Hairspray Inhalation-induced Interstitial Pneumonitis Evaluated by a Transbronchial Lung Cryobiopsy

A 60-year-old Japanese woman was admitted to our hospital with a fever and shortness of breath occurring immediately after using hairspray. Chest high-resolution computed tomography (HRCT) showed ground-glass opacities (GGOs) predominantly distributed around the bronchovascular bundles, and a pathological evaluation by a transbronchial lung cryobiopsy (TBLC) revealed fibrotic non-specific interstitial pneumonia (f-NSIP). Her symptoms disappeared without the use of corticosteroids, and GGOs on HRCT improved markedly over time. This case suggests that a pathological evaluation by a TBLC for lung injury due to inhalation pathogen exposure may provide a more accurate diagnosis and a better understanding of the pathology from bronchial to interstitial lesions than transbronchial lung biopsy.     

Occupational Hypersensitivity Pneumonitis in a Japanese Citrus Farmer

Hypersensitivity pneumonitis (HP) sometimes develops in people working in specific environments. We herein report a case of occupation-related HP in a citrus farmer in Japan. A 66-year-old man developed a fever, dyspnea, and general malaise in March after working near a trash dump filled with moldy tangerines. He presented with leukocytosis, bilateral lung opacities on chest radiographs, and intra-alveolar and interstitial lymphocytic inflammation with fibrotic change on a lung biopsy. His symptoms disappeared after admission and recurred on a revisit to the workplace. Fungal culture and a mycobiome analysis using next-generation sequencing suggested an association with exposure to Penicillium digitatum.     

The Usefulness of a Transbronchial Lung Cryobiopsy for Diffuse Bronchiolitis

We herein report four cases of diffuse bronchiolitis proven by a transbronchial lung cryobiopsy (TBLC). Based on various aspects, including the pathological findings, we definitively diagnosed two patients with diffuse panbronchiolitis (DPB) and the other two with primary ciliary dyskinesia (PCD). One of the DPB patients had more severe peribronchiolar fibrosis than the other, and the disease course was refractory to macrolide therapy. One of the PCD patients was additionally diagnosed with combined constrictive bronchiolitis. This report highlights the importance of a TBLC in the differentiation of bronchiolitis, suggesting its utility for helping pulmonologists formulate a treatment strategy.     

Hypersensitivity Pneumonitis Caused by Exposure to a Gray Parrot (Psittacus erithacus)

A 73-year-old woman complaining of cough and dyspnea was admitted to our hospital. High-resolution computed tomography chest revealed patchy ground-glass attenuation in the upper lung field. The patient suffered an asthma attack and was diagnosed with allergic pneumonitis; prednisolone was administered for treatment. Bird-related hypersensitivity pneumonitis was suspected, as she had a gray parrot (Psittacus erithacus) and a budgerigar (Melopsittacus undulatus) at home. An immunoblotting analysis with the patient''s serum demonstrated IgG-binding fractions to the gray parrot''s feathers only; no binding was noted with the budgerigar antigens. The patient was conclusively diagnosed with hypersensitivity pneumonitis related to exposure to a gray parrot.     

Miliary tuberculosis: clinical manifestations, diagnosis and outcome in 38 adults

OBJECTIVE: The aim of the study was to determine the clinical, radiographic and laboratory characteristics, diagnostic methods, and prognostic variables in patients with miliary tuberculosis (TB). METHODOLOGY: The records of 38 patients (15 male, 23 female; mean age 41 years, range 16-76 years) with miliary TB from 1978 to 1998 were analyzed. Patients were evaluated also as to whether they presented with a fever of unknown origin (FUO). Criteria for the diagnosis of miliary TB were (i) miliary pattern on chest X-ray or (ii) biopsy or autopsy evidence of miliary organ involvement. Paraffin-embedded tissues with granulomata (n = 15) were re-evaluated for the presence of Mycobacterium tuberculosis DNA by polymerase chain reaction (PCR). RESULTS: Predisposing conditions were present in 24% of the patients. The findings were fever, weakness, night sweats, anorexia/weight loss (100% for each), hepatomegaly (37%), splenomegaly (32%), choroidal tubercles (13%), neck stiffness (11%), altered mental status (8%), anaemia (76%), leukopenia (26%), thrombocytopenia (16%), lymphopenia (76%), pancytopenia (8%) and hypertransaminasemia (55%). Eighteen patients (47%) met the criteria for a FUO. Miliary infiltrates were found on chest X-rays of 32 of 38 cases (84%). In six cases without miliary infiltrates, the diagnosis was made by laparotomy in four cases, and autopsy in two cases. Tuberculin skin test was positive in 32% of cases. Acid-fast bacilli were demonstrated in 37% (16/43), and cultures for M. tuberculosis were positive in 90% (9/10) of tested specimens (predominantly sputum and bronchial lavage). Granulomas were found in 85% (11/13) of lung, 100% (15/15) of liver, and 56% (9/16) of bone marrow tissue specimens. Acid-fast bacilli staining was negative in all (0/21), while PCR was positive in 47% (7/15) of specimens with granulomata. Mortality was 18%. Stepwise logistic regression identified male sex (P = 0.005), non-typical miliary pattern (P = 0.015), altered mental status (P = 0.002) and failure to treat for TB (P = 0.00001) as independent predictors of mortality. CONCLUSIONS: Miliary infiltrates on chest X-ray or FUO should raise the possibility of miliary TB. Therapy should be administered urgently to prevent an otherwise fatal outcome.     

Quality of transbronchial biopsy with large forceps compared to cryobiopsy: a randomized controlled,single blinded live animal study

BackgroundTransbronchial forceps biopsy is the widely accepted modality for obtaining tissue specimens for the evaluation of unexplained lung parenchymal abnormalities. However, cryoprobe biopsy provides large specimen sizes and higher yield performance. Utilization of cryoprobe biopsy remains limited by its need to be performed under rigid bronchoscopy and subsequent required operator expertise. We evaluated whether a larger, 2.8 mm forceps could be utilized for parenchymal biopsies. A larger size would surrogate the cryoprobe’s large sample size and forceps mechanism to obviate the need for rigid bronchoscopy and its requirement for removing the sample en bloc.MethodsThis prospective, randomized controlled, single-blinded porcine study compared a 1.9 mm cryoprobe, a 2.4 mm cryoprobe, and a 2.8 mm forceps. Assessment of histopathologic quality, sample quality and surface area, attempts to retrieve specimen samples, fluoroscopy activation time, overall procedural time, and complications were compared.ResultsAlthough cryoprobe yielded larger specimens, there was no statistical difference amongst all tools with respect to alveolar tissue surface area. There was bleeding on all cryoprobe biopsies. No bleeding was observed with forceps. Out of 32 potential combinations of interventions for bleeding control, 18 (56.3%) were made. There was no significant difference in sample quality between all three modalities. There was one pneumothorax in the forceps arm.ConclusionsLarge forceps (LF) biopsy is a feasible technique while providing high diagnostic yield without the need for advanced therapeutic tools. Human studies are needed to further corroborate this technique.     

Miliary tuberculosis and necrotizing tuberculous fasciitis – An unusual coexistence in a rheumatoid arthritis patient

We report a case of a 65‐year‐old Korean female patient with rheumatoid arthritis, who presented with extensive necrotizing fasciitis of the gluteus muscles, as an unusual initial manifestation of miliary tuberculosis. The patient had been previously treated with conventional disease‐modifying antirheumatic drugs and low‐dose steroids for 7 years. However, she recently developed fever, warmth and painful swelling in her right buttock. Magnetic resonance imaging indicated necrotizing fasciitis of the gluteus muscles and a fasciectomy specimen revealed a Mycobacterium tuberculosis infection. Two weeks after a fasciectomy, miliary tuberculosis of the lung was diagnosed by high resolution chest computed tomography. Soft tissue infection due to M. tuberculosis should be included as a differential diagnosis in the immunocompromised host. Clinicians should be alert to the possibility of miliary tuberculosis even in the absence of respiratory symptoms and normal chest radiograph.     

An Open-label Study With Pirfenidone on Chronic Hypersensitivity Pneumonitis

BackgroundChronic hypersensitivity pneumonitis (cHP) represents a severe lung disease often evolving to fibrosis with the subsequent destruction of the lung parenchyma. There are no approved therapies with confirmed efficacy to deal with this disease.MethodsWe performed an open-label, proof of concept study, to evaluate the efficacy and safety of pirfenidone added to immunosuppressive drugs on the treatment of cHP. We included 22 patients assigned to two groups: Group 1, nine patients that received prednisone plus azathioprine and Group 2, thirteen patients, received prednisone plus azathioprine and pirfenidone (ClinicalTrials.gov identifier NCT02496182). There were no significant imbalances in clinically relevant baseline characteristics between two study groups.ResultsAfter 1 year of treatment, inclusion of pirfenidone was not associated with improved forced vital capacity (primary end-point). A not significant tendency to show higher improvement of diffusion capacity of the lung for carbon monoxide (D_LCO) was observed in the group receiving pirfenidone (p = 0.06). Likewise, a significant improvement in the total score on the SGRQ was found in the group 2 (p = 0.02) without differences in other two questionnaires related to quality of life (ATAQ-IPF and EQ-5D-3L). HRCT showed a decrease of the ground glass attenuation without changes in the fibrotic lesions and without differences between both groups.ConclusionsThese findings suggest that the addition of pirfenidone to the anti-inflammatory treatment in patients with chronic HP may improve the outcome with acceptable safety profile. However, prospective randomized double-blind, placebo-controlled trials in largest cohorts are needed to validate its efficacy.     

Pulmonary tuberculosis associated acute fibrinous and organizing pneumonia: A case report and literature review

Background

Acute fibrinous and organizing pneumonia (AFOP) is a rare histological interstitial pneumonia pattern characterized by patches of “fibrin balls” distributed within the alveoli and organizing pneumonia. Currently, there is no consensus on the diagnosis and treatment of this disease.

Methods

We present the case of a 44-year-old male with AFOP secondary to Mycobacterium tuberculosis. We have further reviewed organizing pneumonia (OP) and AFOP caused by tuberculosis.

Conclusion

Tuberculosis secondary to OP or AFOP is rare and challenging to diagnose. We need to constantly adjust the treatment plan based on the patient's symptoms, test results, and response to treatment in order to arrive at an accurate diagnosis and maximize treatment efficacy.     

间质性肺疾病患者行不同活检方式后出血风险及其出血严重程度的相关因素研究

背景现有证据表明,经支气管冷冻肺活检(TBCB)在间质性肺疾病(ILD)诊断中极具潜力,但不同活检方式的诊断效能及安全性鲜见报道。目的比较ILD患者行经支气管镜钳夹活检(TBFB)、TBCB后出血风险及其出血严重程度的相关因素。方法选取2015年3月至2019年2月在湖北医药学院附属太和医院呼吸与危重症医学科就诊的ILD患者729例,排除临床资料不完整的11例患者,最终纳入718例患者,将其随机分为TBFB组和TBCB组,每组359例。TBFB组患者经支气管钳抽取肺组织并进行活检,TBCB组患者经支气管镜低温冷冻抽取肺组织并进行活检。比较两组患者临床资料、肺组织活检情况、术后出血发生率、术后出血严重程度及气胸发生率,TBFB组和TBCB组出血患者出血严重程度的影响因素分析采用单因素分析。结果TBCB组患者凝血酶原时间长于TBFB组,取样数量少于TBFB组,多肺叶活检者所占比例低于TBFB组(P<0.05)。TBCB组患者术后出血发生率高于TBFB组,出血严重程度重于TBFB组(P<0.05)。在TBFB组中,中重度出血患者中服用阿司匹林者所占比例低于轻度出血患者(P<0.05)。在TBCB组中,中重度出血患者中年龄≥65岁、服用阿司匹林者所占比例高于轻度出血患者(P<0.05)。TBCB组患者气胸发生率为6.13%(22/359),高于TBFB组患者的1.67%(6/359)(P<0.05)。结论与TBFB相比,ILD患者行TBCB后出血和气胸发生风险更高;服用阿司匹林可能与ILD患者行TBFB后出血严重程度相关,年龄≥65岁、服用阿司匹林可能与ILD患者行TBFB后出血严重程度相关。     

Efficacy and safety of cryobiopsy versus forceps biopsy for interstitial lung diseases and lung tumours: A systematic review and meta‐analysis

Forceps biopsy (FB) is the most commonly used diagnostic tool for lung pathologies. FB is associated with a high diagnostic failure rate. Cryobiopsy (CB) is a novel technique providing a larger specimen size, few artefacts, more alveolar parts and superior diagnostic yield. CB, however, has drawbacks such as higher bleeding and pneumothorax rate. We conducted a meta‐analysis to investigate the specimen area, diagnostic rate and bleeding severity in CB versus FB in interstitial lung diseases (ILDs) and lung tumours. A systematic literature search of PUBMED, BIOSIS PREVIEW and OVID databases was conducted using specific search terms. Eligible studies including RCTs and non‐RCTs comparing cryobiopsy/cryotransbronchial biopsy (CB/CTBB) and forceps biopsy/forceps transbronchial biopsy (FB/FTBB) for specimen area, diagnostic rate and bleeding rate in ILDs and lung tumours were analysed. Two reviewers independently extracted data and evaluated the quality of the studies. Eight studies involving 916 patients were analysed. Specimen area (mm²) was significantly larger in CB/CTBB than FB/FTBB (standard mean difference = 1.21, 95% confidence interval (0.94, 1.48), P < 0.00001). The diagnostic rate was significantly higher in CB/CTBB than FB/FTBB (Risk ratio 1.36, 95% confidence interval (1.16, 1.59), P = 0.0002). Three studies compared the bleeding severity with only one showing significantly more bleeding in CB. Cryobiopsy/cryotransbronchial shows superiority to FB/FTBB for specimen area and diagnostic rate. CB/CTBB has better efficacy over FB/FTBB.     

Forced Oscillation Technique and Small Airway Involvement in Chronic Hypersensitivity Pneumonitis

ObjectiveHypersensitivity pneumonitis (HP) is an interstitial lung disease caused by the inhalation of specific organic antigens or low-molecular weight substances in genetically susceptible individuals. Although small airway involvement is prominent in patients with chronic HP, conventional pulmonary function tests (PFTs) are relatively insensitive to identify it. Thus, the authors aimed to evaluate resistance (R5) and reactance (X5) values at 5 Hz on inspiration, expiration, and whole breath, as well as small airway resistance (R_5–19) values using a forced oscillation technique (FOT) in patients with chronic HP, and their responses after bronchodilator. In addition, R5 and X5 values according to the presence or absence of mosaic attenuation on computed tomography (CT) were compared.MethodsPFTs with plethysmography, diffusing capacity of the lungs for carbon monoxide (DL_CO) and FOT measurements were performed pre-bronchodilator and post-bronchodilator. High-resolution CT was performed at the same visit, and classified according to the presence or absence of mosaic attenuation. R5 and X5 values were then compared according to the presence or absence of mosaic attenuation on CT.ResultsTwenty-eight patients with chronic HP (57.1% female; mean age, 56 ± 11.5 years; mean forced vital capacity 57 ± 17% predicted) were evaluated. All patients had low X5 values, reflecting lower lung compliance, and only three (8%) demonstrated elevated R5 (whole-breath) values. No patients exhibited bronchodilator response in R5, X5 and R_5–19 values. In patients who exhibited greater extension of mosaic attenuation (n = 11), R5 and X5 values could not discriminate those with a greater presence of these areas on CT.ConclusionsThe results suggest that FOT does not help to additionally characterise concomitant small airway involvement in patients with chronic fibrotic HP who demonstrate restrictive ventilatory pattern in conventional PFTs. Nevertheless, FOT appeared to better characterise decreased lung compliance due to fibrosis through X5. Bronchodilator therapy did not appear to induce an acute response in chronic HP patients with restrictive disease. The precise role of FOT in subacute HP and obstructive chronic HP, therefore, must be evaluated.     

Delayed-onset Organizing Pneumonia Emerging after Recovery from Coronavirus Disease 2019: A Report of Three Cases Diagnosed Using Transbronchial Cryobiopsy and a Review of the Literature

We present three cases with an atypical clinical course of organizing pneumonia (OP) secondary to coronavirus disease 2019 (COVID-19). Three patients were discharged with satisfactory improvement after standard steroid therapy for COVID-19. Shortly after the completion of treatment, the patients experienced a flare-up of symptoms. Imaging results showed new lesions in the lungs. Transbronchial lung cryobiopsy showed histological findings consistent with OP in all cases. Steroids were administered, and a good therapeutic response was observed. This report is the first to describe pathologically confirmed OP that developed after recovery from COVID-19. Careful follow-up is advisable for patients who have recovered from COVID-19.     

Mediastinal Lymphadenopathy: Melioidosis Mimicking Tuberculosis

Melioidosis has protean manifestations and often mimics other disease processes. We present a case of a gentleman presenting with chronic cough whose initial radiographic findings of a cavitatory lung lesion and mediastinal lymphadenopathy were suggestive of tuberculosis. This case highlights the important role that bronchoscopy and endobronchial ultrasound can play in the diagnosis of melioidosis in patients presenting with mediastinal lymphadenopathy whose initial microbiological findings from sputum are negative for tuberculosis.     

Hypersensitivity pneumonitis due to unclean continuous positive airway pressure equipment

Hypersensitivity pneumonitis is a potentially fatal immunological lung disease caused by occupational or environmental exposure to specific antigens. Here, we report on an obstructive sleep apnea patient whose hypersensitivity pneumonitis was aggravated because of the use of unclean continuous positive airway pressure equipment. This report shows that careful history taking is important when diagnosing hypersensitivity pneumonitis. Sleep specialists should be aware of the risks related to unclean continuous positive airway pressure equipment use, and obstructive sleep apnea patients should be instructed and monitored in the regular cleaning of their equipment.     

Sauna lung: Hypersensitivity pneumonitis due to Exophiala jeanselmei

A 55‐year‐old man developed progressive cough and dyspnoea after regular attendance at a public steam bath. Hypoxaemia, diffuse pulmonary infiltrates and a predominance of lymphocytes with an increased percentage of CD8+ T cells in his bronchoalveolar lavage fluid suggested hypersensitivity pneumonitis. Microbial cultures from the steam bath room and tank identified Exophiala jeanselmei. Immunoblotting assays from the patient's serum confirmed the major antigenic stimulus. The patient recovered fully after systemic corticosteroid treatment and cessation of further exposure.