonimiasis Complicated with Multiple Intracerebral Hemorrhages and Diabetes Mellitus Shoko Merrit Yamada1, Shokei Yamada1, Hitomi Takada2, Yayoi Carol Hoshiai3 and Shotetsu Yamada4 1 Department of Neurosurgery, Yamada Hospital, Shizuoka 2 Department of Internal Medicine, Yamada Hospital, Shizuoka 3 Department of Ophthalmology, Yamada Hospital, Shizuoka 4 Department of General Surgery, Yamada Hospital, Shizuoka Abstract Metagonimiasis yokogawai is an unusual intestinal parasitic disease caused by metacercariae of Metagonimus yokogawai. The first clinical manifestations of this disease do not always correlate with gastrointestinal signs. A 61-year-old man with left hemiparesis and disorientation was admitted to our hospital because of atypical nonhypertensive multiple intracerebral hemorrhages, which were conservatively treated. The patient was discharged from our hospital after 2 months without any neurological deficits; however, he was readmitted owing to a body temperature higher than 38℃ for nearly 2 weeks. Examination of stool revealed eggs of M. yokogawai, and the body temperature returned to normal after administration of praziquantel. Furthermore, the control of the patientʼs diabetes mellitus (DM) markedly improved after the treatment, although the patient had had DM for more than 2 years. We conclude that DM is a chronic sign of metagonimiasis in carriers and that intracerebral hemorrhage might be an acute sign in the aggravated phase of the disease. (J Nippon Med Sch 2008; 75: 32―35) Key words: diabetes mellitus, intracerebral hemorrhage, metagonimiasis, parasite, trematode metagonimiasis has also been increasing since 1990 Introduction in Tokyo1. Most patients with metagonimiasis are asymptomatic carriers, although many M. yokogawai Metagonimiasis yokogawai is an unusual parasitic eggs are detected in the stool3, and the infection in disease caused by metacercariae of Metagonimus some of these patients may resolve spontaneously4. yokogawai, an intestinal trematode that infects people Here consuming freshwater fish such as ayu (Plecoglossus yokogawai that was extremely difficult to diagnose altivelis), ugui (Tribolodon hakonensis), and shirauo because the clinical manifestations included multiple 1,2 we present a case of metagonimiasis (Salangichthys microdon) . With the increase in the intracerebral hemorrhages (ICHs) without intestinal popularity of these fish, the number of cases of symptoms. Correspondence to Shoko Merrit Yamada, MD, Department of Neurosurgery, Yamada Hospital, 6―24 Hamada-cho, Shimizu-ku, Shizuoka 424―0834, Japan E-mail: merrityamada@hotmail.co.jp Journal Website (http:! ! www.nms.ac.jp! jnms! ) 32 J Nippon Med Sch 2008; 75 (1) Metagonimiasis with ICH and DM gastrointestinal symptoms were present; however, eggs of M. yokogawai were detected in feces (Fig. 3). Case Presentation The patient received a single 1,200 mg oral dose of was praziquantel, after which the body temperature did admitted to our hospital because of sudden onset of not increase to more than 37℃ and all laboratory disorientation and left hemiparesis (MMT: U! E=4! 5, data normalized (Fig. 2A and 2B). At the time of L! E=4 ! 5). His conscious level was 14 (E4V4M6) on discharge, the body temperature was 36.2℃; the A 61-year-old man residing in Tokyo + the Glasgow coma scale, and he responded slowly to WBC count was 7,400! µL with 3.8% eosinophils; and our questions. Computed tomography (CT) revealed the CRP level was 0.2 mg! dL. Furthermore, the multiple ICHs in the right thalamus and the right fasting blood glucose level decreased to less than 100 frontal lobe (Fig. 1A), and both of the hematomas mg! dL with a daily calorie intake of 2,200 kcal (Fig. were therapy. 2C). Glibenclimide was discontinued, and HbA1c had Angiography showed no vascular abnormalities, decreased to 5.7% 2 months after the second such admission. small as enough tumor for conservative staining or arteriovenous The patient was discharged after malformation (Fig. 1B). He had no history of eradication of M. yokogawai had been confirmed by hypertension, and his blood pressure was 132! 84 the absence of eggs on repeated stool examinations. mmHg on admission. This patient had 2-year history Discussion of diabetes mellitus (DM) and had been receiving 2.5 mg of glibenclimide per day. The fasting blood glucose level was 229 mg! dL, and hemoglobin A1c The most common primary manifestations of (HbA1c) level was high at 10.1%. Ophthalmologic metagonimiasis yokogawai include abdominal pain, examination revealed mild proliferating retinopathy, diarrhea, and loss of appetite2; however, most suggesting retinal changes due to prolonged DM patients are asymptomatic even though many M. (Fig. 1C). The patientʼs daily caloric intake was yokogawai eggs are detected in stool4. Moreover, it is restricted to 1,600 kcal, and physical rehabilitation reported that more than half of the asymptomatic was started within 1 week after admission. On patients are naturally cured without medical 8 admission, body temperature was 37.6℃; the white treatment . We considered retrospectively that the blood cell (WBC) count was 11,000! µL (8.2% of WBCs deteriorated conditions of our patient caused by were eosinophils); and the C-reactive protein (CRP) metagonimiasis yokogawai might improve naturally level was 2.5 mg! dL. As shown in Figure 2, the with transfusion and bed rest during the first body temperature and laboratory data (WBC count admission. Patients infected with M. yokogawai and during generally show 2 types of symptoms: chronic and hospitalization. After 2 months, the patientʼs level of acute. Chronic symptoms include general fatigue, alertness fever, and weight loss8, whereas acute symptoms CRP) fluctuated improved, but and normalized the left hemiparesis recovered completely. Although a definite conclusion include abdominal pain, diarrhea2, and pleural 4 could not be made regarding the cause of the ICHs, effusion . In our patient, DM might have been a the patient was discharged from our hospital with a chronic symptom and ICH an acute symptom. normal body temperature and normal WBC count Metagonimiasis cannot be diagnosed without stool (Fig. 2A and 2B). examination. Some patients who exhibit only chronic After 2 weeks, the patient was readmitted to our symptoms may be misdiagnosed to have common hospital with a body temperature higher than 38℃ cold or another disease; however, the diagnosis is that On also difficult in patients with acute symptoms if they readmission, the body temperature was 38℃ (Fig. are not associated with gastrointestinal findings. 2A), and laboratory data suggested severe infectious Metagonimiasis yokogawai symptoms vary from disease with eosinophilia (WBC count, 10,200! µL patient to patient; hence, our patient did not with 10% eosinophils; CRP, 12.5 mg! dL; Fig. 2B). No demonstrate any signs of ICH during the second had persisted for nearly J Nippon Med Sch 2008; 75 (1) 2 weeks. 33 S.M. Yamada, et al A. BT (℃) 39 BT 38 37 36 35 B 0 20 40 1st admission 60 80 100 120 (days) 2nd admission 2nd discharge 1st discharge B. WBC & CRP WBC (/μL) 15 15 10 10 5 5 0 Fi g.1 A)Pl a i n CT s c a no na dmi s s i o n:Mul t i pl e I CHswe r ei de nt i f i e di nt her i ghtt ha l a mus a ll o be . A mi l dma s se f f e c t a ndt her i ghtf r o nt wa so bs e r ve da r o und bo t h he ma t o ma s .B) Angi o gr a phy:No va s c ul a ra bno r ma l i t i e s , sa r t e r i o ve no us ma l f o r ma t i o na nd s uc ha t umo rs t a i ni ng, we r eo bs e r ve di nt hi ss t udy. r a t e C) Opt i cf undus e xa mi na t i o n: Mo de a r t e r i o s c l e r o s i swa so bs e r ve di n bo t ho pt i c f undi . Mi l d ne o va s c ul a r i z a t i o n wa sa l s o t es po t t yl e s i o nsi n bo t h de t e c t e d wi t h whi r e t i na s ,c o ns i s t e ntwi t h mi l d pr o l i f e r a t i ng r e t i no pa t hy. Fi g.3 St o o le xa mi na t i o nr e ve a l sa n M.y o k o g a wa i e ggi nt hepa t i e nt ' sf e c e s . 34 CRP (mg/dL) WBC (×1000) CRP 0 20 40 60 80 0 120 (days) 100 C. FBG & HbA1c (mg/dL) 300 (10.1) (8.7) 200 (8.3) FRG HbA1c (8.4) (6.1) 100 glibenclamide 2.5mg 0 0 20 1800kcal/day 40 1.25mg off 60 80 100 120 (days) 2200kcal/day Fi g.2 Cha nge si nt hel a bo r a t o r yda t ao ft hepa t i e nt dur i ng ho s pi t a l i z a t i o n.A)Cha nge si n bo dy ur e : Thepa t i e ntwa sdi s c ha r ge d6 0 t e mpe r a t da ysa f t e rt hef i r s ta dmi s s i o nwi t hano r ma l e ks a t bo dy t e mpe r a t ur ea nd s pe nt2 we ho me ; he wa st he n r e a dmi t t e d t oo ur ho s pi t a lwi t ha ne l e va t e dbo dyt e mpe r a t ur e . o unta ndCRPl e ve l : B)Cha nge si nt heWBCc TheWBC c o unta nd CRP l e ve lf l uc t ua t e d gr e a t l ydur i ngt hef i r s ta dmi s s i o n.Ho we ve r , l ue so fbo t hva r i a bl e sno r ma l i z e da f t e r t heva t het r e a t me ntdur i ngt hes e c o nda dmi s s i o n. et o l e r a nc ea ndde c r e a s e d C) I mpr o ve dgl uc o s he mo gl o bi n A1 c ( HbA1 c ) l e ve l : Af t e r t r e a t me ntwi t h pr a z i qua nt e l ,f a s t i ng bl o o d gl uc o s e de c r e a s e d ma r ke dl yt o7 2 mg/ dL whe nt heda i l yc a l o r i ci nt a kewa sr e duc e dt o a l o r i ci nt a kewa st he n 1 , 8 0 0kc a l ;t heda i l yc i nc r e a s e dt o2 , 2 0 0kc a l . J Nippon Med Sch 2008; 75 (1) Metagonimiasis with ICH and DM admission despite the worsening of his general should be considered in patients with chronic conditions and laboratory data. metabolic diseases or atypical ICH. We did not consider parasitic disease in our patient when CT revealed multiple ICHs. Despite References the presence of fever and abnormal laboratory data, we first thought that this patient with DM had cerebrovascular disease. Hypertension is the main risk factor for ICH, and DM is a risk factor for intracerebral ischemia but not always for ICH9,10. The causes of nonhypertensive ICH include cerebral amyloid angiopathy, arterial or venous angiomas, brain tumors, anticoagulant medication, and bleeding tendency, such as leukemia, thrombocytopenia, and hemophilia. Although infectious diseases rarely cause ICH, they should not be ruled out. Some authors have reported 5 ICH caused 6 by 7 schistosomiasis , paragonimiasis , or sparganosis . We concluded that in our patient, ICH was associated with metagonimiasis although a definitive diagnosis of cerebral metagonimiasis was not made. DM, a chronic metabolic disease, may be caused by intestinal parasitic diseases. Without aggravation of the disease, the clinical process of DM is similar to that of non-insulin-dependent diabetes. In our patient, optic fundus examination revealed mild proliferating retinopathy (Fig. 1B), which is a typical retinal change due to chronic diabetes. However, DM resolved several months after the complete resolution of metagonimiasis, and restriction of daily caloric intake was not required after treatment. 1.Suzuki J, Murata R, Morozumi S, et al.: Investigation of Metagonimus yokogawai metacercariae infection in Salangichthys microdon (Shirauo) retailed in Tokyo. Shokuhin Eiseigaku Zassi 2000; 41: 353―356. (in Japanese) 2.Ichiki Y, Tanaka T, Haraguchi Y, Tanaka K, Nawa Y: A case of severe metagonimiasis with abdominal symptoms. 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