- Email: [email protected]
‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis
DMB-114916; No of Pages 3 Diagnostic Microbiology and Infectious Disease xxx (xxxx) xxx
Contents lists available at ScienceDirect
Diagnostic Microbiology and Infectious Disease journal homepage: www.elsevier.com/locate/diagmicrobio
‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis Jennifer H. Ku ⁎, Gregory Ranches, Sarah A.R. Siegel, Kevin L. Winthrop Oregon Health & Science University – Portland State University School of Public Health, 3181 SW Sam Jackson Park Road GH104, Portland, OR 97239
a r t i c l e
i n f o
Article history: Received 24 June 2019 Received in revised form 15 September 2019 Accepted 22 September 2019 Available online xxxx Keywords: Atypical mycobacteria Nontuberculous mycobacteria Pulmonary infection Bronchiectasis Kyphoscoliosis Pectus excavatum
a b s t r a c t Pulmonary nontuberculous mycobacterial (NTM) disease occurs frequently in older women, and phenotypes of men with NTM disease are largely undescribed. We conducted a case–control study of 34 men with non-cystic fibrosis pulmonary NTM disease (cases), and three male and female control groups with or without NTM disease. Cases were median 71 years of age (range 30–94) and mostly non-Hispanic white (85.3%). These men had similarly low BMI as their female NTM patient counterparts, which was lower than both healthy men (p b 0.001) and bronchiectatic men without NTM (p = 0.06). Kyphoscoliosis was also more common in cases than healthy men (p= 0.007) or bronchiectatic men without NTM (p = 0.02). Our study was the first study to our knowledge to examine demographic features and phenotypes of men with NTM disease. Larger studies are needed to ascertain whether these phenotypes are characteristic of men with NTM disease. © 2019 Elsevier Inc. All rights reserved.
Nontuberculous mycobacteria (NTM) are ubiquitous environmental organisms that cause chronic, debilitating pulmonary disease in older individuals (Falkinham, 2009; Griffith et al., 2007). Pulmonary NTM disease is increasing in the U.S. with prevalence estimates of 4.7/100,000 in men and 6.4/100,000 in women and an annual increase in incidence by 2.9% in men and 2.6% in women (Cassidy et al., 2009; Henkle et al., 2015; Prevots et al., 2010). “Lady Windermere syndrome” was first described in 6 women with lingular/right middle lobe (RML) disease among 29 pulmonary Mycobacterium avium complex (MAC) cases (Reich and Johnson, 1992). The eponym has then been used to describe older, immunocompetent women with no smoking history or underlying pulmonary disease with RML/lingular MAC infection (Griffith et al., 2007). NTM infection is most commonly caused by MAC and frequently affects post-menopausal women with scoliosis, pectus excavatum without cardiopulmonary disease or smoking history (Griffith et al., 2007). Accordingly, prior reports predominately described such women with RML/lingular bronchiectasis and MAC infection (Bhatt et al., 2009). Pulmonary NTM disease also occurs in men, most often in the setting of an underlying lung disease such as chronic obstructive pulmonary disease (COPD) and bronchiectasis. To our knowledge, men with NTM disease outside these settings have been poorly described, and it is unclear whether these men present similarly to their female counterparts. Therefore, we conducted a case–control study of bronchiectatic men with pulmonary NTM disease seen at Oregon Health & Science ⁎ Corresponding author. Tel.: +1-503-494-8121; fax: +1-503-346-8277. E-mail address: [email protected] (J.H. Ku).
University (OHSU) to describe their demographic and clinical characteristics. We searched OHSU's medical record system to identify men with an ICD-9 code for NTM (031.xx) and bronchiectasis (494.0 or 494.1), excluding cystic fibrosis (277.00 through 277.09 and V77.6). We reviewed each patient and collected data from 34 who met the American Thoracic Society / Infectious Disease Society of America criteria for NTM disease (Griffith et al., 2007). Pectus excavatum and scoliosis/kyphoscoliosis were determined based on the most recent computerized tomography scan using Haller Index (Yang et al., 2011). We collected thoracic and spine phenotype data from three control groups: (1) bronchiectatic women with NTM; (2) healthy men without either NTM or bronchiectasis; and (3) bronchiectatic men without NTM. Bronchiectatic women with NTM disease were identified from OHSU patients enrolled in the Bronchiectasis Research Registry (Aksamit et al., 2017). From this group, one patient was randomly selected at a time to match with a case on age (± 3 years) until we had a sufficient number of controls. Healthy men were identified by reviewing all adult men visiting the OHSU Family Medicine clinic between 2012 and 2017 reverse-chronologically; the first match identified for each case (age ± 3 years) was included. Bronchiectatic men without NTM were identified through an ICD9-search for bronchiectasis (494.0 or 494.1), excluding cystic fibrosis (277.00–277.09 and V77.6) and NTM (031.xx). Each control patient's medical records were reviewed to confirm eligibility and culture negativity for either sputum or bronchioalveolar lavage. Patients with significant comorbidities (e.g. renal failure) or
https://doi.org/10.1016/j.diagmicrobio.2019.114916 0732-8893/© 2019 Elsevier Inc. All rights reserved.
Please cite this article as: J.H. Ku, G. Ranches, S.A.R. Siegel, et al., ‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis, Diagnostic Microbiology and Infectious Disease, https://doi.org/10.1016/j.diagmicrobio.2019.114916
2
J.H. Ku et al. / Diagnostic Microbiology and Infectious Disease xxx (xxxx) xxx
Table 1 Demographic and clinical features of 34 male bronchiectatic patients with pulmonary nontuberculous mycobacterial disease. Characteristics
Median (range) or (%)
Age at the time of data collection (years, if alive at the time of data collection) Deceased at the time of data collection Body mass index at initial consultation (kg/ m2) Ethnicity Non-Hispanic white Asian, Southeast Pacific Islander Native-American or Alaskan Primary species Mycobacterium avium complex Mycobacterium abscessus Mycobacterium goodii Mycobacerium kansasii Mycobacterium neoaurum Comorbidities Chrocnic obstructive pulmonary disease Gastroesophageal reflux disease Autoimmune disease+ Asthma Renal disease Diabetes Liver disease Former smoker Current smoker Location of bronchiectasis Right middle lobe Right lower lobe Left upper lobe Right upper lobe Left lower lobe Lingula
71 (30–94) 8.8 22.7 (17.6–35.3) 85.3 8.8 5.9 82.4 8.8 2.9 2.9 2.9 35.3 29.4 17.6 11.8 5.9 5.9 0 52.9 8.8 41.2 23.5 23.5 23.5 20.6 20.6
+ Crohn's disease (n = 2), polymyalgia rheumatica (n = 1), rheumatoid arthritis (n = 1), sarcoidosis (n = 1), unknown (n = 1).
immunosuppressive conditions were excluded. All data were collected by a trained staff and a pulmonary physician blinded to case status. Descriptive statistics were stratified by smoking status. Differences between groups were assessed by Rank-sum and Chisquare test (or Fisher's exact test); a p-value less than 0.05 was considered statistically significant. Statistical analyses were performed using Stata version 14 (STATA Corp., TX, USA). This study was approved by the OHSU Institutional Review Board. Cases were 34 men with bronchiectasis and pulmonary NTM disease seen at OHSU between 2003 and 2017, who were aged 71 years at median (range 30–94) and mostly non-Hispanic white (85.3%) (Table 1). Cases had a lower BMI [22.7 (range 17.6–35.3)] than both healthy men [28.1 (range 21.2–70.6), p b 0.001] and men without NTM [27.3
(range 18.3–40.3), p b 0.06)]. Otherwise, demographic characteristics were similar across the groups. In cases, 14 (41.2%) had predominant bronchiectasis in the RML and 7 (20.6%) in lingula. Similarly, bronchiectasis was most common in the RML (52.9%) and ligula (44.1%) in the female NTM counterparts. Pectus excavatum was present in 16 cases (47.1%) while present in 26 (76.5%, p = 0.01) in bronchiectatic women with NTM (Table 2). Kyphoscoliosis was significantly more common in cases (61.9%) than in healthy men (41.2%, p = 0.007), or bronchiectatic men without NTM (26.7%, p = 0.02). Of the 34 cases, infiltrates were present in 19 (55.9%) on radiographs, nodules in 18 (52.9%), and cavities in 9 (26.5%). Prior to the initial consultation at OHSU, 17 (50.0%) received antibiotic treatment for NTM disease. Since initial consultation, 25 (73.5%) received antibiotic therapy for a median 90.0 weeks (range 42.3–255.9), most commonly including azithromycin (64.7%) and rifampin (52.9%). Nine (26.5%) used at least one antibiotic for at least 1 week for bronchiectasis exacerbation, and 6 (17.6%) received video-assisted thoracoscopic surgery for NTM disease. Of the 20 who received therapy and had culture data, 7 (35.0%) remained persistently positive, 6 (30.0%) converted from positive to persistent negative either during or after therapy, and 1 (5.0%) stayed negative. The remaining 6 (30.0%) had no cultures after therapy. Gastroesophageal reflux disease (GERD) was more common in patients with kyphoscoliosis (33.3%) than those without (23.1%), although this difference was not statistically significant. Stratification by smoking did not modify the results. Our results indicate that men with pulmonary NTM disease present similar phenotypes to female counterparts with nodular bronchiectatic MAC lung disease. Most interestingly, these men had similarly low BMI as their female counterparts, which was lower than both healthy men and bronchiectatic men without NTM. A lack of adipose tissue, which make adiponectins, has been proposed as a risk factor for both NTM and tuberculosis (Kartalija et al., 2013), and low BMI is associated with tuberculosis reactivation (Yen et al., 2016). Similar to their female NTM counterparts, kyphoscoliosis was more common in male NTM patients, compared to either healthy men or men without NTM. The role of kyphoscoliosis in NTM disease is unknown, although an association with GERD, which is a potential risk factor for NTM, has been speculated (Hosogane et al., 2017). We did not observe significant differences in the proportion of GERD diagnoses between our patient groups however. Our study was the first to our knowledge to examine phenotypes of men with NTM disease and to provide a preliminary observation for this population. Generalizability of our findings is limited by the small sample size and because all our subjects came from a single academic center. Further comparison in larger, real-world studies is needed to better ascertain phenotypes characteristic of men with NTM disease.
Table 2 Phenotypes of male bronchiectatic patients with pulmonary nontuberculous mycobacterial disease and three control groups.
Haller Index (overall) Less than 2 (normal) 2 ≤ and b 3.2 (mild pectus excavatum) 3.2 ≤ and b 3.5 (moderate pectus excavatum) 3.5 or greater (severe pectus excavatum) Kyphoscoliosis (on CT or CXR) Body mass index (kg/cm2)
Male bronchiectatic patients with NTM disease (cases) (n = 34) N (%) or median (range)
Female bronchiectatic patients with NTM disease (n = 34) N (%) or median (range)
Male bronchiectatic patients without Healthy males NTM disease (n = 15) (n = 34) N (%) or median (range) N (%) or median (range)
1.9 (1.5–2.7) 18 (52.9) 16 (47.1)
2.3 (1.4–3.9)⁎ 8 (23.5) 24 (70.6)⁎
1.9 (1.5–3.0) 20 (58.8) 14 (41.2)
2.0 (1.5–2.6) 11 (73.3) 4 (26.7)
0
1 (2.9)
0
0
0
1 (2.9)
0
0
21 (61.8) 22.7 (17.6–35.0)
17 (50.0) 20.9 (15.5–41.3)
14 (41.2)⁎ 28.1 (21.2–70.6)⁎
4 (26.7)⁎ 27.3 (18.4–40.3)
Abbreviations: CT (computed tomography); CXR (chest x-ray); NTM (nontuberculous mycobacteria). ⁎ significantly different from male patients with bronchiectasis and NTM disease.
Please cite this article as: J.H. Ku, G. Ranches, S.A.R. Siegel, et al., ‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis, Diagnostic Microbiology and Infectious Disease, https://doi.org/10.1016/j.diagmicrobio.2019.114916
J.H. Ku et al. / Diagnostic Microbiology and Infectious Disease xxx (xxxx) xxx
References Aksamit TR, O'Donnell AE, Barker A, et al. Adult Patients With Bronchiectasis: A First Look at the US Bronchiectasis Research Registry. Chest 2017;151(5):982–92. Bhatt SP, Nanda S, Kintzer Jr JS. The Lady Windermere syndrome. Prim Care Respir J 2009; 18(4):334–6. Cassidy PM, Hedberg K, Saulson A, McNelly E, Winthrop KL. Nontuberculous mycobacterial disease prevalence and risk factors: a changing epidemiology. Clin infect Dis 2009;49(12):e124–9. Falkinham III JO. Surrounded by mycobacteria: nontuberculous mycobacteria in the human environment. J Appl Microbiol 2009;107(2):356–67. Griffith DE, Aksamit T, Brown-Elliott BA, et al. An official ATS/IDSA statement: diagnosis, treatment, and prevention of nontuberculous mycobacterial diseases. Am J Respir Crit Care Med 2007;175(4):367–416. Henkle E, Hedberg K, Schafer S, Novosad S, Winthrop KL. Population-based Incidence of Pulmonary Nontuberculous Mycobacterial Disease in Oregon 2007 to 2012. Ann Am Thorac Soc 2015;12(5):642–7. Hosogane N, Watanabe K, Yagi M, Kaneko S, Toyama Y, Matsumoto M. Scoliosis is a Risk Factor for Gastroesophageal Reflux Disease in Adult Spinal Deformity. Clin Spine Surg 2017;30(4):E480–4.
3
Kartalija M, Ovrutsky AR, Bryan CL, et al. Patients with nontuberculous mycobacterial lung disease exhibit unique body and immune phenotypes. Am J Respir Crit Care Med 2013;187(2):197–205. Prevots DR, Shaw PA, Strickland D, et al. Nontuberculous mycobacterial lung disease prevalence at four integrated health care delivery systems. Am J Respir Crit Care Med 2010;182(7):970–6. Reich JM, Johnson RE. Mycobacterium avium complex pulmonary disease presenting as an isolated lingular or middle lobe pattern. The Lady Windermere syndrome. Chest 1992;101(6):1605–9. Yang JH, Kasat NS, Suh SW, Kim SY. Improvement in reflux gastroesophagitis in a patient with spinal muscular atrophy after surgical correction of kyphoscoliosis: a case report. Clin Orthop Relat Res 2011;469(12):3501–5. Yen YF, Chuang PH, Yen MY, et al. Association of Body Mass Index With Tuberculosis Mortality: A Population-Based Follow-Up Study. Medicine (Baltimore) 2016;95(1), e2300.
Please cite this article as: J.H. Ku, G. Ranches, S.A.R. Siegel, et al., ‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis, Diagnostic Microbiology and Infectious Disease, https://doi.org/10.1016/j.diagmicrobio.2019.114916
Contents lists available at ScienceDirect
Diagnostic Microbiology and Infectious Disease journal homepage: www.elsevier.com/locate/diagmicrobio
‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis Jennifer H. Ku ⁎, Gregory Ranches, Sarah A.R. Siegel, Kevin L. Winthrop Oregon Health & Science University – Portland State University School of Public Health, 3181 SW Sam Jackson Park Road GH104, Portland, OR 97239
a r t i c l e
i n f o
Article history: Received 24 June 2019 Received in revised form 15 September 2019 Accepted 22 September 2019 Available online xxxx Keywords: Atypical mycobacteria Nontuberculous mycobacteria Pulmonary infection Bronchiectasis Kyphoscoliosis Pectus excavatum
a b s t r a c t Pulmonary nontuberculous mycobacterial (NTM) disease occurs frequently in older women, and phenotypes of men with NTM disease are largely undescribed. We conducted a case–control study of 34 men with non-cystic fibrosis pulmonary NTM disease (cases), and three male and female control groups with or without NTM disease. Cases were median 71 years of age (range 30–94) and mostly non-Hispanic white (85.3%). These men had similarly low BMI as their female NTM patient counterparts, which was lower than both healthy men (p b 0.001) and bronchiectatic men without NTM (p = 0.06). Kyphoscoliosis was also more common in cases than healthy men (p= 0.007) or bronchiectatic men without NTM (p = 0.02). Our study was the first study to our knowledge to examine demographic features and phenotypes of men with NTM disease. Larger studies are needed to ascertain whether these phenotypes are characteristic of men with NTM disease. © 2019 Elsevier Inc. All rights reserved.
Nontuberculous mycobacteria (NTM) are ubiquitous environmental organisms that cause chronic, debilitating pulmonary disease in older individuals (Falkinham, 2009; Griffith et al., 2007). Pulmonary NTM disease is increasing in the U.S. with prevalence estimates of 4.7/100,000 in men and 6.4/100,000 in women and an annual increase in incidence by 2.9% in men and 2.6% in women (Cassidy et al., 2009; Henkle et al., 2015; Prevots et al., 2010). “Lady Windermere syndrome” was first described in 6 women with lingular/right middle lobe (RML) disease among 29 pulmonary Mycobacterium avium complex (MAC) cases (Reich and Johnson, 1992). The eponym has then been used to describe older, immunocompetent women with no smoking history or underlying pulmonary disease with RML/lingular MAC infection (Griffith et al., 2007). NTM infection is most commonly caused by MAC and frequently affects post-menopausal women with scoliosis, pectus excavatum without cardiopulmonary disease or smoking history (Griffith et al., 2007). Accordingly, prior reports predominately described such women with RML/lingular bronchiectasis and MAC infection (Bhatt et al., 2009). Pulmonary NTM disease also occurs in men, most often in the setting of an underlying lung disease such as chronic obstructive pulmonary disease (COPD) and bronchiectasis. To our knowledge, men with NTM disease outside these settings have been poorly described, and it is unclear whether these men present similarly to their female counterparts. Therefore, we conducted a case–control study of bronchiectatic men with pulmonary NTM disease seen at Oregon Health & Science ⁎ Corresponding author. Tel.: +1-503-494-8121; fax: +1-503-346-8277. E-mail address: [email protected] (J.H. Ku).
University (OHSU) to describe their demographic and clinical characteristics. We searched OHSU's medical record system to identify men with an ICD-9 code for NTM (031.xx) and bronchiectasis (494.0 or 494.1), excluding cystic fibrosis (277.00 through 277.09 and V77.6). We reviewed each patient and collected data from 34 who met the American Thoracic Society / Infectious Disease Society of America criteria for NTM disease (Griffith et al., 2007). Pectus excavatum and scoliosis/kyphoscoliosis were determined based on the most recent computerized tomography scan using Haller Index (Yang et al., 2011). We collected thoracic and spine phenotype data from three control groups: (1) bronchiectatic women with NTM; (2) healthy men without either NTM or bronchiectasis; and (3) bronchiectatic men without NTM. Bronchiectatic women with NTM disease were identified from OHSU patients enrolled in the Bronchiectasis Research Registry (Aksamit et al., 2017). From this group, one patient was randomly selected at a time to match with a case on age (± 3 years) until we had a sufficient number of controls. Healthy men were identified by reviewing all adult men visiting the OHSU Family Medicine clinic between 2012 and 2017 reverse-chronologically; the first match identified for each case (age ± 3 years) was included. Bronchiectatic men without NTM were identified through an ICD9-search for bronchiectasis (494.0 or 494.1), excluding cystic fibrosis (277.00–277.09 and V77.6) and NTM (031.xx). Each control patient's medical records were reviewed to confirm eligibility and culture negativity for either sputum or bronchioalveolar lavage. Patients with significant comorbidities (e.g. renal failure) or
https://doi.org/10.1016/j.diagmicrobio.2019.114916 0732-8893/© 2019 Elsevier Inc. All rights reserved.
Please cite this article as: J.H. Ku, G. Ranches, S.A.R. Siegel, et al., ‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis, Diagnostic Microbiology and Infectious Disease, https://doi.org/10.1016/j.diagmicrobio.2019.114916
2
J.H. Ku et al. / Diagnostic Microbiology and Infectious Disease xxx (xxxx) xxx
Table 1 Demographic and clinical features of 34 male bronchiectatic patients with pulmonary nontuberculous mycobacterial disease. Characteristics
Median (range) or (%)
Age at the time of data collection (years, if alive at the time of data collection) Deceased at the time of data collection Body mass index at initial consultation (kg/ m2) Ethnicity Non-Hispanic white Asian, Southeast Pacific Islander Native-American or Alaskan Primary species Mycobacterium avium complex Mycobacterium abscessus Mycobacterium goodii Mycobacerium kansasii Mycobacterium neoaurum Comorbidities Chrocnic obstructive pulmonary disease Gastroesophageal reflux disease Autoimmune disease+ Asthma Renal disease Diabetes Liver disease Former smoker Current smoker Location of bronchiectasis Right middle lobe Right lower lobe Left upper lobe Right upper lobe Left lower lobe Lingula
71 (30–94) 8.8 22.7 (17.6–35.3) 85.3 8.8 5.9 82.4 8.8 2.9 2.9 2.9 35.3 29.4 17.6 11.8 5.9 5.9 0 52.9 8.8 41.2 23.5 23.5 23.5 20.6 20.6
+ Crohn's disease (n = 2), polymyalgia rheumatica (n = 1), rheumatoid arthritis (n = 1), sarcoidosis (n = 1), unknown (n = 1).
immunosuppressive conditions were excluded. All data were collected by a trained staff and a pulmonary physician blinded to case status. Descriptive statistics were stratified by smoking status. Differences between groups were assessed by Rank-sum and Chisquare test (or Fisher's exact test); a p-value less than 0.05 was considered statistically significant. Statistical analyses were performed using Stata version 14 (STATA Corp., TX, USA). This study was approved by the OHSU Institutional Review Board. Cases were 34 men with bronchiectasis and pulmonary NTM disease seen at OHSU between 2003 and 2017, who were aged 71 years at median (range 30–94) and mostly non-Hispanic white (85.3%) (Table 1). Cases had a lower BMI [22.7 (range 17.6–35.3)] than both healthy men [28.1 (range 21.2–70.6), p b 0.001] and men without NTM [27.3
(range 18.3–40.3), p b 0.06)]. Otherwise, demographic characteristics were similar across the groups. In cases, 14 (41.2%) had predominant bronchiectasis in the RML and 7 (20.6%) in lingula. Similarly, bronchiectasis was most common in the RML (52.9%) and ligula (44.1%) in the female NTM counterparts. Pectus excavatum was present in 16 cases (47.1%) while present in 26 (76.5%, p = 0.01) in bronchiectatic women with NTM (Table 2). Kyphoscoliosis was significantly more common in cases (61.9%) than in healthy men (41.2%, p = 0.007), or bronchiectatic men without NTM (26.7%, p = 0.02). Of the 34 cases, infiltrates were present in 19 (55.9%) on radiographs, nodules in 18 (52.9%), and cavities in 9 (26.5%). Prior to the initial consultation at OHSU, 17 (50.0%) received antibiotic treatment for NTM disease. Since initial consultation, 25 (73.5%) received antibiotic therapy for a median 90.0 weeks (range 42.3–255.9), most commonly including azithromycin (64.7%) and rifampin (52.9%). Nine (26.5%) used at least one antibiotic for at least 1 week for bronchiectasis exacerbation, and 6 (17.6%) received video-assisted thoracoscopic surgery for NTM disease. Of the 20 who received therapy and had culture data, 7 (35.0%) remained persistently positive, 6 (30.0%) converted from positive to persistent negative either during or after therapy, and 1 (5.0%) stayed negative. The remaining 6 (30.0%) had no cultures after therapy. Gastroesophageal reflux disease (GERD) was more common in patients with kyphoscoliosis (33.3%) than those without (23.1%), although this difference was not statistically significant. Stratification by smoking did not modify the results. Our results indicate that men with pulmonary NTM disease present similar phenotypes to female counterparts with nodular bronchiectatic MAC lung disease. Most interestingly, these men had similarly low BMI as their female counterparts, which was lower than both healthy men and bronchiectatic men without NTM. A lack of adipose tissue, which make adiponectins, has been proposed as a risk factor for both NTM and tuberculosis (Kartalija et al., 2013), and low BMI is associated with tuberculosis reactivation (Yen et al., 2016). Similar to their female NTM counterparts, kyphoscoliosis was more common in male NTM patients, compared to either healthy men or men without NTM. The role of kyphoscoliosis in NTM disease is unknown, although an association with GERD, which is a potential risk factor for NTM, has been speculated (Hosogane et al., 2017). We did not observe significant differences in the proportion of GERD diagnoses between our patient groups however. Our study was the first to our knowledge to examine phenotypes of men with NTM disease and to provide a preliminary observation for this population. Generalizability of our findings is limited by the small sample size and because all our subjects came from a single academic center. Further comparison in larger, real-world studies is needed to better ascertain phenotypes characteristic of men with NTM disease.
Table 2 Phenotypes of male bronchiectatic patients with pulmonary nontuberculous mycobacterial disease and three control groups.
Haller Index (overall) Less than 2 (normal) 2 ≤ and b 3.2 (mild pectus excavatum) 3.2 ≤ and b 3.5 (moderate pectus excavatum) 3.5 or greater (severe pectus excavatum) Kyphoscoliosis (on CT or CXR) Body mass index (kg/cm2)
Male bronchiectatic patients with NTM disease (cases) (n = 34) N (%) or median (range)
Female bronchiectatic patients with NTM disease (n = 34) N (%) or median (range)
Male bronchiectatic patients without Healthy males NTM disease (n = 15) (n = 34) N (%) or median (range) N (%) or median (range)
1.9 (1.5–2.7) 18 (52.9) 16 (47.1)
2.3 (1.4–3.9)⁎ 8 (23.5) 24 (70.6)⁎
1.9 (1.5–3.0) 20 (58.8) 14 (41.2)
2.0 (1.5–2.6) 11 (73.3) 4 (26.7)
0
1 (2.9)
0
0
0
1 (2.9)
0
0
21 (61.8) 22.7 (17.6–35.0)
17 (50.0) 20.9 (15.5–41.3)
14 (41.2)⁎ 28.1 (21.2–70.6)⁎
4 (26.7)⁎ 27.3 (18.4–40.3)
Abbreviations: CT (computed tomography); CXR (chest x-ray); NTM (nontuberculous mycobacteria). ⁎ significantly different from male patients with bronchiectasis and NTM disease.
Please cite this article as: J.H. Ku, G. Ranches, S.A.R. Siegel, et al., ‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis, Diagnostic Microbiology and Infectious Disease, https://doi.org/10.1016/j.diagmicrobio.2019.114916
J.H. Ku et al. / Diagnostic Microbiology and Infectious Disease xxx (xxxx) xxx
References Aksamit TR, O'Donnell AE, Barker A, et al. Adult Patients With Bronchiectasis: A First Look at the US Bronchiectasis Research Registry. Chest 2017;151(5):982–92. Bhatt SP, Nanda S, Kintzer Jr JS. The Lady Windermere syndrome. Prim Care Respir J 2009; 18(4):334–6. Cassidy PM, Hedberg K, Saulson A, McNelly E, Winthrop KL. Nontuberculous mycobacterial disease prevalence and risk factors: a changing epidemiology. Clin infect Dis 2009;49(12):e124–9. Falkinham III JO. Surrounded by mycobacteria: nontuberculous mycobacteria in the human environment. J Appl Microbiol 2009;107(2):356–67. Griffith DE, Aksamit T, Brown-Elliott BA, et al. An official ATS/IDSA statement: diagnosis, treatment, and prevention of nontuberculous mycobacterial diseases. Am J Respir Crit Care Med 2007;175(4):367–416. Henkle E, Hedberg K, Schafer S, Novosad S, Winthrop KL. Population-based Incidence of Pulmonary Nontuberculous Mycobacterial Disease in Oregon 2007 to 2012. Ann Am Thorac Soc 2015;12(5):642–7. Hosogane N, Watanabe K, Yagi M, Kaneko S, Toyama Y, Matsumoto M. Scoliosis is a Risk Factor for Gastroesophageal Reflux Disease in Adult Spinal Deformity. Clin Spine Surg 2017;30(4):E480–4.
3
Kartalija M, Ovrutsky AR, Bryan CL, et al. Patients with nontuberculous mycobacterial lung disease exhibit unique body and immune phenotypes. Am J Respir Crit Care Med 2013;187(2):197–205. Prevots DR, Shaw PA, Strickland D, et al. Nontuberculous mycobacterial lung disease prevalence at four integrated health care delivery systems. Am J Respir Crit Care Med 2010;182(7):970–6. Reich JM, Johnson RE. Mycobacterium avium complex pulmonary disease presenting as an isolated lingular or middle lobe pattern. The Lady Windermere syndrome. Chest 1992;101(6):1605–9. Yang JH, Kasat NS, Suh SW, Kim SY. Improvement in reflux gastroesophagitis in a patient with spinal muscular atrophy after surgical correction of kyphoscoliosis: a case report. Clin Orthop Relat Res 2011;469(12):3501–5. Yen YF, Chuang PH, Yen MY, et al. Association of Body Mass Index With Tuberculosis Mortality: A Population-Based Follow-Up Study. Medicine (Baltimore) 2016;95(1), e2300.
Please cite this article as: J.H. Ku, G. Ranches, S.A.R. Siegel, et al., ‘Lady Windermere's counterpart? Pulmonary nontuberculous mycobacteria in men with bronchiectasis, Diagnostic Microbiology and Infectious Disease, https://doi.org/10.1016/j.diagmicrobio.2019.114916