Skip to main content
Top
Published in: Journal of Medical Case Reports 1/2012

Open Access 01-12-2012 | Case report

Tuberous sclerosis diagnosed by incidental computed tomography findings of multifocal micronodular pneumocyte hyperplasia: a case report

Authors: Makoto Ishii, Koichiro Asano, Nobufumi Kamiishi, Yuichiro Hayashi, Daisuke Arai, Mizuha Haraguchi, Hiroaki Sugiura, Katsuhiko Naoki, Sadatomo Tasaka, Kenzo Soejima, Koichi Sayama, Tomoko Betsuyaku

Published in: Journal of Medical Case Reports | Issue 1/2012

Login to get access

Abstract

Introduction

The majority of multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis complex is diagnosed with the classical clinical triad of seizures, mental retardation, and skin lesions. We report a rare case of tuberous sclerosis complex with no classical clinical findings, which was diagnosed through incidental computed tomography findings of multiple nodular lesions of multifocal micronodular pneumocyte hyperplasia.

Case presentation

A chest computed tomography scan of a 51-year-old Japanese woman showed multiple nodular ground-glass opacities that were not seen on chest X-ray. Video-assisted thoracoscopic surgery was performed. A histological examination demonstrated type II pneumocyte hyperplasia with thickened fibrotic alveolar septa, which was consistent with multifocal micronodular pneumocyte hyperplasia. Brain magnetic resonance imaging displayed multiple cortical tubers, and abdominal computed tomography showed bilateral renal angiomyolipoma. Our patient was finally diagnosed as having tuberous sclerosis complex with multifocal micronodular pneumocyte hyperplasia, although she had no episodes of epilepsy, no skin lesions, and no family history.

Conclusions

Multifocal micronodular pneumocyte hyperplasia with latent tuberous sclerosis complex should be considered in the differential diagnosis of multiple ground-glass opacities.
Appendix
Available only for authorised users
Literature
1.
go back to reference Bourneville D: Sclerose tubereuse des circonvolutions cerebrales: idiotie et epilepsie hemiplegique. Arch Neurol (Paris). 1880, 1: 81-91. Bourneville D: Sclerose tubereuse des circonvolutions cerebrales: idiotie et epilepsie hemiplegique. Arch Neurol (Paris). 1880, 1: 81-91.
2.
go back to reference Crino PB, Nathanson KL, Henske EP: The tuberous sclerosis complex. N Engl J Med. 2006, 355: 1345-1356. 10.1056/NEJMra055323.CrossRefPubMed Crino PB, Nathanson KL, Henske EP: The tuberous sclerosis complex. N Engl J Med. 2006, 355: 1345-1356. 10.1056/NEJMra055323.CrossRefPubMed
3.
go back to reference Castro M, Shepherd CW, Gomez MR, Lie JT, Ryu JH: Pulmonary tuberous sclerosis. Chest. 1995, 107: 189-195. 10.1378/chest.107.1.189.CrossRefPubMed Castro M, Shepherd CW, Gomez MR, Lie JT, Ryu JH: Pulmonary tuberous sclerosis. Chest. 1995, 107: 189-195. 10.1378/chest.107.1.189.CrossRefPubMed
4.
go back to reference Franz DN, Brody A, Meyer C, Leonard J, Chuck G, Dabora S, Sethuraman G, Colby TV, Kwiatkowski DJ, McCormack FX: Mutational and radiographic analysis of pulmonary disease consistent with lymphangioleiomyomatosis and micronodular pneumocyte hyperplasia in women with tuberous sclerosis. Am J Respir Crit Care Med. 2001, 164: 661-668.CrossRefPubMed Franz DN, Brody A, Meyer C, Leonard J, Chuck G, Dabora S, Sethuraman G, Colby TV, Kwiatkowski DJ, McCormack FX: Mutational and radiographic analysis of pulmonary disease consistent with lymphangioleiomyomatosis and micronodular pneumocyte hyperplasia in women with tuberous sclerosis. Am J Respir Crit Care Med. 2001, 164: 661-668.CrossRefPubMed
5.
go back to reference Muir TE, Leslie KO, Popper H, Kitaichi M, Gagne E, Emelin JK, Vinters HV, Colby TV: Micronodular pneumocyte hyperplasia. Am J Surg Pathol. 1998, 22: 465-472. 10.1097/00000478-199804000-00012.CrossRefPubMed Muir TE, Leslie KO, Popper H, Kitaichi M, Gagne E, Emelin JK, Vinters HV, Colby TV: Micronodular pneumocyte hyperplasia. Am J Surg Pathol. 1998, 22: 465-472. 10.1097/00000478-199804000-00012.CrossRefPubMed
6.
go back to reference Cancellieri A, Poletti V, Corrin B: Respiratory failure due to micronodular type II pneumocyte hyperplasia. Histopathology. 2002, 41: 263-265.CrossRefPubMed Cancellieri A, Poletti V, Corrin B: Respiratory failure due to micronodular type II pneumocyte hyperplasia. Histopathology. 2002, 41: 263-265.CrossRefPubMed
7.
go back to reference Umeoka S, Koyama T, Miki Y, Akai M, Tsutsui K, Togashi K: Pictorial review of tuberous sclerosis in various organs. Radiographics. 2008, 28: e32-10.1148/rg.e32.CrossRefPubMed Umeoka S, Koyama T, Miki Y, Akai M, Tsutsui K, Togashi K: Pictorial review of tuberous sclerosis in various organs. Radiographics. 2008, 28: e32-10.1148/rg.e32.CrossRefPubMed
8.
go back to reference Roach ES, Gomez MR, Northrup H: Tuberous sclerosis complex consensus conference: revised clinical diagnostic criteria. J Child Neurol. 1998, 13: 624-628. 10.1177/088307389801301206.CrossRefPubMed Roach ES, Gomez MR, Northrup H: Tuberous sclerosis complex consensus conference: revised clinical diagnostic criteria. J Child Neurol. 1998, 13: 624-628. 10.1177/088307389801301206.CrossRefPubMed
9.
go back to reference Vogt H: Zur Diagnostik der tuberosen sklerose. Z Erforsch Behandl Jugeudl Schwachsinns. 1908, 2: 1-16. Vogt H: Zur Diagnostik der tuberosen sklerose. Z Erforsch Behandl Jugeudl Schwachsinns. 1908, 2: 1-16.
10.
go back to reference Curatolo P, Verdecchia M, Bombardieri R: Tuberous sclerosis complex: a review of neurological aspects. Eur J Paediatr Neurol. 2002, 6: 15-23. 10.1053/ejpn.2001.0538.CrossRefPubMed Curatolo P, Verdecchia M, Bombardieri R: Tuberous sclerosis complex: a review of neurological aspects. Eur J Paediatr Neurol. 2002, 6: 15-23. 10.1053/ejpn.2001.0538.CrossRefPubMed
11.
go back to reference Thiele EA: Managing epilepsy in tuberous sclerosis complex. J Child Neurol. 2004, 19: 680-686.PubMed Thiele EA: Managing epilepsy in tuberous sclerosis complex. J Child Neurol. 2004, 19: 680-686.PubMed
12.
go back to reference Schwartz RA, Fernandez G, Kotulska K, Jozwiak S: Tuberous sclerosis complex: advances in diagnosis, genetics, and management. J Am Acad Dermatol. 2007, 57: 189-202. 10.1016/j.jaad.2007.05.004.CrossRefPubMed Schwartz RA, Fernandez G, Kotulska K, Jozwiak S: Tuberous sclerosis complex: advances in diagnosis, genetics, and management. J Am Acad Dermatol. 2007, 57: 189-202. 10.1016/j.jaad.2007.05.004.CrossRefPubMed
13.
go back to reference Rose VM, Au KS, Pollom G, Roach ES, Prashner HR, Northrup H: Germ-line mosaicism in tuberous sclerosis: how common?. Am J Hum Genet. 1999, 64: 986-992. 10.1086/302322.CrossRefPubMedPubMedCentral Rose VM, Au KS, Pollom G, Roach ES, Prashner HR, Northrup H: Germ-line mosaicism in tuberous sclerosis: how common?. Am J Hum Genet. 1999, 64: 986-992. 10.1086/302322.CrossRefPubMedPubMedCentral
14.
go back to reference McCormack FX: Lymphangioleiomyomatosis: a clinical update. Chest. 2008, 133: 507-516. 10.1378/chest.07-0898.CrossRefPubMed McCormack FX: Lymphangioleiomyomatosis: a clinical update. Chest. 2008, 133: 507-516. 10.1378/chest.07-0898.CrossRefPubMed
15.
go back to reference Kobashi Y, Sugiu T, Mouri K, Irei T, Nakata M, Oka M: Multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis: differentiation from multiple atypical adenomatous hyperplasia. Jpn J Clin Oncol. 2008, 38: 451-454. 10.1093/jjco/hyn042.CrossRefPubMed Kobashi Y, Sugiu T, Mouri K, Irei T, Nakata M, Oka M: Multifocal micronodular pneumocyte hyperplasia associated with tuberous sclerosis: differentiation from multiple atypical adenomatous hyperplasia. Jpn J Clin Oncol. 2008, 38: 451-454. 10.1093/jjco/hyn042.CrossRefPubMed
Metadata
Title
Tuberous sclerosis diagnosed by incidental computed tomography findings of multifocal micronodular pneumocyte hyperplasia: a case report
Authors
Makoto Ishii
Koichiro Asano
Nobufumi Kamiishi
Yuichiro Hayashi
Daisuke Arai
Mizuha Haraguchi
Hiroaki Sugiura
Katsuhiko Naoki
Sadatomo Tasaka
Kenzo Soejima
Koichi Sayama
Tomoko Betsuyaku
Publication date
01-12-2012
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2012
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/1752-1947-6-352

Other articles of this Issue 1/2012

Journal of Medical Case Reports 1/2012 Go to the issue