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