Ramírez-López AB, Zúñiga-Lagunes CG, Martínez-Viveros A, Medina-Torres EA, Murata C, Espinosa-Padilla SE, Lugo-Reyes SO

Language: Spanish
References: 18
Page: 204-214
PDF size: 666.46 Kb.

ABSTRACT

Introduction: Primary immunodeficiencies (PID) are underdiagnosed all around the world, even at tertiary care centers. Antibody defects make the most prevalent defect group, and usually manifest themselves clinically after 6 months of age with recurrent respiratory infections caused by encapsulated bacteria. A number of ear-nose and throat surgeries are indicated in patients with recurrent or complicated respiratory infections, who have not adequately responded to medical therapy. These children who underwent a surgical procedure after a history of recurrent respiratory infections might constitute a high-risk group for PID.
Objetive: To enquire how frequent antibody defects are among children with a history of respiratory infections who underwent any of three otolaryngology surgeries.
Methods: We reviewed the electronic medical records of children who underwent adenoid-tonsillectomy (ATT), endoscopic paranasal sinus drainage (ESD), and tympanic ventilation tube placement (TVT) at our center during 2011-2012, for serum immunoglobulins (IgE, IgG, IgA, IgM) levels.
Results: We found 112 surgical procedures in 87 patients. Of these, the indication for surgery was infectious in 37 (21 male, 1 dead, mean age 7.3 years), more often chronic rhinosinusitis (19/37) and tonsillitis (9/37). The procedures included: 24 ATT, 13 ESD, and 6 TVT, for a total of 43. Eight patients (21.6%) underwent more than one surgery. Serum IgE was found in 27 (72.9%), and “at-least-IgG” in 18 (48.6%). Only 70% of these tests were ordered before surgery. Abnormal results enferincluded: High IgE 10/27, high IgG 8/18, low IgG 1/18, high IgM 3/17, and high IgA 5/17. Two children with known Chronic granulomatous disease (CGD) were identified through this electronic search.
Discussion: We describe 37 patients who underwent ENT surgery for a history of complicated, recurrent or refractory respiratory infections, of whom at least nine were allergic and at least two had PID (CGD). Only 18 of those 37 had IgG measured as part of their workup; an alarmingly low index of suspicion for antibody defects. Previous studies in adults with refractory CRS have found antibody defects. We intend to complete the immunological evaluation of the 37, including for specific antibody deficiency (SAD). “Red flags” such as IgE ›2,000 IU/mL, complicated pneumonia, or Aspergillus sp. culture growth may prove to be useful to detect patients with previously undiagnosed PID.

REFERENCES

1. Ocampo, C. J. & Peters, A. T. Antibody deficiency in chronic rhinosinusitis: Epidemiology and burden of illness. Am. J. Rhinol. Allergy. 2013;27:34–38.

2. Aghamohammadi, A. et al. Immunologic evaluation of patients with recurrent ear, nose, and throat infections. Am. J. Otolaryngol. 2008;29:385–392.

3. Conley, M. E., Notarangelo, L. D., Casanova, J., Ellen, M. & Mec, C. Definition of primary immunodeficiency in 2011 : a ‘trialogue’ among friends. 2011;1238:1–6.

4. Notarangelo, L.D., Hammarström, L. Presentation to European Union (EU) Parliament Scientific and Technological Assessment Unit. in Karolinska Institute, Karolinska Univ. Hosp. Huddinge, Sweden (March, 2004).

5. Dahl, M., Tybjærg-hansen, A. & Schnohr, P. A Population- based Study of Morbidity and Mortality in Mannose-binding Lectin Deficiency. J. Exp. Med. 2004;199:1391–1399.

6. Cunningham-Rundles, C., Sidi, P., Estrella, L. & Doucette, J. Identifying undiagnosed primary immunodeficiency diseases in minority subjects by using computer sorting of diagnosis codes. J. Allergy Clin. Immunol. 2004;113:747–55.

7. Wang, L.-L. et al. Distribution and clinical features of primary immunodeficiency diseases in Chinese children (2004-2009). J. Clin. Immunol. 2011;31:297–308.

8. Fried, A. J. & Bonilla, F. a. Pathogenesis, diagnosis, and management of primary antibody deficiencies and infections. Clin. Microbiol. Rev. 2009;22:396–414.

9. Staines Boone, Tamara, A. et al. Gastric Adenocarcinoma in the Context of X-linked Agammaglobulinemia. J Clin Immunol 2013;10–13. doi:10.1007/s10875-013-9971-5.

10. De Vincentiis, G. C. et al.Otolaryngologic manifestations of pediatric immunodeficiency. Int. J. Pediatr. Otorhinolaryngol. 2009;73:S42–S48.

11. Moteki, H., Yasuo, M., Hamano, H., Uehara, T. & Usami, S. IgG4-related chronic rhinosinusitis: a new clinical entity of nasal disease. Acta Otolaryngol. 2011;131:518–526.

12. Ramos, S. D., Mukerji, S. & Pine, H. S. Tonsillectomy and adenoidectomy. Pediatr. Clin. North Am. 2013;60:793– 807.

13. Alqudah, M., Graham, S. M. & Ballas, Z. K. High prevalence of humoral immunodeficiency patients with refractory chronic rhinosinusitis. Am. J. Rhinol. Allergy. 2010;24:409–412.

14. Frieri, M. Good’s Syndrome, CVID, and Selective Antibody Deficiency in Patients with Chronic Rhinosinusitis. Curr. Allergy Asthma Rep. 2014;14:438: 1-7.

15. Carr, T. F. et al. Characterization of specific antibody deficiency in adults with medically refractory chronic rhinosinusitis. Am. J. Rhinol. Allergy. 2011:25:241–244.

16. May, a., Zielen, S., Von Ilberg, C. & Weber, a. Immunoglobulin deficiency and determination of pneumococcal antibody titers in patients with therapy-refractory recurrent rhinosinusitis. Eur. Arch. Oto-Rhino-Laryngology 1999;256:445–449.

17. Shapiro, G. G., Virant, F. S., Furukawa, C. T., Pierson, W. E. & Bierman, C. W. Immune defects in patients with refractory sinusitis. Pediatrics.1991;87:311–316.

18. Vanlerberghe, L., Joniau, S. & Jorissen, M. The prevalence of humoral immunodeficiency in refractory rhinosinusitis: a retrospective analysis. B-ENT. 2006;2:161–166.