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Weekly Chest CasesArchive of Old Cases

Case No : 1230 Date 2021-05-19

  • Courtesy of Bo Mi Gil and Myung Hee Chung / Bucheon St. Mary’s Hospital, The Catholic University of Korea, Korea.
  • Age/Sex 50 / M
  • Chief Complaintmild cough and sputum, 1month ago. industrial dust inhalation exposure for 10 yrs
  • Figure 1
  • Figure 2

Diagnosis With Brief Discussion

Diagnosis
(secondary) Pulmonary alveolar proteinosis
Radiologic Findings
Fig. 1. Chest posteroanterior radiograph showing heterogeneous patchy ground-glass opacities (GGOs) in the middle and lower lung fields.
Fig. 2. Computed tomography scan showing geographic GGOs and interlobular septal thickening in the entire lungs with middle and lower predominance.
Brief Review
Bronchoscopy revealed redness of the bronchial mucosa, and Streptococcus pneumoniae was detected. Video-assisted thoracoscopic surgery was performed for the confirmation of unimproved radiologic ground-glass opacity (GGO) findings. The surgical specimen was positive for periodic acid-Schiff (PAS) stain, leading to the final diagnosis of pulmonary alveolar proteinosis (PAP). After bronchoalveolar lavage treatment, the radiologic findings improved on the follow-up chest computed tomography (CT) and chest posteroanterior radiography, and PAP did not recur for 7 years.
PAP is a rare disease characterized by abnormal intra-alveolar accumulation of lipoproteins (1, 2). Three distinct subgroups of PAP are currently recognized: idiopathic, secondary, and congenital (3). Secondary PAP (5%–10% of cases) is recognized in patients with inhalation exposure to industrial materials such as silica particles, cement dust, aluminum dust, titanium dioxide, nitrogen dioxide, and fiberglass; in those with underlying hematologic malignancies; or in those with immunodeficiency disorders (including cytotoxic or immunosuppressive therapy and human immunodeficiency virus infection) (3). Our case was considered a secondary form caused by the recent heavy exposure to dust. PAP was originally defined by histopathologic findings of amorphous PAS-positive lipoproteinaceous materials filling the alveoli, with a relatively normal background lung architecture.
The “crazy-paving” pattern on CT is defined as a network of smoothly thickened reticular (septal) lines superimposed on areas of GGO. Areas of crazy-paving in PAP are typically widespread and bilateral, often with sharply marginated regions of geographic or lobular sparing. Crazy-paving has a broad radiologic differential diagnosis, including left heart failure, pneumonia (especially pneumocystis pneumonia), alveolar hemorrhage, bronchoalveolar carcinoma, lymphangitic carcinomatosis, diffuse alveolar damage (adult respiratory distress syndrome), radiation- or drug-induced pneumonitis, hypersensitivity pneumonitis, and pulmonary veno-occlusive disease.
In unusual cases, clinical remission or quiescence of PAP may occur; however, intervention is required in most patients, and the treatment depends on the particular form of the disease (3). Standard treatment includes therapeutic whole-lung bronchoalveolar lavage to remove alveolar material. Supplementation with granulocyte-macrophage colony-stimulating factor has been used, with varying effectiveness (1, 2, 3).
Please refer to
Case 818, Case 843, Case 880, Case 883, Case 1033, Case 1200,
References
1) Godwin JD, Müller NL, Takasugi JE. Pulmonary alveolar proteinosis: CT findings. Radiology. 1988;169 (3): 609-13.
2) Ioachimescu OC, Kavuru MS. Pulmonary alveolar proteinosis. Chron Respir Dis 2006;3:149–159.
3) Frazier AA, Franks TJ, Cooke EO et-al. From the archives of the AFIP: pulmonary alveolar proteinosis. Radiographics. 28 (3): 883-99.
Keywords

No. of Applicants : 91

▶ Correct Answer : 65/91,  71.4%
  • - Saitama-Sekishinkai Hosptal , Japan MIHOKO YAMAZAKI
  • - , Japan HIROAKI ARAKAWA
  • - , Japan KEI SASAKI
  • - ZIGONG TCM HOSPITAL OF CHINA , China CAO CUN YOU
  • - Kinki University Faculty of Medicine, , Japan MITSURU MATSUKI
  • - Showa University Fujigaoka Hospital , Japan KYOKO NAGAI
  • - Showa General Hospital , Japan MASAFUMI KAIUME
  • - Mayo Clinic , United States AKITOSHI INOUE
  • - Chungbuk National University Hospital , Korea (South) YEONGTAE PARK
  • - Other , Korea (South) HYEJIN YANG
  • - , Korea (South) HYEYOUNG CHOI
  • - Affilitated Hospital of Jining Medical college , China JIANG SHENG HUA
  • - Chonnam National University Hwasun Hospital , Korea (South) SEONGWOO CHO
  • - Chonbuk National University Hospital , Korea (South) MINGI SHIN
  • - McGill University Health Center , Canada ALEXANDRE SEMIONOV
  • - Osaka Red Cross Hospital , Japan YUSUKE UTSUNOMIYA
  • - NIMS, HYDERABAD , India BHASKAR K
  • - National Center of Neurology and Psychiatry , Japan MOTO NAKAYA
  • - , Italy PAOLO BALDASSARI
  • - Tiger Gate Hospital , Japan SHIN-ICHI CHO
  • - Vita Hospital , Brazil DIOGO LAGO PINHEIRO
  • - Other , Korea (South) SEONGSU KANG
  • - , Korea (South) JIN YOUNG LEE
  • - Chonbuk National University Hospital , Korea (South) DAEUL LEE
  • - Chonbuk National University Hospital , Korea (South) JUNGHWAN KIM
  • - National Center for Global Health and Medicine , Japan MASATOSHI HOTTA
  • - , Korea (South) HYEWON CHOI
  • - The University of Tokyo Hospital , Japan TOSHIHIRO FURUTA
  • - HHS , Canada S LEE
  • - Seoul National University Bundang Hospital , Korea (South) SUNG HYUN YOON
  • - Narayana Multispeciality Hospital Jaipur Rajasthan , India JAINENDRA JAIN
  • - Kyeongpook National University Hospital , Korea (South) HYEONJUN JANG
  • - Yeungnam University Medical Center , Korea (South) JONGSOO PARK
  • - VDC, HYDERABAD , India KARTHIK RAYASAM
  • - , Turkey AKIN KAYA
  • - McGill , Canada MUTAZ ADNAN KHAIRO
  • - Mallinckrodt Institute of Radiology , United States NAGANATHAN MANI
  • - Osaka City General Hospital , Japan SHU MATSUSHITA
  • - Chungbuk National University Hospital , Korea (South) MIHYEON PARK
  • - Other , Korea (South) MINSU KIM
  • - University of Tsukuba, Dept of Radiology , Japan MANABU MINAMI
  • - Seoul National University Hospital , Korea (South) SE WOO KIM
  • - Other , Korea (South) DONGYOUNG JEONG
  • - , Japan HIROAKI ARAKAWA
  • - Wuhan Union Hospital , China QIGUANG CHENG
  • - Holy Family Hopsital , India RAJESH GOTHI
  • - Kyoto University Hospital , Japan SATOSHI IKEDA
  • - Chonbuk National University Hospital , Korea (South) MINJEE KIM
  • - , Korea (South) SUBIN LEE
  • - Toranomon Hospital , Japan KAORU SUMIDA
  • - Chonbuk National University Hospital , Korea (South) DONG HAN SHIN
  • - Chonbuk National University Hospital , Korea (South) SEOYUN CHOI
  • - Ichinomiya Nishi Hospital , Japan Takao Kiguchi
  • - , Japan KAZUMA TERAUCHI
  • - Chonnam National University Hospital , Korea (South) SEUNG WAN KANG
  • - Chonbuk National University Hospital , Korea (South) YOUNG JU SONG
  • - Private sector , Greece VASILIOS TZILAS
  • - College of Medicine, CHA University , Korea (South) PARK JONG WON
  • - Columbia asia radiology group , India VIKHYATH SHETTY Y
  • - Chonnam National University Hospital , Korea (South) JINWANG PARK
  • - , Japan YUKI HAYASHI
  • - Chonnam National University Hwasun Hospital , Korea (South) WONGI JEONG
  • - Kyoto university , Japan AKIHIKO SAKATA
  • - Hanyang University Hospital , Korea (South) SEUNG JIN YOO
  • - Jiangsu province hospital , China WANGJIAN ZHA
▶ Correct Answer as Differential Diagnosis : 9/91,  9.9%
  • - CHCB , France LE GUEN RAPHAEL
  • - Ichinomiya Nishi Hospital , Japan CHIHIRO KIGUCHI
  • - Inje University Pusan Paik Hospital , Korea (South) JIYEON HAN
  • - Gifu University Hospital , Japan Yo Kaneko
  • - , Korea (South) EUNJIN LEE
  • - Soonchunhyang University Hospital Bucheon , Korea (South) SEONGHWAN BYUN
  • - AIMS , India NITHYA HARIDAS MALIYAM
  • - Other , Korea (South) KYU-CHONG LEE
  • - The University of Tokyo Hospital , Japan HIROSHI TAKUMIDA
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