Hypovitaminosis D: a novel finding in primary ciliary dyskinesia

Italian Journal of Pediatrics

Francesca Santamaria²  Silvia Montella²  Laida Lisa Di Micco²  Claudia Mazzarella³  Giuseppe Perruolo³  Rossella Valentino³  Marco Maglione²  Carlo Caffarelli¹  Virginia Mirra² 

[1] Department of Pediatrics, Department of Clinical and Experimental Medicine, Azienda Ospedaliera-Universitaria, University of Parma, Parma, Italy;Department of Translational Medical Sciences, Federico II University, Via Sergio Pansini, Naples, 5-80131, Italy;National Council of Research, Institute of Experimental Endocrinology and Oncology, Naples, Italy

关键词: Quality of life;    Vitamin D;    Primary ciliary dyskinesia;   

Others  :  1133510
DOI  :  10.1186/s13052-015-0119-5

 received in 2014-12-22, accepted in 2015-02-11,  发布年份 2015

【 摘 要 】

Background

A relationship between low levels of serum vitamin D and respiratory infections has been established. No study has examined the frequency and clinical relevance of vitamin D deficiency in patients with primary ciliary dyskinesia (PCD).

Methods

Vitamin D levels were measured in 22 PCD patients (7 females, 10.5 years, range, 2–34 years). In PCD, pulmonary function tests (PFTs), sputum microbiology, self-reported physical activity (PA) level, and quality of life (QoL) by means of the Saint George’s Respiratory Questionnaire (SGRQ), were also assessed.

Results

Seventy-two percent of PCD patients were vitamin-D deficient-to-insufficient and 28% were sufficient. No differences in PFTs parameters were found between vitamin D deficiency-to-insufficiency and sufficiency groups. Patients with vitamin D deficiency-to-insufficiency had significantly higher SGRQ total scores, and thus poorer QoL (p = 0.03). Seventy-nine percent of PCD subjects had limitations in performing vigorous activities, and 53% performed less than 3 hours of PA per week. Vitamin D deficiency-to-insufficiency and sufficiency groups did not show any differences in age at PCD diagnosis or at onset of respiratory symptoms, BMI, atopy, current asthma or bronchiectasis. However, 79% of patients with bronchiectasis had vitamin D deficiency-to-insufficiency. No differences were found in the rate of positive sputum cultures and in the number of antibiotic courses between the two groups.

Conclusions

Hypovitaminosis D is common in PCD patients, and is associated with poorer QoL. We recommend the assessment and treatment of hypovitaminosis D to be included in the routine management of PCD.

【 授权许可】

   
2015 Mirra et al.; licensee BioMed Central.

【 图 表 】

Figure 1.

【 参考文献 】

• [1]Bush A, Hogg C: Primary ciliary dyskinesia: recent advances in epidemiology, diagnosis, management and relationship with the expanding spectrum of ciliopathy. Expert Rev Respir Med 2012, 6:663-682.
• [2]Sagel SD, Davis SD, Campisi P, Dell SD: Update of respiratory tract disease in children with primary ciliary dyskinesia. Proc Am Thorac Soc 2011, 8:438-443.
• [3]Marthin JK, Petersen N, Skovgaard LT, Nielsen KG: Lung function in patients with primary ciliary dyskinesia: a cross-sectional and 3-decade longitudinal study. Am J Respir Crit Care Med 2010, 181:1262-1268.
• [4]Maglione M, Bush A, Nielsen KG, Hogg C, Montella S, Marthin JK, et al.: Multicenter analysis of body mass index, lung function, and sputum microbiology in primary ciliary dyskinesia. Pediatr Pulmonol 2014, 49(Suppl 12):1243-50.
• [5]Peterlik M, Cross HS: Vitamin D and calcium deficits predispose for multiple chronic diseases. Eur J Clin Invest 2005, 35:290-304.
• [6]Holick MF: Vitamin D deficiency. N Engl J Med 2007, 357:266-281.
• [7]Urashima M, Segawa T, Okazaki M, Kurihara M, Wada Y, Ida H: Randomized trial of vitamin D supplementation to prevent seasonal influenza A in schoolchildren. Am J Clin Nutr 2010, 91:1255-1260.
• [8]Camargo CA Jr, Ingham T, Wickens K, Thadhani R, Silvers KM, Epton MJ, et al.: Cord-blood 25-hydroxyvitamin D levels and risk of respiratory infection, wheezing, and asthma. Pediatrics 2011, 127:180-187.
• [9]Camargo CA Jr, Ganmaa D, Frazier AL, Kirchberg FF, Stuart JJ, Kleinman K, et al.: Randomized trial of vitamin D supplementation and risk of acute respiratory infection in Mongolia. Pediatrics 2012, 130:561-567.
• [10]Tran B, Armstrong BK, Ebeling PR, English DR, Kimlin MG, van der Pols JC, et al.: Effect of vitamin D supplementation on antibiotic use: a randomized controlled trial. Am J Clin Nutr 2014, 99:156-161.
• [11]Chalmers JD, McHugh BJ, Docherty C, Govan JR, Hill AT: Vitamin-D deficiency is associated with chronic bacterial colonization and disease severity in bronchiectasis. Thorax 2013, 68:39-47.
• [12]Asher MI, Keil U, Anderson HR, Beasley R, Crane J, Martinez F, et al.: International Study of Asthma and Allergies in Childhood (ISAAC): rationale and methods. Eur Respir J 1995, 8:483-491.
• [13]Renzoni E, Forastiere F, Biggeri A, Viegi G, Bisanti L, Chellini E, et al.: Differences in parental- and self-report of asthma, rhinitis and eczema among Italian adolescents. SIDRIA collaborative group. Studi Italiani sui Disordini Respiratori dell’ Infanzia e l’Ambiente. Eur Respir J 1999, 14:597-604.
• [14]Maglione M, Bush A, Montella S, Mollica C, Manna A, Esposito A, et al.: Progression of lung disease in primary ciliary dyskinesia: is spirometry less accurate than CT? Pediatr Pulmonol 2012, 47:498-504.
• [15]Savastano S, Valentino R, Di Somma C, Orio F, Pivonello C, Passaretti F, et al.: Serum 25-Hydroxyvitamin D Levels, phosphoprotein enriched in diabetes gene product (PED/PEA-15) and leptin-to-adiponectin ratio in women with PCOS. Nutr Metab 2011, 8:84. BioMed Central Full Text
• [16]Miller MR, Hankinson J, Brusasco V, Burgos F, Casaburi R, Coates A, et al.: Standardisation of spirometry. Eur Respir J 2005, 26:319-338.
• [17]Wilson CB, Jones PW, O'Leary CJ, Cole PJ, Wilson R: Validation of the St George’s Respiratory Questionnaire in bronchiectasis. Am J Respir Crit Care Med 1997, 156:536-541.
• [18]Maglione M, Montella S, Mirra V, Bruzzese D, Santamaria F: Long-term Assessment of Quality of Life in Primary Ciliary Dyskinesia: Time for New Tools? Chest 2014, 146:232-233.
• [19]Pifferi M, Bush A, Di Cicco M, Pradal U, Ragazzo V, Macchia P, et al.: Health-related quality of life and unmet needs in patients with primary ciliary dyskinesia. Eur Respir J 2010, 35:787-94.
• [20]Madsen A, Green K, Buchvald F, Hanel B, Nielsen KG: Aerobic fitness in children and young adults with primary ciliary dyskinesia. PLoS One 2013, 8:e71409.
• [21]Baïz N, Dargent-Molina P, Wark JD, Souberbielle JC, Slama R, Annesi-Maesano I, et al.: Gestational exposure to urban air pollution related to a decrease in cord blood vitamin D levels. J Clin Endocrinol Metab 2012, 97:4087-4095.
• [22]Carnevale V, Modoni S, Pileri M, Di Giorgio A, Chiodini I, Minisola S, et al.: Longitudinal evaluation of vitamin D status in healthy subjects from southern Italy: seasonal and gender differences. Osteoporos Int 2001, 12:1026-1030.
• [23]Adami S, Bertoldo F, Braga V, Fracassi E, Gatti D, Gandolini G, et al.: 25-hydroxy vitamin D levels in healthy premenopausal women: association with bone turnover markers and bone mineral density. Bone 2009, 45:423-426.
• [24]Valerio G, Giallauria F, Montella S, Vaino N, Vigorito C, Mirra V, et al.: Cardiopulmonary assessment in primary ciliary dyskinesia. Eur J Clin Invest 2012, 42:617-622.
• [25]Whalley S, McManus IC: Living with primary ciliary dyskinesia: a prospective qualitative study of knowledge sharing, symptom concealment, embarrassment, mistrust, and stigma. BMC Pulm Med 2006, 6:25. BioMed Central Full Text
• [26]Carotenuto M, Esposito M, Di Pasquale F, De Stefano S, Santamaria F: Psychological, cognitive and maternal stress assessment in children with primary ciliary dyskinesia. World J Pediatr 2013, 9:312-317.
• [27]Yim S, Dhawan P, Ragunath C, Christakos S, Diamond G: Induction of cathelicidin in normal and CF bronchial epithelial cells by 1,25- dihydroxyvitamin D(3). J Cyst Fibros 2007, 6:403-410.
• [28]Coussens AK, Wilkinson RJ, Hanifa Y, Nikolayevskyy V, Elkington PT, Islam K, et al.: Vitamin D accelerates resolution of inflammatory responses during tuberculosis treatment. Proc Natl Acad Sci U S A 2012, 109:15449-15454.
• [29]Camargo CA Jr, Rifas-Shiman SL, Litonjua AA, Rich-Edwards JW, Weiss ST, Gold DR, et al.: Maternal intake of vitamin D during pregnancy and risk of recurrent wheeze in children at 3 y of age. Am J Clin Nutr 2007, 85:788-795.
• [30]Laaksi I, Ruohola JP, Tuohimaa P, Auvinen A, Haataja R, Pihlajamäki H, et al.: An association of serum vitamin D concentrations < 40 nmol/L with acute respiratory tract infection in young Finnish men. Am J Clin Nutr 2007, 86:714-717.
• [31]Brehm JM, Celedón JC, Soto-Quiros ME, Avila L, Hunninghake GM, Forno E, et al.: Serum vitamin D levels and markers of severity of childhood asthma in Costa Rica. Am J Respir Crit Care Med 2009, 179:765-771.
• [32]Sutherland ER, Goleva E, Jackson LP, Stevens AD, Leung DY: Vitamin D levels, lung function, and steroid response in adult asthma. Am J Respir Crit Care Med 2010, 181:699-704.
• [33]Carraro S, Giordano G, Reniero F, Carpi D, Stocchero M, Sterk PJ, et al.: Asthma severity in childhood and metabolomic profiling of breath condensate. Allergy 2013, 68:110-117.
• [34]Skaaby T, Husemoen LL, Thuesen BH, Pisinger C, Jørgensen T, Fenger RV, et al.: Vitamin D status and chronic obstructive pulmonary disease: a prospective general population study. PLoS One 2014, 9:e90654.
• [35]Hall WB, Sparks AA, Aris RM: Vitamin D deficiency in cystic fibrosis. Int J Endocrinol 2010, 2010:218691.
• [36]Santamaria F, Montella S, Tiddens HA, Guidi G, Casotti V, Maglione M, et al.: Structural and functional lung disease in primary ciliary dyskinesia. Chest 2008, 134:351-57.
• [37]Montella S, Alving K, Maniscalco M, Sofia M, De Stefano S, Raia V, et al.: Measurement of nasal nitric oxide by hand-held and stationary devices. Eur J Clin Invest 2011, 41:1063-1070.
• [38]Boon M, Meyts I, Proesmans M, Vermeulen FL, Jorissen M, De Boeck K: Diagnostic accuracy of nitric oxide measurements to detect Primary Ciliary Dyskinesia. Eur J Clin Invest 2014, 44(Suppl 5):477-485.
• [39]Santamaria F, De Stefano S, Montella S, Barbarano F, Iacotucci P, Ciccarelli R, et al.: Nasal nitric oxide assessment in primary ciliary dyskinesia using aspiration, exhalation, and humming. Med Sci Monit 2008, 14:80-85.
• [40]Walker WT, Jackson CL, Lackie PM, Hogg C, Lucas JS: Nitric oxide in primary ciliary dyskinesia. Eur Respir J 2012, 40:1024-1032.
• [41]Rockett KA, Brookes R, Udalova I, Vidal V, Hill AV, Kwiatkowski D: 1,25-Dihydroxyvitamin D3 induces nitric oxide synthase and suppresses growth of Mycobacterium tuberculosis in a human macrophage-like cell line. Infect Immun 1998, 66:5314-5321.
• [42]Chang JM, Kuo MC, Kuo HT, Hwang SJ, Tsai JC, Chen HC, et al.: 1-alpha,25-Dihydroxyvitamin D3 regulates inducible nitric oxide synthase messenger RNA expression and nitric oxide release in macrophage-like RAW 264.7 cells. J Lab Clin Med 2004, 143:14-22.
• [43]Bartley J, Garrett J, Grant CC, Camargo CA Jr: Could vitamin D have a potential anti-inflammatory and anti-infective role in bronchiectasis? Curr Infect Dis Rep 2013, 15:148-157.
• [44]Chishimba L, Thickett DR, Stockley RA, Wood AM: The vitamin D axis in the lung: a key role for vitamin D-binding protein. Thorax 2010, 65:456-462.
• [45]Lehouck A, Boonen S, Decramer M, Janssens W: COPD, bone metabolism, and osteoporosis. Chest 2011, 139:648-657.