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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 23  |  Issue : 4  |  Page : 158-160

Vibrio alginolyticus tympanostomy tube otorrhea in a child newly diagnosed with primary ciliary dyskinesia with no seawater exposure


1 College of Medicine, King Saud bin Abdulaziz University for Health Sciences; King Abdullah International Medical Research Center, Riyadh, Saudi Arabia
2 King Abdullah International Medical Research Center; Department of Otorhinolaryngology, King Abdulaziz Medical City, Ministry of the National Guard Health Affairs, Riyadh, Saudi Arabia

Date of Submission29-May-2021
Date of Acceptance26-Jun-2021
Date of Web Publication20-Oct-2021

Correspondence Address:
Dr. Abdulrahman O Alomar
College of Medicine, King Saud bin Abdulaziz University for Health Sciences, Al Hars Al Watani, Ar Rimayah, Riyadh 14611
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sjoh.sjoh_26_21

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  Abstract 


Vibrio alginolyticus is a rare pathogenic organism that has been associated with seawater contact causing gastrointestinal disease in most cases and in a narrower spectrum, otitis media, and externa. Here, we report an 8-year-old male child who presented with tympanostomy tube otorrhea caused by V. alginolyticus although his family reported strict water precaution adherence without any exposure to swimming pools nor seawater. Before this presentation, he had multiple otological and respiratory complaints since his birth, and he was investigated for primary ciliary dyskinesia (PCD), for which nasal biopsy was done and returned negative. However, genetic panel testing was positive and confirmed PCD.

Keywords: Case report, primary ciliary dyskinesia, tympanostomy tube otorrhea, Vibrio alginolyticus


How to cite this article:
Alomar AO, Alsanea M, Almajed A, Alsaab F. Vibrio alginolyticus tympanostomy tube otorrhea in a child newly diagnosed with primary ciliary dyskinesia with no seawater exposure. Saudi J Otorhinolaryngol Head Neck Surg 2021;23:158-60

How to cite this URL:
Alomar AO, Alsanea M, Almajed A, Alsaab F. Vibrio alginolyticus tympanostomy tube otorrhea in a child newly diagnosed with primary ciliary dyskinesia with no seawater exposure. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2021 [cited 2022 Jan 21];23:158-60. Available from: https://www.sjohns.org/text.asp?2021/23/4/158/328726




  Background Top


Primary ciliary dyskinesia (PCD) is a rare inherited autosomal recessive disorder that is characterized by disruptions in the cilia, leading to defective mucus clearance and therefore chronic inflammation.[1] It was known as immotile cilia syndrome back in 1977, yet this name was not a precise description since the affected cilia show some motility.[2] This pathology leads to multiple abnormalities surfacing in patients as infections of the respiratory tract, otitis media with effusion (OME), sinusitis, and rhinitis.[1]

The management of OME unresponsive to medical treatment includes the insertion of pressure-equalizing tubes, the temporary ventilation tube (V-tube), or the permanent T-tube.[3] Common organisms affecting patients as such are Streptococcus pneumoniae,  Moraxella More Details catarrhalis, and Haemophilus influenzae. Vibrio species are a known cause of gastrointestinal infections after ingestion of raw seafood. This is due to the fact that Vibrio is found with high concentrations in warm seawater, especially in the months between May and October.[4],[5] V. alginolyticus is an unusual cause of otitis media that has been reported in patients who recently swam in the sea.[6],[7] In the USA, a 24-year long observational study reported that 96% of 1331 V. alginolyticus infection cases occurred in coastal states.[8] It reported 437 ear infections, only 3% of which were otitis media cases.


  Case Report Top


An 8-year-old Arabic boy with attention-deficit hyperactivity disorder presented to our otolaryngology clinic in Riyadh at the age of 4 years having speech delay and mild hearing loss with flat tympanic membrane. The patient had a history of cough, recurrent OME, recurrent sinusitis, and admission for bronchiolitis during his 1st year of life in 2012. The parent's consanguinity is negative. He has two siblings, one of them is asthmatic. The patient had levocardia and no situs inversus.

During the last 4 years, he underwent a total of five tympanostomy procedures for recurrent OME. The procedures in September 2017, April 2018, December 2018, and August 2019 were to insert a temporary ventilation tube. In March 2020, a permanent T-tube was inserted, and a nasal brush border biopsy was taken because of the suspicion of PCD. The biopsy returned negative. Later, in June, a genetic panel testing was ordered by pulmonology. A pathogenic variant was identified in the RSPH 9 gene confirming the diagnosis of autosomal recessive PCD type 12. The patient had improved hearing after the T-tube with less pain and discharge with only one visit to the emergency department that was in July 2020. The patient was presented with ear purulent discharge and the clinical picture of tympanostomy tube otorrhea. He was managed by ofloxacin 0.3% ear drops and augmentin 90 mg/kg/day for 10 days with no improvement. Ear culture was taken which had heavy growth of V. alginolyticus although the patient was following strict water precautions without any exposure to swimming pools nor seawater. Antibiotic therapy was changed to ciprofloxacin 20 mg/kg/day for 10 days with the continuation of ofloxacin. The patient showed improvement later. The patient now follows up with pulmonology and genetics for PCD and with otolaryngology for his tympanostomy. He has future scheduled tympanometry and audiometry for further evaluation of his status.


  Discussion Top


Recurrent OME and ventilation tube procedures should raise the question of underlying diagnoses such as PCD. A report indicated that 80%–84% of children, aged <11 years, who have PCD have OME.[9] However, only 48% of those between 12 and 16 years had OME. It also showed that more than half of their patients underwent V-tube insertion which improved their hearing significantly. The complications of insertion included otorrhea and persistent perforation. Therefore, these symptoms must be kept in mind for the diagnosis of PCD, in addition to the other respiratory symptoms, especially in younger patients without situs inversus.

Nasal brush biopsy may not be conclusive due to the variants of the disease itself. Takeuchi et al. reviewed 15 patients with PCD.[10] Four (27%) had a negative biopsy yet were confirmed by genetic testing, as in our case. Another study showed that out of 119 confirmed PCD cases, 42 (35%) had a negative transmission electron microscopy result.[11] High-speed video-microscopy analysis, nasal nitric oxide (<233 ppb), and genetic testing had the highest positive results (84%, 84%, and 80%, respectively). The previous results, in addition to our case, necessitate that PCD diagnosis should not be ruled out based on the nasal brush border biopsy result alone. However, it should be further investigated by other means, such as the genetic panel testing. Furthermore, the involvement of other disciplines such as medical genetics, pulmonology, and fertility is useful for diagnosing and managing cases as such.

Riyadh lies at the center of the Arabian Peninsula with the closest seashore being more than 400 km (249 miles) far. Therefore, the growth of V. Alginolyticus in our patient's culture is of utmost interest as the organism is extremely rare in this area, especially with our patient having no travel and water exposure history. Studies in Saudi Arabia have isolated this organism from sea-foods and linked it to intestinal disease, yet, to our knowledge, this might be the first reported case of tympanostomy tube otorrhea caused by V. alginolyticus in the kingdom.[12] The contamination method of our patient remains unknown.


  Conclusion Top


To sum up, recurrent OME and ventilation tube procedures should raise the suspicion of PCD, especially if accompanied by a history of respiratory illness and sinusitis. Diagnosis should not be ruled out with the first negative result as it might be a false negative. Further investigation is encouraged with the engagement of pulmonology, medical genetics, and fertility departments for both diagnosis and management.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Werner C, Onnebrink JG, Omran H. Diagnosis and management of primary ciliary dyskinesia. Cilia 2015;4:2.  Back to cited text no. 1
    
2.
Eliasson R, Mossberg B, Camner P, Afzelius BA. The immotile-cilia syndrome. A congenital ciliary abnormality as an etiologic factor in chronic airway infections and male sterility. N Engl J Med 1977;297:1-6.  Back to cited text no. 2
    
3.
Prulière-Escabasse V, Coste A, Chauvin P, Fauroux B, Tamalet A, Garabedian EN, et al. Otologic features in children with primary ciliary dyskinesia. Arch Otolaryngol Head Neck Surg 2010;136:1121-6.  Back to cited text no. 3
    
4.
Cavallo RA, Stabili L. Presence of vibrios in seawater and Mytilus galloprovincialis (Lam.) from the Mar Piccolo of Taranto (Ionian Sea). Water Res 2002;36:3719-26.  Back to cited text no. 4
    
5.
National Center for Emerging and Zoonotic Infectious Diseases (NCEZID), Division of Foodborne, Waterborne and ED. Vibrio Species Causing Vibriosis. Centers for Disease Control and Prevention; 2019. Available from: https://www.cdc.gov/vibrio/faq.html. [Last accessed on 2020 Sep 05].  Back to cited text no. 5
    
6.
Feingold MH, Kumar ML. Otitis media associated with Vibrio alginolyticus in a child with pressure-equalizing tubes. Pediatr Infect Dis J 2004;23:475-6.  Back to cited text no. 6
    
7.
Schets FM, van den Berg HH, Demeulmeester AA, van Dijk E, Rutjes SA, van Hooijdonk HJ, et al. Vibrio alginolyticus infections in the Netherlands after swimming in the North Sea. Euro Surveill 2006;11:E061109.3.  Back to cited text no. 7
    
8.
Jacobs Slifka KM, Newton AE, Mahon BE. Vibrio alginolyticus infections in the USA, 1988-2012. Epidemiol Infect 2017;145:1491-9.  Back to cited text no. 8
    
9.
Andersen TN, Alanin MC, von Buchwald C, Nielsen LH. A longitudinal evaluation of hearing and ventilation tube insertion in patients with primary ciliary dyskinesia. Int J Pediatr Otorhinolaryngol 2016;89:164-8.  Back to cited text no. 9
    
10.
Takeuchi K, Kitano M, Sakaida H, Usui S, Masuda S, Ogawa S, et al. Analysis of otologic features of patients with primary ciliary dyskinesia. Otol Neurotol 2017;38:e451-6.  Back to cited text no. 10
    
11.
Abitbul R, Amirav I, Blau H, Alkrinawi S, Aviram M, Shoseyov D, et al. Primary ciliary dyskinesia in Israel: Prevalence, clinical features, current diagnosis and management practices. Respir Med 2016;119:41-7.  Back to cited text no. 11
    
12.
Elhadi N, Aljeldah M, Aljindan R. Microbiological contamination of imported frozen fish marketed in Eastern Province of Saudi Arabia. Int Food Res J 2016;26:2723-31.  Back to cited text no. 12
    




 

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