Unusual Radiographic Findings in a Patient with Sarcoidosis

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1 J Radiol Sci 2011; 36: Unusual Radiographic Findings in a Patient with Sarcoidosis Swei-Hsiung Tsung 1 Mee-Sun Tsai 2 Wen-Hsian Ho 3 Hao-Chun Chang 1 Department of Pathology 1, Department of Medicine 2, Department of Radiology 3, St. Mary Hospitl, Luodong, Yilan, Taiwan Abstract We recently encountered a patient presenting with a solitary nodule on chest radiograph. The solitary pulmonary nodule (SPN) has extensive differential diagnosis, including both benign and malignant etiology. Management is frequently challenging. After wedge resection, the nodule was proven to be sarcoidosis. In Taiwan, sarcoidosis presenting as a SPN has never been documented. Recognition of this phenomenon aids in the proper diagnosis and subsequent conservative management. We wish to report this case to alert clinicians, radiologists and pathologists that solitary pulmonary nodule can develop in the lung in patients with sarcoidosis with or without hilar lymphadenopathy. A solitary pulmonary nodule (SPN) is noted in 1 of 500 chest radiographs. When solitary pulmonary nodule (SPN) is seen on chest radiographs; they could be either malignant or benign lesions. The most common benign causes are infectious granulomas (about 80%), and hamartomas (about 10%). The other benign etiologies are rheumatoid arthritis, intrapulmonary lymph nodes and sarcoidosis [1]. We recently encountered a patient with sarcoidosis presenting with a solitary pulmonary nodule on chest radiograph. Herein, we wish to report this case to share our experience. Case Report A 48-year-old female had chest tightness off and on for more than one month. She came to our hospital to seek medical attention. Physical examination showed nothing of note. Laboratory tests including routine chemistry and complete blood count with a differential were all within normal limits. The sputum cytology was negative. Chest radiography revealed a solitary nodule in her right lower lung field (Fig. 1). Computed tomography (CT) revealed a cm nodular opacity (Fig. 2) with hilar lymphadenopathy (Fig. 3). On lung window, the margins of this nodule are circumscribed. A malignant neoplasm was suspected. The patient underwent a wedge resection with a frozen section examination which was reported as noncaseating granulomas Hilar lymph node biopsy was also taken. Histologically, sections from the lung and hilar lymph nodes showed a similar microscopic appearance. They contained multiple noncaseating granulomas composed of histiocytes, lymphocytes and multinucleated giant cells. (Fig. 4, 5). Both acid fast stain and periodic acid Schiff stain failed to demonstrate TB bacilli or fungal elements. Cultures for TB and fungus later yielded negative results. The patient was started on glycosteroid hormone. Six months after surgery, she returned to the clinic. CT scan showed minimal decrease of her hilar lymphadenopathy. Correspondence Author to: Mee-Sun Tsai Department of Medicine, St. Mary Hospital, Luodong, Yilan, Taiwan No. 160, Chong-Chun S. Road, Luodong, Yilan 265, Taiwan 209

2 Figure 1 Figure 1. Plain chest x-ray film shows a solitary nodule in the right lower lung field. Discussion Our patient presented the typical radiographic findings as symmetrical hilar lymph- adenopathy. Histologically, both the lung nodule and hilar lymph nodes showed noncaseating granulomas. Acid fast stain was negative, as was periodic acid-schiff stain. She was treated with glycosteroid hormone with minimal response. The diagnosis of sarcoidosis was most likely. The first two Taiwanese cases of sarcoidosis were reported in the 1960s [2, 3]. In 1997, Perng et al noticed an increasing incidence in Northern Taiwan [4]. However, as there has been no island-wide survey, the true annual incidence of sarcoidosis in Taiwan still remains unknown. Thereafter, only a few sporadic cases have been reported [5-8]. The largest series was the report by Hsieh et al [8]. Typical chest radiographic findings of sarcoidosis include bilateral hilar lymphadenopathy (BHL), pulmonary infiltration, small nodules in a perilymphatic distribution [9]. However, Baughman et al [10] reported that 20-25% of patients of sarcoidosis lacked bilateral lymphadenopathy. In our case, the solitary pulmonary nodule caused confusion. Initially, we considered that the findings and BHL were unrelated. That was why the surgeon requested a frozen section examination. There are several reports that sarcoidosis presents as a solitary pulmonary nodule [11-22], mostly from the western literature, with a few reports from Japan. The patient reported by Koh et al had pulmonary sarcoidosis without BHL [14]. As a result, the correct diagnosis was delayed. From the literature, we could say that the radiologic findings of nodular sarcoidosis are not specific, and often mandate a 210

3 Figure 2 Figure 2. Computed tomography shows a solitary nodule in the right lower lung field. Figure 3 Figure 3. Computed tomography shows symmetrical hilar lymphadenopathy. 211

4 Figure 4 Figure 4. Histological section from lung, H & E. x 100. Showing multiple noncaseating granulomas. Figure 5 Figure 5. H & E. x400. The noncaseating granulomas contain histiocytes, lymphocytes and multinucleated giant cells. tissue diagnosis. In conclusion, we present this case to alert the clinicians, radiologists, and pathologists that solitary pulmonary nodules can develop in patient with sarcoidosis, with or without bilateral hilar lymphadenopathy. Recognition of this phenomenon aids in the proper diagnosis and subsequent conservative management. We acknowledge and appreciate the English revise of this article from Dr. Stephen S. Tsung. 212

5 References 1. Vahid B, Leone FT. The solitary pulmonary nodule:not always bronchogenic carcinoma. Primary care Resp J 2006; 15: Wu MC.Sarcoidosis. One case report with a general discussion. J Formosa Med Assoc 1960; 59: Hsing CT, Han FC, Liu HC. Sarcoidosis among Chinese. Am Rev Resp Dis 1964; 89: Perng RP, Chen JH, Tsai TT, Hsieh WC. Sarcoidosis among Chinese in Taiwan. J Formosa Med Assoc 1997; 96: Lee YY, Chao SC, Yang MH, Yan JT. Saroidosis in Taiwan: Clinical characteristics and atypical mycobacteria. J Formosa Med Assoc 2002; 101: Lee WY, Jeng CM, Hsu CY, Wu JT, Liu JS, Wang YC. Pulmonary sarcoidosis. Report of four cases. Chin J Radiol 2001; 26: Chang CS, Chen WL, Li CT, Wang PY. Carvenous sinus syndrome due to sarcoidosis: a case report. Acta Neurologica Taiwanica 2009; 18: Hsieh CW, Chen DY, Lan JL. Late onset and rare faradvanced pulmonary involvement in patients with sarcoisosis in Taiwan. J Formosa Med Assoc 2006; 105: Park HJ, Jung JI, Cheng MH, Sun WS, Kim HL, Baik JH. Typical and atypical manifestation of intrathoracic sarcoidosis. Korean J Radiol 2009; 10: Baughman BP, Tebrstein AS, Judsm MA, et al. Clinical characteristics of patients in a care control study of sarcoidosis. Am J Respir Crit Care Med 2001; 161: Sharma OP, Hewlet R, Gondosman I. Nodular Sarcoidosis: an unusual Radiographic appearance. Chest 1973; 84: Koh A, Doudsi Y, Yamamura M, et al. Pulmonary Sarcoidosis Showing a Solitary Large Nodule with a Pseudo-alveolar Pattern. Acta Med Nagasaki 2006; 84: Itoh T, Kobayashi D, Rensha K, Minami K. A case of sarcoidosis presenting as a solitary pulmonary nodule. Nibon Kyuki Gakkai Zasshi 2008; 46: Imokayna S, Sato A, Tahiguchi M, et al. A case of sarcoidosis presenting as pulmonary nodules associated with pleural indentation fifteen years after onset.,nibon Kyoba Shikkan Gakkai Zasshi 1993; 10: Chrisholm JC, Lang GR. Solitary circumscribed pulmonary nodule. An unusual manifestation of sarcoidosis. Arch Intern Med 1966; 118: Nutting S, Carr I, Cole PM. Solitary pulmonary nodules due to sarcoidosis. Can J Surg 1979; 22: Pinsker KL. Solitary pulmonary nodule in sarcoidosis. JAMA 1978; 240: Rubinstein I, Baum GL, Lieberman. Asymptomatic pulmonary nodule in sarcoidosis. Eur J Respir Dis 1985; 66: Steele JD, The solitarypulmonary nodule: report of a cooperative study of resected. Asymptomatic pulmonary nodules in male. J Thorac and Cardiovasc Surg 1963; 46: Rose RM, Lee RG, Costello P. Solitary nodular sarcoidosis. Clin Radiol 1985; 36: Gotway MB, Tchao NK, Leung JWT, Hanks DK, Thomas AN. Sarcoidosis presenting as an enlarging solitary pulmonary nodule. J Thoracic Imaging 2001; 16: Malaisamy S, Dalal B, Bimenyuy C, Soubani AO. The clinical and radiologic features of nodular pulmonary sarcoidosis. Lung 2009; 187: