medicina Case Report Pneumoconiosis with a Sarcoid-Like Reaction Other than Beryllium Exposure: A Case Report and Literature Review Fumiko Hayashi 1, Takashi Kido 1,*, Noriho Sakamoto 1, Yoshiaki Zaizen 2, Mutsumi Ozasa 1,2, Mitsuru Yokoyama 3, Hirokazu Yura 1, Atsuko Hara 1 , Hiroshi Ishimoto 1, Hiroyuki Yamaguchi 1 , Taiga Miyazaki 4, Yasushi Obase 1, Yuji Ishimatsu 5, Yoshinobu Eishi 6, Junya Fukuoka 2 and Hiroshi Mukae 1 1 Department of Respiratory Medicine, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki 852-8501, Japan; [email protected] (F.H.); [email protected] (N.S.); [email protected] (M.O.); [email protected] (H.Y.); [email protected] (A.H.); [email protected] (H.I.); [email protected] (H.Y.); [email protected] (Y.O.); [email protected] (H.M.) 2 Department of Pathology, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki 852-8520, Japan; [email protected] (Y.Z.); [email protected] (J.F.) 3 Department of Anatomy, University of Occupational and Environmental Health, 1-1, Iseigaoka, Yahatanishiku, Kitakyushu City, Fukuoka 807-8555, Japan; [email protected] 4 Department of Infectious Diseases, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki 852-8501, Japan; [email protected] 5 Department of Nursing, Nagasaki University Graduate School of Biomedical Sciences, 1-7-1 Sakamoto, Nagasaki 852-8520, Japan; [email protected] 6 Department of Human Pathology, Tokyo Medical and Dental University, Yushima, Bunkyo-ku, Tokyo 113-8519, Japan; [email protected] * Correspondence: [email protected]; Tel.: +81-95-819-7273; Fax: +81-95-849-7285 Received: 12 October 2020; Accepted: 20 November 2020; Published: 22 November 2020 Abstract: Background: Chronic beryllium disease (CBD) is a granulomatous disease that resembles sarcoidosis but is caused by beryllium. Clinical manifestations similar to those observed in CBD have occasionally been reported in exposure to dusts of other metals. However, reports describing the clinical, radiographic, and pathological findings in conditions other than beryllium-induced granulomatous lung diseases, and detailed information on mineralogical analyses of metal dusts, are limited. Case presentation: A 51-year-old Japanese man with rapidly progressing nodular shadows on chest radiography, and a 10-year occupation history of underground construction without beryllium exposure, was referred to our hospital. High-resolution computed tomography showed well-defined multiple centrilobular and perilobular nodules, and thickening of the intralobular septa in the middle and lower zones of both lungs. No extrathoracic manifestations were observed. Pathologically, the lung specimens showed 5–12 mm nodules with dust deposition and several non-necrotizing granulomas along the lymphatic routes. X-ray analytical electron microscopy of the same specimens revealed aluminum, iron, titanium, and silica deposition in the lung tissues. The patient stopped smoking and changed his occupation to avoid further dust exposure; the chest radiography shadows decreased 5 years later. Conclusion: The radiological appearances of CBD and sarcoidosis are similar, although mediastinal or hilar lymphadenopathy is less common in CBD and is usually seen in the presence of parenchymal opacities. Extrathoracic manifestations are also rare. Despite limited evidence, these findings are similar to those observed in pneumoconiosis with a sarcoid-like reaction due to exposure to dust other than of beryllium. Aluminum is frequently detected in patients with pneumoconiosis with a sarcoid-like reaction and is listed as an inorganic agent in the etiology of sarcoidosis. It was also detected in our patient and may have contributed to the etiology. Medicina 2020, 56, 630; doi:10.3390/medicina56110630 www.mdpi.com/journal/medicina Medicina 2020, 56, x 630 FOR PEER REVIEW 22 of of 11 12 is listed as an inorganic agent in the etiology of sarcoidosis. It was also detected in our patient and Additionally,may have contributed our case suggests to the thatetio cessationlogy. Additionally, of dust exposure our case may contributesuggests that to improvement cessation ofunder dust exposurethe aforementioned may contribute conditions. to improvement under the aforementioned conditions. Keywords: aluminum;aluminum; berylliosis; berylliosis; chronic chronic beryllium beryllium disease; disease; pneumoconiosis; pneumoconiosis; sarcoidosis; sarcoidosis; sarcoid- likesarcoid-like reaction; reaction; X-ray analytical X-ray analytical electronelectron microscopy microscopy 1. Introduction Introduction Studies havehave shown shown that that exposure exposure to industrial to indust dustrial may dust lead tomay the developmentlead to the ofdevelopment granulomatous of granulomatouslung disease. Hardy lung and disease. Tabershaw Hardy have and described Tabershaw a granulomatous have described disease a granulomatous that resembles disease sarcoidosis that resemblesbut is caused sarcoidosis by beryllium, but is caused known by as beryllium, berylliosis known or chronic as berylliosis beryllium or disease chronic (CBD) beryllium [1]. Clinicaldisease (CBD)manifestations [1]. Clinical similar manifestations to those observed similar in to CBD those have observed occasionally in CBD been have reported occasionally in cases been of exposurereported into dustcases of of other exposure metals [to2– 13dust]. However, of other reports metals describing [2–13]. However, the clinical, reports radiographic, describing and pathologicalthe clinical, radiographic,findings of conditions and pathological other than findings beryllium-induced of conditions pneumoconiosis other than beryllium-induced with a sarcoid-like pneumoconiosis reaction, and withdetailed a sarcoid-like information reaction, on mineralogical and detailed analyses information of metal on dusts mineralogical are limited. analyses Herein, of wemetal report dusts a case are limited.of pneumoconiosis Herein, we withreport a sarcoid-likea case of pneumoconiosis reaction other with than a CBD sarcoid-like and review reaction similar other reported than CBD cases and in reviewthe literature. similar reported cases in the literature. 2. Case Case Presentation Presentation A 51-year-old, asymptomatic Japanese man wi withth rapidly progressing nodular shadows, as observed on chest radiography perf performedormed as part of his routine physical examinations, was referred to our hospital. Two Two years ago, hishis chestchest radiographradiograph hadhad revealedrevealed nono abnormalities.abnormalities. Small nodules had appeared a year later, mainly in both the lower lung fields fields (Figure 11A);A); however,however, hehe diddid notnot visitvisit a hospital. Further, Further, he reported a smoking history of 30 pack-years. As As a a civil civil engineer, he had been engaged in underground construction with welding, cutting, and drilling of reinforced concrete and metals for 10 years (between the ages of 41 and 51 years); though he had never used and/or and/or been exposed to beryllium. (A) Figure 1. Cont. Medicina 2020, 56, 630 3 of 12 Medicina 2020, 56, x FOR PEER REVIEW 3 of 11 (B) Figure 1. 1. (A(A) )Chest Chest radiograph radiograph 1 year 1 year prior prior to referral to referral to our to ourhospital, hospital, showing showing small small nodules nodules in both in theboth lower the lower lung lungfields. fields. (B) Chest (B) Chest radiograph radiograph on onadmission, admission, show showinging an an increase increase in in the the number number of of nodules in 1 year. The patient did not not display display clinical clinical respiratory respiratory sy symptomsmptoms such such as as dyspnea, dyspnea, exercise exercise intolerance, intolerance, or any other relevant manifestations upon referral to our hospital. The The patient’s patient’s body body temperature, temperature, pulse rate, and respiratory rate were 36.9 °C,◦C, 58 58 be beatsats/min,/min, and and 20 20 breaths/min, breaths/min, respectively. respectively. On chest auscultation, fine fine crackles were audible in the bilateral lower lung fields. fields. Clubbed fingers fingers were not observed. Laboratory Laboratory data data on on admission admission revealed revealed elevated elevated levels levels of of PR3-ANCA PR3-ANCA (15.4 (15.4 U/mL, U/mL, cutoff cutoff level:level: 3.5 3.5 U/mL). U/mL). Other Other representative representative tests tests for for coll collagenagen disease-related disease-related autoantibodies autoantibodies were were negative. negative. Serum levels levels of of angiotensin-conver angiotensin-convertingting enzyme, enzyme, carcinoembryonic carcinoembryonic antigen, antigen, KL-6, KL-6, and and sIL-2R sIL-2R were were in thein the normal normal range, range, and and the the results results of of interferon interferon-gamma-gamma release release assay assay were were negative. The The chest radiograph on admission showedshowed anan increaseincrease inin thethe numbernumber ofof nodules nodules compared compared with with those those found found 1 1year year previously previously (Figure (Figure1B). 1B). High-resolution computed computed tomography (HRCT) (HRCT) (Figure (Figure 22)) showedshowed well-definedwell-defined multiplemultiple centrilobular andand perilobularperilobular nodules, nodules, and and thickening thickening of theof the intralobular intralobular septa septa in the in middle the middle and lower and lowerzones