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Weekly Chest CasesArchive of Old Cases

Case No : 962 Date 2016-04-04

  • Courtesy of So Yeon Ki, Hyun Ju Seon, Soo Hyun Kim, Sung Min Moon, Yun-Hyeon Kim / Chonnam National University Hospital
  • Age/Sex 36 / F
  • Chief ComplaintIncidentally detected abnormal lung lesion
  • Figure 1
  • Figure 2
  • Figure 3
  • Figure 4

Diagnosis With Brief Discussion

Diagnosis
Multifocal micronodular pneumocyte hyperplasia in tuberous sclerosis
Radiologic Findings
HRCT images (Fig. 1-2) show a random distribution of multiple and variable-sized ground glass opacity nodules in both lungs. Abdomen CT images (Fig. 3-4) show lobulated osteosclerotic lesion involving the lumbar spine and a well-defined nodule containing fat attenuation and lacy, enhancing area in the left kidney. VATS biopsy was performed and pathologic diagnosis was multifocal micronodular pneumocyte hyerplasia (MMPH). These findings suggest tuberous sclerosis complex (TSC) of the patient with MMPH, TS-associated osteosclerotic lesion, and an angiomyolipoma of kidney.
Brief Review
TSC is an autosomal dominant syndrome characterized by hamartomatous growth in the skin, eyes, kidneys, and central nervous system. Pulmonary involvement occurs in 1% of cases and usually manifests as lymphangioleiomyomatosis (LAM). MMPH is another, extremely rare pulmonary manifestation of TSC, which was first described by Popper et al. in 1991. MMPH is a hamartomatous process of the lung that exhibits multiple tiny pulmonary nodules. High-resolution CT in MMPH reveals nodules 1–8 mm in diameter that are diffusely scattered throughout the lung in a random distribution with regard to the secondary lobule. Pathologically, the minute pulmonary nodules represent clusters of cuboidal epithelial cells with mild cellular atypia lining thickened alveolar septa. The cell of origin is the type II pneumocyte.
The presence of multiple tiny pulmonary nodules in a random distribution in a TSC patient should suggest the presence of MMPH. The differential diagnosis should also include a miliary granulomatous infection, Langerhans’ cell histiocytosis (LCH), or hematogenous metastases. MMPH occurs more frequently in patients with TSC than with LAM and rarely presents in patients without TSC or LAM. The clinical manifestations resulting from isolated MMPH are dyspnea, cough, and mild to moderate hypoxemia. The clinical course of MMPH is usually not progressive, and treatment is unnecessary.

References
Ross L. Ristagno, Paul W. Biddinger, Elsira M. Pina, Cris A. Meyer. Multifocal Micronodular Pneumocyte Hyperplasia in Tuberous Sclerosis. AJR 2005;184:S37-39.
2. Yoshihiro Kobashi, Tadaaki Sugiu, Keiji Mouri, Tsutomu Irei. Clinicopathological analysis of multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis in Japan. Respirology. 2008;13:1076-1081.
Keywords
Lung, Multiple organ, Interstitial lung disease, ILD,

No. of Applicants : 118

▶ Correct Answer : 86/118,  72.9%
  • - Ishikawa Matto Central Hospital , Japan Manabu Akimoto
  • - SAISEIKAI KURIHASHI HOSPITAL , Japan YASUO OOKUBO
  • - Saitama-Sekishinkai Hosptal , Japan Mihoko Yamazaki
  • - Ostu Municipal Hospital , Japan Noriatsu Ichiba
  • - Osaka Rosai Hospital , Japan Hiromitsu Sumikawa
  • - Kinki University Faculty of Medicine, , Japan Mitsuru Matsuki
  • - Yokohama-asahi-chuo-general hospital , Japan Kyoko Nagai
  • - dayanand , India reena gupta
  • - Niigata University , Japan Atsushi Uehara
  • - Juntendo University Hospital , Japan Akifumi Hagiwara
  • - King Abdulaziz University Hospital , Saudi Arabia Amr Ajlan
  • - , Korea (South)
  • - Eren Hastanesi Istanbul , Turkey Armagan Sarac
  • - McGill University Health Center , Canada Alexandre Semionov
  • - Khoo Teck Phuat Hospital Singapore , Singapore ashish chawla
  • - 異⑸ , Korea (South) sooncheul baek
  • - Nihon University Itabashi Hospital , Japan Yusuke Watanabe
  • - Seoul national university , Korea (South) Boram Kim
  • - Ondokuz Mayis University , Turkey Cetin Celenk
  • - Policlinico Umberto I , Italy Paolo Baldassari
  • - Kyungpook national university , Korea (South) Chungeun Lim
  • - Chonnam national university hospital , Korea (South) GYEONG MOON
  • - Chonnam national university hospital , Korea (South) INSOO JANG
  • - Tokyo university, Japan , Korea (South)
  • - Busan national univercity hospital radiology department , Korea (South) Il-wan Son
  • - NEUROCITI DIAGNOSTICS , India Justin Moses
  • - Asan Medical Center , Korea (South) Hyung Jung Koo
  • - Seoul National University Bundang Hospital , Korea (South) Ji Eun Park
  • - �stanbul , Turkey Ayhan Yilmaz
  • - Oita University, Faculty of Medicine , Japan Fumito Okada
  • - The Jikei university , Japan Taku Gomi
  • - The first affiliated Hospital of Xiamen University , China Qing Qiang Guo
  • - Pusan national university hospital , Korea (South) SeungHyun Lee
  • - , Korea (South) BAEK YOOLIM
  • - Onomichi municipal hospital , Japan Hirofumi Mifune
  • - The University of Tokyo Hospital , Japan Toshihiro Furuta
  • - Chonnam national university hospital , Korea (South) Jong-hyeon Kim
  • - Chung buk national university hospital , Korea (South) hong Byun
  • - Chonnam Nationl University Radiology , Korea (South) juwon kim
  • - Kizawa Memorial Hospital , Japan Yo Kaneko
  • - Bahcesehir University School of Medicine, Radiology Department , Turkey Mustafa Kemal Demir
  • - 異⑸ , Korea (South) Junghwan Kim
  • - Chonnam National University Hospital , Korea (South) So yeon Ki
  • - Chungbuk national university , Korea (South) Yong Kim
  • - Medicheck health care , Korea (South) Chae Lim
  • - Kyung Hee University Hospital at Gangdong , Korea (South) Han Na Lee
  • - chungbuk national university hospital , Korea (South) lee jung hwan
  • - University of Tsukuba Hospital , Japan Hiroaki Takahashi
  • - Goa Medical College , India Paresh Desai
  • - Mallinckrodt Institute of Radiology , United States Naganathan Mani
  • - Pusan national university hospital , Korea (South) hie bum suh
  • - CLINIQUE STE CLOTILDE , Reunion patrick MASCAREL
  • - Okayama University Hospital , Japan Toshiyuki Komaki
  • - Diskapi Yildirim Beyazit Hospital, Ankara , Turkey Meric Tuzun
  • - University of Toronto , Canada Micheal McInnis
  • - Asan Medical Center, Ulsan University , Korea (South) Mi Young Kim
  • - University of Tsukuba, Dept of Radiologt , Japan Manabu Minami
  • - Avrasya Hospital , Turkey Murat Ulusoy
  • - Les cedres St malo , France nicolas gautier
  • - Ajou university hospital , Korea (South) Hye Lin Kim
  • - Hallym University Sacred Heart Hospital , Korea (South) Jee Hye Hur
  • - Insung Hospital , Korea (South) Kyung bum Nam
  • - Seoul national university hospital , Korea (South) Kumju Chae
  • - CHU Poitiers , France CHAN paul
  • - GHICL , France manuel toledano
  • - Himeji Redcross Hospital , Japan Soichiro Okamoto
  • - Kanto Rosai Hospital , Japan Taku Tajima
  • - Daya general Hospital , India Raishad Khan
  • - Daejin Medical Center Bundang Jesaeng General Hospital , Korea (South) Donghwan Kim
  • - bundang jesaeng , Korea (South) kyung su kwag
  • - Centre Imagerie de Fribourg , Switzerland Benoit RIZK
  • - Maternity & child hospital , Saudi Arabia Sania ALBlushi
  • - Ajou University Hospital , Korea (South) Seulgi You
  • - Fortis hospital , Mohali , India Shaleen Rana
  • - Kizawa Memorial Hospital , Japan Shoji Okuda
  • - National Center hospital of Neurology and Psychiatry , Japan Kaoru Sumida
  • - Ichinomiya Nishi Hospital , Japan Takao Kiguchi
  • - University of Tsukuba , Japan Toshitaka Ishiguro
  • - Private sector , Greece Vasilios Tzilas
  • - Diagnose.me (BV) / Royal Perth Hospital , Australia Yuranga Weerakkody
  • - Pusan national university hospital , Korea (South) Jin Joo Kim
  • - Kyoto University , Japan Akihiko Sakata
  • - chungbuk university hospital , Korea (South) Hyeonmi Ryu
  • - Chonnam national university hospital , Korea (South) Lee Bin
  • - Asan medical center , Korea (South) Cherry Kim
  • - Seirei Hamamatsu General Hospital , Japan Kenichi Mizuki
▶ Correct Answer as Differential Diagnosis : 12/118,  10.2%
  • - Shiga University of Medical Science , Japan Akitoshi Inoue
  • - PNUH , Korea (South) Seungbaek Hong
  • - pusan national university hospital , Korea (South) nam soo won
  • - Pusan National University Hospital , Korea (South) LEE YOU JIN
  • - Pusan national university hospital , Korea (South) MIRI JEONG
  • - Department of Radiology, Seoul National University Bundang Hospital , Korea (South) Sung Hyun Yoon
  • - pusan national university hospital , Korea (South) kim jeongmyeong
  • - Pusan National University Hospital , Korea (South) Jeongbin Park
  • - NASA SCANS , India RAKESH BHATIA
  • - Korea University Guro Hospital , Korea (South) Jeong Woo Kim
  • - Pusan national university hospital , Korea (South) So Park
  • - Pusan National University Yangsan Hospital , Korea (South) SEUNG-MIN LEE
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