Aggregatibacter aphrophilus chronic lacrimal canaliculitis: A case report

Abstract

Background
Chronic canaliculitis is often misdiagnosed as conjunctivitis, delaying proper documentation and management. Aggregatibacter aphrophillus has not been implicated in chronic canaliculitis.

Case presentation
We report a case of unilateral chronic epiphora associated with chronic lacrimal canaliculitis resistant to prolonged topical antibiotic treatment in a 65-year-old woman without notable medical history. Canaculotomy, curettage with removal of concretions and tubing with silicone stent for six weeks resolved this chronic infection. Culturing lacrimal secretions and concretions yielded Aggregatibacter aphrophilus in pure culture. Histological analyses showed elongated seed clusters surrounded by neutrophils. Fluorescence in Situ Hybridization confirmed the presence of bacteria in two distinctive concretions.

Conclusion
This first documented case of A. aphrophilus chronic lacrimal canaliculitis illustrates that optimal surgical management of chronic lacrimal canaliculitis allows for both accurate microbiological diagnosis and treatment.

Full text

C A S E R E P O R T Open Access
Aggregatibacter aphrophilus chronic lacrimal
canaliculitis: a case report
Marie Boulze-Pankert
1
, Cécile Roux
1
, Vanessa D. Nkamga
2
, Frédérique Gouriet
2
, Marie-Christine Rojat-Habib
3
,
Michel Drancourt
2,4*
and Louis Hoffart
1
Abstract
Background: Chronic canaliculitis is often misdiagnosed as conjunctivitis, delaying proper documentation and
management. Aggregatibacter aphrophillus has not been implicated in chronic canaliculitis.
Case presentation: We report a case of unilateral chronic epiphora associated with chronic lacrimal canaliculitis
resistant to prolonged topical antibiotic treatment in a 65-year-old woman without notable medical history.
Canaculotomy, curettage with removal of concretions and tubing with silicone stent for six weeks resolved this
chronic infection. Culturing lacrimal secretions and concretions yielded Aggregatibacter aphrophilus in pure culture.
Histological analyses showed elongated seed clusters surrounded by neutrophils. Fluorescence in Situ Hybridization
confirmed the presence of bacteria in two distinctive concretions.
Conclusion: This first documented case of A. aphrophilus chronic lacrimal canaliculitis illustrates that optimal
surgical management of chronic lacrimal canaliculitis allows for both accurate microbiological diagnosis and
treatment.
Keywords: Canaliculitis, Aggregatibacter aphrophilus,Haemophilus aphrophilus, diagnosis, Canaculitis, Aggregatibacter
aphrophillus, Canaliculotomy, Case report
Background
Chronic canaliculitis is often misdiagnosed as conjunc-
tivitis, delaying proper management. Its diagnosis should
include appropriate microbiological investigations based
on the analysis of surgical clinical specimens, as treat-
ment should include both surgery of the canaliculus and
pathogen-targeted antibiotic treatment. Based on this
approach, we here report on a case of Aggregatibacter
aphrophilus chronic canaliculitis, firmly diagnosed using
advanced microbiological methods.
Case presentation
A 65-year-old woman was referred to perform a dacryo-
cystorhinostomy for chronic epiphora with mucopuru-
lent secretions resistant to several topical antibiotic
treatments. The patient had no history of lacrimal plug,
palpebral surgery or trauma. This patient had been ini-
tially diagnosed with chronic conjunctivitis and dacryocys-
tis. However, her clinical presentation included a lower
eyelid erythema and a red, pouting punctum expressing a
mucopurulent discharge after bidigital massage (Fig. 1a).
Slit lamp examination showed pericanalicular inflam-
mation without lacrimal sac involvement. Probing and
irrigation showed permeability of the lacrimal drainage
system. Chronic canaliculitis was finally diagnosed and
the patient underwent canaliculotomy under general an-
aesthesia involving a linear incision into the conjunc-
tival side of the canaliculus, curetting of concretions
and tubing with a silicone stent (Mini Monoka silicone
stent, FCI Ophthalmics, Paris, France) for six weeks;
followed by topical dexamethasone 1 mg/mL combined
with tobramycin 0.3 % QID for 15 days. Culture of the
secretions and concretions on 5 %-sheep blood
Colombia agar incubated under a strict anaerobic at-
mosphere for seven days remained sterile but culture
on a chocolate agar (PolyViteX, bioMérieux, Marcy
l’Etoile, France) incubated in a 5 % CO
2
-enriched
* Correspondence: michel.drancourt@univ-amu.fr
2
Fédération de Microbiologie Clinique et Unité des Rickettsies, CNRS UMR
6020, Aix-Marseille Université, IHU Méditerranée Infection, Marseille, France
4
Unité de Recherche sur les Maladies Infectieuses et Tropicales Emergentes,
Faculté de Médecine, 27, Boulevard Jean Moulin, 13385 Marseille Cedex 5,
France
Full list of author information is available at the end of the article
© 2016 The Author(s). Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0
International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and
reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to
the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver
(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
Boulze-Pankert et al. BMC Ophthalmology (2016) 16:132
DOI 10.1186/s12886-016-0312-3
Content courtesy of Springer Nature, terms of use apply. Rights reserved.

atmosphere yielded Aggregatibacter aphrophilius iden-
tified by matrix-assisted laser desorption ionization
time-of-flight mass-spectrometry (MALDI-TOF-MS)
with an identification score of 1.737. Using the disk
diffusion method, the isolate tested susceptible to
amoxicillin (minimum inhibitory concentration (MIC),
0.5 mg/L), ceftriaxone (MIC <2 g/L), gentamicin (MIC,
0.25 mg/L) and rifampicin (MIC <2 g/L). The microbial
community structure of the canaliculitis was studied by
Fluorescence in situ hybridization (FISH) incorporating
probe EUB338 5′-GCTGCCTCCCGTAGGAGT-3′la-
beled with Alexa fluor-546, specific for Eubacteria 16S
rRNA gene and probe ARC915 5′-GTGCTCCC
CCGCCAATTCCT-3’labeled with Alexa fluor-488,
specific for Archaea 16S rRNA gene. FISH revealed
cocci detected by EUB338 probe (red fluorescence),
while the ARC915 probe (green fluorescence) remained
negative (Fig. 1b). Histological analysis after hematoxylin
and eosin staining showed clusters of elongated microor-
ganisms surrounded by neutrophils. After ablation of the
silicone stent at six weeks, the four-month follow-up
showed no sign of infection and the patient was judged
cured.
Conclusions
Chronic lacrimal canaliculitis is rarely detected in clinical
practice, accounting for 2 % of lacrimal duct diseases. This
inflammation is caused by infection or punctual plug in-
sertion. Generally, canaliculitis is a primitive unilateral
condition caused by Streptococcus spp., Staphylococcus
spp. or Actinomyces spp. [1]. In the patient here reported,
A. aphrophilus,formerlyHaemophilus aphrophilus,a
fastidious Gram-negative inhabitant of the oropharyn-
geal microbiota, was detected by FISH in two distinct
concretions, cultured and firmly identified by mass
spectrometry. Additional next-generation sequencing is
a more research-oriented method, which can also be
used in selected cases to disclose microorganisms. Only
four cases of A. aphrophilus ocular infection have been
previously reported [2–4] including two cases of en-
dophthalmitis, one case of trabeculectomy bleb infec-
tion and one cited and as yet undescribed case of
canaliculitis [3]. Other infections mainly include brain
abscess [5] and endocarditis [6].
Topical antibiotics are ineffective for curing chronic
canaliculitis due to chronic colonized concretions [7], as
illustrated by the case here reported in which antibiotics
failed, despite an exquisitely antibiotic-sensitive strain of
A. aphrophilus. We therefore recommend surgical treat-
ment, canaliculotomy with incision of the punctum and
curetting of the concretions, as the standard treatment
of chronic canaliculitis.
Abbreviations
FISH, fluorescence in situ hybridization; MALDI-TOF-MS, matrix-assisted laser
desorption ionization time-of-flight mass-spectrometry; MIC: minimum inhibitory
concentration
Availability of data and materials
The dataset supporting the conclusions of this article is included within
the article.
Authors’contribution
MBR, CR, LH managed the patient. VN, FG and MD performed microbiological
analyses including FISH. MRH performed histological analyses. MBP drafted the
report. All authors wrote the report and approved its final version. Consent to
publication was obtained.
Competing interests
The authors declare that they have no competing interests.
Fig. 1 Chronic Aggregatibacter aphrophilus lacrimal canaliculitis. aPhotography of right eye showing swollen lower canaliculis (arrow), inflamed
conjunctiva and mucopurulent secretions (b) FISH detection of A. aphrophilus in canaliculitis concretions. Fluorescent microscopy revealed
bacteria labeled by EUB338 probe (red fluorescence) (arrow) when combining non-specific DNA label by DAPI (blue fluorescence) and negative
control probe (green fluorescence). Scale bar, 50 microns
Boulze-Pankert et al. BMC Ophthalmology (2016) 16:132 Page 2 of 3
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Consent for publication
Written informed consent was obtained from the patient for publication of
this case and any accompanying images. A copy of the written consent is
available for review by the editor of this journal.
Ethics approval and consent to participate
Not applicable.
Author details
1
Service d’Ophtalmologie, Hôpital de la Timone, Aix-Marseille-Université,
Marseille, France.
2
Fédération de Microbiologie Clinique et Unité des
Rickettsies, CNRS UMR 6020, Aix-Marseille Université, IHU Méditerranée
Infection, Marseille, France.
3
Service d’anatomopathologie, Hôpital de la
Timone, Marseille, France.
4
Unité de Recherche sur les Maladies Infectieuses
et Tropicales Emergentes, Faculté de Médecine, 27, Boulevard Jean Moulin,
13385 Marseille Cedex 5, France.
Received: 31 March 2016 Accepted: 28 July 2016
References
1. Freedman JR, Markert MS, Cohen AJ. Primary and secondary lacrimal
canaliculitis: a review of literature. Surv Ophthalmol. 2011;56:336–47.
2. Macken PL, Boyd SR, Campbell I, Chang D, Rootman DS, Trope GE.
Haemophilus aphrophilus bleb infection after a mitomycin trabeculectomy.
Aust N Z J Ophthalmol. 1995;23:323–5.
3. Huang S-T, Lee H-C, Lee N-Y, Liu K-H, Ko W-C. Clinical characteristics of
invasive Haemophilus aphrophilus infections. J Microbiol Immunol Infect Wei
Mian Yu Gan Ran Za Zhi. 2005;38:271–6.
4. Shum JWH, Tsang FCW, Fung KSC, Li KKW. Presumed Aggregatibacter
aphrophilus endogenous endophthalmitis. Int Ophthalmol. 2015;35:269–73.
5. Maraki S, Papadakis IS, Chronakis E, Panagopoulos D, Vakis A. Aggregatibacter
aphrophilus brain abscess secondary to primary tooth extraction: Case report
and literature review. J Microbiol Immunol Infect. 2016;49:119–22.
6. Nørskov-Lauritsen N. Classification, identification, and clinical significance of
Haemophilus and Aggregatibacter species with host specificity for humans.
Clin Microbiol Rev. 2014;27:214–40.
7. Kaliki S, Ali MJ, Honavar SG, Chandrasekhar G, Naik MN. Primary canaliculitis:
clinical features, microbiological profile, and management outcome.
Ophthal Plast Reconstr Surg. 2012;28:355–60.
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