Multifocal micronodular pneumocyte hyperplasia (MMPH) is a rare pulmonary disease, generally manifesting as a tuberous sclerosis complex (TSC), characterised by multiple, small ground-glass nodular shadows on chest computed tomography (CT). Histological examination typically reveals multicentric, well-demarcated, nodular type II pneumocystic growth. Herein, we describe three cases of this rare pulmonary disease occurring within one family. Using reverse transcription polymerase chain reaction (RT-PCR) and direct DNA sequencing, we identified a novel germline mutation, a point mutation in TSC1 intron 5, which yielded a splice variant and loss of function of TSC1. Furthermore, immunohistochemical staining indicated the expression of phospho-p70S6K and phospho-4E-BP1, suggesting that TSC1 function was impaired by the novel gene mutation in MMPH cells.

Download full-text PDF

Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6386448PMC
http://journals.plos.org/plosone/article?id=10.1371/journal.pone.0212370PLOS

Publication Analysis

Top Keywords

multifocal micronodular
8
micronodular pneumocyte
8
pneumocyte hyperplasia
8
mutation tsc1
8
three cases
8
rare pulmonary
8
pulmonary disease
8
familial multifocal
4
hyperplasia novel
4
novel splicing
4

Similar Publications

Article Synopsis
  • - This study compares the clinical and radiological features of tuberous sclerosis complex (TSC)-associated lymphangioleiomyomatosis (LAM) and sporadic LAM by reviewing medical records of 90 confirmed cases.
  • - Patients with sporadic LAM showed more clinical symptoms at diagnosis, while TSC-LAM patients presented all four analyzed CT phenotypes, including multiple sclerotic bone lesions and fat-containing lesions in various organs.
  • - The findings indicate significant differences between the two LAM types, which could lead to updated diagnostic criteria, while also highlighting similarities that may help understand the underlying mechanisms of sporadic LAM.
View Article and Find Full Text PDF

Tuberous sclerosis complex: A clinical diagnosis in Ethiopian patients.

Medicine (Baltimore)

February 2024

Department of Radiology, College of Medicine and Health Sciences, University of Gondar, Gondar, Ethiopia.

Article Synopsis
  • - Tuberous sclerosis complex (TSC) is a rare genetic disorder causing nonmalignant growths in various organs, with delayed diagnosis often resulting from a lack of physician familiarity.
  • - Two patients, a 27-year-old man with kidney issues and a 28-year-old woman with seizures, were diagnosed with TSC through established diagnostic criteria and exhibited various skin lesions.
  • - Both patients were advised to receive ongoing care: the male was referred to a chronic illness clinic, while the female was scheduled for dermatological treatment for her facial lesions.
View Article and Find Full Text PDF

Heavy metals even at low concentrations can damage all systems in the human body from the cellular level by causing disruptions in DNA repair mechanisms, cell division and apoptosis. A 49-year-old man who had been working in the sanding and deburring department of a factory producing underground water pipes for 15 years, presented with complaints of effort dyspnea, cough and loss of strength in his left hand. Computed tomography of the lung revealed diffuse micronodular appearance in all zones in both lungs, subpleural nodule and bronchial dilatation.

View Article and Find Full Text PDF

A 52-year-old woman with multifocal micronodular pneumocyte hyperplasia in bilateral lungs and multiple sclerotic bone lesions (SBLs) visited our hospital. Tuberous sclerosis complex (TSC) was suspected but did not meet the diagnostic criteria. Ten years later, at age 62, the patient developed ureteral cancer.

View Article and Find Full Text PDF

Want AI Summaries of new PubMed Abstracts delivered to your In-box?

Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!