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Pulmonary dirofilariasis in a 59-year-old man

Pulmonary dirofilariasis in a 59-year-old man JSCR 2014;8 ( 3 pages) doi:10.1093/jscr/rju082 Case Report Pulmonary dirofilariasis in a 59-year-old man 1,* 2 2 3 3 4 Tetsuya So , Reiko Mitsueda , Takeaki Miyata , Atsushi Sekimura , Takashi Yoshimatsu , Naohiro Nose , 5 5 1 Hiroshi Itoh , Shigeo Nakano and Kosei Yasumoto 1 2 Department of Thoracic Surgery, Shinkomonji Hospital, Kitakyushu 800-0057, Japan, Department of Thoracic Surgery, Fukuoka-Wajiro Hospital, Fukuoka 811-0213, Japan, Department of Thoracic Surgery, Shintakeo Hospital, Takeo City, Saga 843-0024, Japan, Department of Thoracic Surgery, Shinyukuhashi Hospital, Yukuhashi City, Fukuoka 824-0026, Japan and Department of Pathology, Shinkomonji Hospital, Kitakyushu 800-0057, Japan *Correspondence address. Tetsuya So, Department of Thoracic Surgery, Shinkomonji Hospital, 2-5 Dairishinmachi, Moji-ku, Kitakyushu 800-0057, Japan. Tel: þ81-93-391-1001; Fax: þ81-93-391-7001; E-mail: tetsuya@shinkomonji-hp.jp Received 11 July 2014; revised 29 July 2014; accepted 2 August 2014 We present a case of a human pulmonary dirofilariasis in a 59-year-old man. At the medical examination, a chest computed tomography (CT) revealed a mass, measuring 18  15 mm in diameter, with an irregular margin on the bottom of the right lower lobe. We could not neglect the possibility of a primary lung cancer, and therefore, a lung partial resection was performed under video-assisted thoracoscopic surgery. The intra-operative pathological findings revealed inflammatory granuloma with coagulation necrosis and no malignant cells. The permanent pathological examination showed occlusion of the peripheral pulmonary artery by worms and formation of a necrotic mass surrounded by reactive inflammation and hemorrhage. Human pul- monary dirofilariasis is an extremely rare zoonotic infection, and sometimes it is difficult to dis- tinguish it from a primary lung cancer on radiographic findings. INTRODUCTION were all within their normal ranges. In May 2013, a lung partial resection was performed under video-assisted thoracic surgery Human pulmonary dirofilariasis is a rare zoonotic infection (VATS). At the time of thoracotomy, the mass located at the per- caused by the dog heartworm Dirofilaria immitis, which is trans- iphery of S9 of the right lower lobe and the surface was whitely mitted via a vector/intermediate host, generally the mosquito transparent (Fig. 2A). The mass was solid and hard on palpation [1, 2]. Owing to the increase in the number of a medical examin- (Fig. 2B). The intra-operative pathological findings (frozen ation, this disease has sometimes been reported recently. We section) revealed inflammatory granuloma with coagulation ne- herein report the rare case of a human pulmonary dirofilariasis. crosis. The permanent pathological examination showed occlu- sion of the peripheral pulmonary artery by worms and CASE REPORT formation of a necrotic mass surrounded by reactive inflamma- tory cells and hemorrhage in hematoxylin–eosin stain (Fig. 3A) A 59-year-old Japanese man visited our hospital in April 2013 and Elastica van Gieson stain (Fig. 3B). Post-operative course for a workplace medical examination. He was a former smoker was uneventful, and he is doing well 15 months after surgery. (from 20 to 26-year-old, 1 pack per day), and had neither phys- ical symptoms nor family history of malignancies. The chest X-ray did not detect an abnormal shadow, but a CT showed a mass with an irregular margin measuring 18  15 mm in diam- DISCUSSION eter at the subpleural area of S of the right lower lobe (Fig. 1). The CT detected no nodules in the other lobes or regional Human pulmonary dirofilariasis is a rare zoonotic infection lymph nodes swelling. A FDG-PET was performed, but did not caused by the canine heartworm Dirofilaria immitis [1, 2]. yield a definitive FDG accumulation (data not shown). Such According to the medical database, De Magelhaes first tumor markers as CEA, SLX, SCC, CYFRA, NSE, and ProGRP described the discovery of filarial worm in the left ventricle of Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved.# The Author 2014. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http:// creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Page 2 of 3 T. So et al. Figure 1: Pre-operative chest CT scan demonstrated the mass measuring 18  15 mm with spiculation at the periphery of S9 of the right lower lobe. Figure 3: (A) Histopathological findings of the pulmonary nodule. Two trans- verse sections of an immature worm were seen in the peripheral pulmonary with fibrous tissue, necrotic tissue, and granulation (hematoxylin and eosin staining, 6). (B) The transverse section in the peripheral pulmonary revealed layers of cuticle, internal longitudinal cuticular ridges and lateral chords, being identified as Dirofilaria immitis (Elastica van Gieson stain, 20). a Brazilian boy in 1887 [3]. Flieder and Moran [4] investi- gated that there were 31 of 41 nodules in the right side of the lung, and especially 19 of 31 nodules in the right lower lobe. In our case, he was asymptomatic and the mass was located in the right lower lobe. Furthermore, he bred a dog 20 years ago in our case. Milanez et al. [5] mentioned that the filarial infection rates of dogs were more important than the history of dog breeding. The radiographic images of human pulmonary dirofilariasis have some specific features. It has been reported that the chest X-ray showed a solitary, round nodule in a lung field in many cases. In our case, however, the chest X-ray did not reveal the mass because of overlapping of the diaphragm. A chest CT can detect the disease as a mass measuring 18  15 mm in diameter at the periphery of the right lower lobe close to the lateral pleura. Recently, a FDG-PET has been applying for the qualitative diagnosis of lung nodules. The FDG-PET had been described in three cases of human pulmonary dirofi- Figure 2: (A) Intra-operative thoracoscopy showed the white tumor with pleural lariasis for the differential diagnosis of pulmonary nodule [6]. indentation. (B) Macroscopic examination revealed white and solid tumor. Human pulmonary dirofilariasis Page 3 of 3 The standardized uptake value (SUV) of them was reported ACKNOWLEDGEMENTS from negative to 7.5, and the SUV was 1.4 in our case. In this We thank Dr Tamotsu Kanazawa of University of Environmental way, the FDG-PET cannot deny a primary lung cancer pre- and Occupational Health, Japan, for critical comments. operatively. Human pulmonary dirofilariasis is usually diagnosed by REFERENCES the surgical specimens, and it seems to be difficult to diag- nose this disease by using the other modalities. For example, 1. Shibasaki M, Shito I, Konno A, Yusa T, Shibuya K, Kubosawa H, et al. A case of pulmonary diroflariasis: cytological, pathological and a filarial infection in human causes eosinophilia in general. immunological diagnosis-(in Japanese with English abstract). J Jpn Soc Yoshimura et al. mentioned that human pulmonary dirofilar- Clin Cytol 1997;36:174–8. iasis had little specific findings in hematological analysis 2. Asimacopoulos PJ, Katras A, Christie B. Pulmonary dirofilariasis. The largest single-hospital experience. Chest 1992;102:851–5. and the average of eosinocytes in the peripheral blood was 3. Sakurai A, Gohara H, Tajiri N, Ando Y, Maruyama S, Yokoyama S, et al. 6.5% [7]. In our case, the eosinocytes in the peripheral Human pulmonary dirofilariasis; a case report and a review of 117 cases in blood was 1.3 and 1.9% pre-operatively. The other examin- Japan (in Japanese with English abstract). Jpn J Clin Radiol 2006;51:169–73. 4. Flieder DB, Moran CA. Pulmonary dirofilariasis: a clinicopathologic ation to detect dirofilaria-specific antibody, such as an study of 41 lesions in 39 patients. Hum Pathol 1999;30:251–6. enzyme-linked immunosorbent assay, immunological elec- 5. Milanez de Campos JR, Barbas CS, Filomeno LT, Fernandez A, trophoresis and agglutination of red blood cells, has been Minamoto H, Filho JV, et al. Human pulmonary dirofilariasis: analysis of 24 cases from Sao Paulo, Brazil. Chest 1997;112:729–33. reported to be useful [1, 7 – 9]. These immuno-serological 6. Shimokawa H, Hanagiri T, Takenoyama M, Yamada S, Kanazawa T, analyses, however, have some problem as cross-reactivity Yasumoto K. A case of pulmonary dirofilariasis with cavity formation and non-specificity between Dirofilaria immitis and the (in Japanese with English abstract). J Jpn Assoc Chest Surg 2011;25:21–4. 7. Sato M, Koyama A, Iwai K, Kawabata Y, Kojima S. Human pulmonary other filariasis. As a treatment, a lung resection has been per- dirofilariasis with special reference to the ELISA for the diagnosis and formed which leads to final diagnosis and cure. Recently, a follow-up study. Z parasitenkd 1985;71:561–3. VATS seems to be a mainstream, and the benefit is less inva- 8. Mori S, Yasunobu H, Mizoguchi A, Kawabata M, Nakamura F, Nawa Y, et al. Pulmonary dirofilariasis with serologic study on familial infection sive for the patient [6, 10]. with Dirofilaria immitis. Intern Med 2004;43:327–30. In conclusion, human pulmonary dirofilariasis is an ex- 9. Kunst H, Mack D, Kon OM, Banerjee AK, Chidini P, Grant A. Parasitic tremely rare zoonotic infection, and sometimes mimick a infections of the lung: a guide for the respiratory physician. Thorax 2011;66:528–36. primary lung cancer on radiographic findings. We, however, 10. Miyoshi T, Tsubouchi H, Iwasaki A, Shiraishi T, Nabeshima K, should be able to list up the one of differential diagnosis of Shirakusa T. Human pulmonary dirofilariasis: a case report and review lung cancer. of the recent Japanese literature. Respirology 2006;11:343–7. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Pulmonary dirofilariasis in a 59-year-old man

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Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author 2014.
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Abstract

JSCR 2014;8 ( 3 pages) doi:10.1093/jscr/rju082 Case Report Pulmonary dirofilariasis in a 59-year-old man 1,* 2 2 3 3 4 Tetsuya So , Reiko Mitsueda , Takeaki Miyata , Atsushi Sekimura , Takashi Yoshimatsu , Naohiro Nose , 5 5 1 Hiroshi Itoh , Shigeo Nakano and Kosei Yasumoto 1 2 Department of Thoracic Surgery, Shinkomonji Hospital, Kitakyushu 800-0057, Japan, Department of Thoracic Surgery, Fukuoka-Wajiro Hospital, Fukuoka 811-0213, Japan, Department of Thoracic Surgery, Shintakeo Hospital, Takeo City, Saga 843-0024, Japan, Department of Thoracic Surgery, Shinyukuhashi Hospital, Yukuhashi City, Fukuoka 824-0026, Japan and Department of Pathology, Shinkomonji Hospital, Kitakyushu 800-0057, Japan *Correspondence address. Tetsuya So, Department of Thoracic Surgery, Shinkomonji Hospital, 2-5 Dairishinmachi, Moji-ku, Kitakyushu 800-0057, Japan. Tel: þ81-93-391-1001; Fax: þ81-93-391-7001; E-mail: tetsuya@shinkomonji-hp.jp Received 11 July 2014; revised 29 July 2014; accepted 2 August 2014 We present a case of a human pulmonary dirofilariasis in a 59-year-old man. At the medical examination, a chest computed tomography (CT) revealed a mass, measuring 18  15 mm in diameter, with an irregular margin on the bottom of the right lower lobe. We could not neglect the possibility of a primary lung cancer, and therefore, a lung partial resection was performed under video-assisted thoracoscopic surgery. The intra-operative pathological findings revealed inflammatory granuloma with coagulation necrosis and no malignant cells. The permanent pathological examination showed occlusion of the peripheral pulmonary artery by worms and formation of a necrotic mass surrounded by reactive inflammation and hemorrhage. Human pul- monary dirofilariasis is an extremely rare zoonotic infection, and sometimes it is difficult to dis- tinguish it from a primary lung cancer on radiographic findings. INTRODUCTION were all within their normal ranges. In May 2013, a lung partial resection was performed under video-assisted thoracic surgery Human pulmonary dirofilariasis is a rare zoonotic infection (VATS). At the time of thoracotomy, the mass located at the per- caused by the dog heartworm Dirofilaria immitis, which is trans- iphery of S9 of the right lower lobe and the surface was whitely mitted via a vector/intermediate host, generally the mosquito transparent (Fig. 2A). The mass was solid and hard on palpation [1, 2]. Owing to the increase in the number of a medical examin- (Fig. 2B). The intra-operative pathological findings (frozen ation, this disease has sometimes been reported recently. We section) revealed inflammatory granuloma with coagulation ne- herein report the rare case of a human pulmonary dirofilariasis. crosis. The permanent pathological examination showed occlu- sion of the peripheral pulmonary artery by worms and CASE REPORT formation of a necrotic mass surrounded by reactive inflamma- tory cells and hemorrhage in hematoxylin–eosin stain (Fig. 3A) A 59-year-old Japanese man visited our hospital in April 2013 and Elastica van Gieson stain (Fig. 3B). Post-operative course for a workplace medical examination. He was a former smoker was uneventful, and he is doing well 15 months after surgery. (from 20 to 26-year-old, 1 pack per day), and had neither phys- ical symptoms nor family history of malignancies. The chest X-ray did not detect an abnormal shadow, but a CT showed a mass with an irregular margin measuring 18  15 mm in diam- DISCUSSION eter at the subpleural area of S of the right lower lobe (Fig. 1). The CT detected no nodules in the other lobes or regional Human pulmonary dirofilariasis is a rare zoonotic infection lymph nodes swelling. A FDG-PET was performed, but did not caused by the canine heartworm Dirofilaria immitis [1, 2]. yield a definitive FDG accumulation (data not shown). Such According to the medical database, De Magelhaes first tumor markers as CEA, SLX, SCC, CYFRA, NSE, and ProGRP described the discovery of filarial worm in the left ventricle of Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved.# The Author 2014. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http:// creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Page 2 of 3 T. So et al. Figure 1: Pre-operative chest CT scan demonstrated the mass measuring 18  15 mm with spiculation at the periphery of S9 of the right lower lobe. Figure 3: (A) Histopathological findings of the pulmonary nodule. Two trans- verse sections of an immature worm were seen in the peripheral pulmonary with fibrous tissue, necrotic tissue, and granulation (hematoxylin and eosin staining, 6). (B) The transverse section in the peripheral pulmonary revealed layers of cuticle, internal longitudinal cuticular ridges and lateral chords, being identified as Dirofilaria immitis (Elastica van Gieson stain, 20). a Brazilian boy in 1887 [3]. Flieder and Moran [4] investi- gated that there were 31 of 41 nodules in the right side of the lung, and especially 19 of 31 nodules in the right lower lobe. In our case, he was asymptomatic and the mass was located in the right lower lobe. Furthermore, he bred a dog 20 years ago in our case. Milanez et al. [5] mentioned that the filarial infection rates of dogs were more important than the history of dog breeding. The radiographic images of human pulmonary dirofilariasis have some specific features. It has been reported that the chest X-ray showed a solitary, round nodule in a lung field in many cases. In our case, however, the chest X-ray did not reveal the mass because of overlapping of the diaphragm. A chest CT can detect the disease as a mass measuring 18  15 mm in diameter at the periphery of the right lower lobe close to the lateral pleura. Recently, a FDG-PET has been applying for the qualitative diagnosis of lung nodules. The FDG-PET had been described in three cases of human pulmonary dirofi- Figure 2: (A) Intra-operative thoracoscopy showed the white tumor with pleural lariasis for the differential diagnosis of pulmonary nodule [6]. indentation. (B) Macroscopic examination revealed white and solid tumor. Human pulmonary dirofilariasis Page 3 of 3 The standardized uptake value (SUV) of them was reported ACKNOWLEDGEMENTS from negative to 7.5, and the SUV was 1.4 in our case. In this We thank Dr Tamotsu Kanazawa of University of Environmental way, the FDG-PET cannot deny a primary lung cancer pre- and Occupational Health, Japan, for critical comments. operatively. Human pulmonary dirofilariasis is usually diagnosed by REFERENCES the surgical specimens, and it seems to be difficult to diag- nose this disease by using the other modalities. For example, 1. Shibasaki M, Shito I, Konno A, Yusa T, Shibuya K, Kubosawa H, et al. A case of pulmonary diroflariasis: cytological, pathological and a filarial infection in human causes eosinophilia in general. immunological diagnosis-(in Japanese with English abstract). J Jpn Soc Yoshimura et al. mentioned that human pulmonary dirofilar- Clin Cytol 1997;36:174–8. iasis had little specific findings in hematological analysis 2. Asimacopoulos PJ, Katras A, Christie B. Pulmonary dirofilariasis. The largest single-hospital experience. Chest 1992;102:851–5. and the average of eosinocytes in the peripheral blood was 3. Sakurai A, Gohara H, Tajiri N, Ando Y, Maruyama S, Yokoyama S, et al. 6.5% [7]. In our case, the eosinocytes in the peripheral Human pulmonary dirofilariasis; a case report and a review of 117 cases in blood was 1.3 and 1.9% pre-operatively. The other examin- Japan (in Japanese with English abstract). Jpn J Clin Radiol 2006;51:169–73. 4. Flieder DB, Moran CA. Pulmonary dirofilariasis: a clinicopathologic ation to detect dirofilaria-specific antibody, such as an study of 41 lesions in 39 patients. Hum Pathol 1999;30:251–6. enzyme-linked immunosorbent assay, immunological elec- 5. Milanez de Campos JR, Barbas CS, Filomeno LT, Fernandez A, trophoresis and agglutination of red blood cells, has been Minamoto H, Filho JV, et al. Human pulmonary dirofilariasis: analysis of 24 cases from Sao Paulo, Brazil. Chest 1997;112:729–33. reported to be useful [1, 7 – 9]. These immuno-serological 6. Shimokawa H, Hanagiri T, Takenoyama M, Yamada S, Kanazawa T, analyses, however, have some problem as cross-reactivity Yasumoto K. A case of pulmonary dirofilariasis with cavity formation and non-specificity between Dirofilaria immitis and the (in Japanese with English abstract). J Jpn Assoc Chest Surg 2011;25:21–4. 7. Sato M, Koyama A, Iwai K, Kawabata Y, Kojima S. Human pulmonary other filariasis. As a treatment, a lung resection has been per- dirofilariasis with special reference to the ELISA for the diagnosis and formed which leads to final diagnosis and cure. Recently, a follow-up study. Z parasitenkd 1985;71:561–3. VATS seems to be a mainstream, and the benefit is less inva- 8. Mori S, Yasunobu H, Mizoguchi A, Kawabata M, Nakamura F, Nawa Y, et al. Pulmonary dirofilariasis with serologic study on familial infection sive for the patient [6, 10]. with Dirofilaria immitis. Intern Med 2004;43:327–30. In conclusion, human pulmonary dirofilariasis is an ex- 9. Kunst H, Mack D, Kon OM, Banerjee AK, Chidini P, Grant A. Parasitic tremely rare zoonotic infection, and sometimes mimick a infections of the lung: a guide for the respiratory physician. Thorax 2011;66:528–36. primary lung cancer on radiographic findings. We, however, 10. Miyoshi T, Tsubouchi H, Iwasaki A, Shiraishi T, Nabeshima K, should be able to list up the one of differential diagnosis of Shirakusa T. Human pulmonary dirofilariasis: a case report and review lung cancer. of the recent Japanese literature. Respirology 2006;11:343–7.

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Published: Aug 28, 2014

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