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Orbital and Pulmonary Actinomycosis: The First Case Report and Literature Review.

1. Introduction

Actinomyces species are anaerobic Gram-positive filamentous bacteria and ubiquitous in soil and microbiota of both animals and humans. The infection caused by Actinomyces species, actinomycosis, is usually indolent and slowly progressive [1]. There are wide clinical manifestations of actinomycosis depending on the infection site including orocervicofacial, thoracic, abdominal, and pelvic diseases [1]. To date, there have been handful cases of orbital actinomycosis. The source of infection is usually following the head injury or from the infection of adjacent paraorbital organs including paranasal sinus, nose, cavernous sinus, and brain. We report herein a case of a 58-year-old man who presented with chronic progressive course of total ophthalmoparesis in association with productive cough, leading to the diagnosis after extensive investigation.

2. Case Report

A 58-year-old Thai previously healthy male farmer, living in Roi Et Province (Northeast Thailand), was referred to our hospital, King Chulalongkorn Memorial Hospital, due to problems with total ophthalmoparesis and productive cough for 2 months. Two months prior to admission (PTA), he developed productive cough and low-grade fever with no response to many courses of antibiotic treatment. One month PTA, he noted binocular horizontal diplopia of the right eye, before progression to visual loss and ptosis despite antibiotic and steroid treatment. His past medical history was unremarkable except heavy alcoholic drinking.

The right eye examination revealed eyelid swelling, proptosis, marked chemosis, and complete ptosis; light perception of visual acuity; absent afferent pupillary reflex; and optic disk swelling and retinal hemorrhage. Neurological examination revealed right eye total ophthalmoparesis and decreased pinprick sensation of right cranial VI nerve area. Multiple dental caries were noted. Pulmonary examination revealed fine crackles at both the lungs. Other examination was unremarkable.

Complete blood count showed hemoglobin of 9.3g/dL, white blood cells of 25,540 cells/[mm.sup.3] (85.1% neutrophil, 9.8% lymphocyte, and 4.8% monocyte), and platelets of 697,000 cells/[mm.sup.3]. Blood chemistry was normal. Anti-HIV test was negative. Chest X-ray revealed bilateral diffuse reticulonodular and alveolar infiltrations. Orbital computed tomography revealed a 1.5 x 2.9 x 1.5 cm rim-enhancing hypodense lesion at the right orbital apex abutting to the optic nerve sheath complex and right medial rectus muscle (Figure 1).

A diagnosis of orbital and pulmonary infections was made, and emergent anterior orbitotomy was performed and revealed purulent discharge from the orbit. The pus and sputum Gram stain exhibited many Gram-positive filamentous bacilli, but non-acid-fast, on both standard and modified acid-fast bacilli stains. Hence, Actinomyces species was suspected, and Nocardia species could be excluded due to negative results on modified AFB stain. The pus cultures finally grew Actinomyces israelii (Figures 2(a) and 2(b)). Further bacterial identification by base sequencing of 16S ribosomal DNA was A. israelii with 99% identity (NCBI BLAST Search).

He was given penicillin G sodium of 24 million units per day for 6 weeks and amoxicillin of 2 g per day for another 12 months. After 10 days of treatment, there was a much improvement of lung infiltrates on his chest X-ray. The patient was doing well when last seen 1 year after discharge with mild residual impaired ophthalmoparesis and ptosis but still moderate visual impairment.

3. Discussion

To our knowledge, our case is the first report of both orbital and pulmonary actinomycosis. All previously reported cases were only orbital actinomycosis.

A portal of entry to the orbit in our case is probably from the hematogenous spread from the lungs, even though only unilateral orbit is involved. Due to sequence of respiratory and eye symptoms, the imaging reveals that there is no infection of paraorbital organs and presumed aspiration pneumonia from heavy alcohol drinking. In contrast, orbital actinomycosis in all previously reported cases was caused by infection in paraorbital organs or following the injury.

A literature review of all 10 cases (including our case) of orbital actinomycosis is shown in Table 1. The median age is 45 (IQR 37, 56) years with the male:female ratio of 2:1. Europe and Asia are the most common continents (4, 40% each), followed by North America and South America (1, 10% each). Teeth extraction history and chronic steroid use are the most common predisposing factors (4, 40% each), followed by sinusitis (2, 20%) and trauma (1, 10%). The median duration of illness is 21 (IQR 11, 51) days. Ophthalmoparesis and ptosis are the most common presentations (7, 70% each), followed by proptosis (4, 40%) and visual loss (3, 30%). Six (60%) patients are afebrile. On imaging, there are 5 and 3 extraconal and intraconal lesions (1 infraorbital nerve and 1 unknown data). Of 5 extraconal lesions, there are 3 and 2 intradural and extradural lesions. Of 3 intraconal lesions, there are 2 and 1 central and lateral lesions. Of 10 patients (no data in 1 patient), 8 (80%) undergo surgical intervention. Penicillin is the most common prescribed antibiotic (6, 60%), followed by tetracycline and gentamicin (1, 10% each). There is 12% mortality rate. Full recovery and partial recovery are observed in 2 (20%) and 6 (60%) patients (no data in 2 patients).

4. Conclusions

To our knowledge, our case is the first report of both orbital and pulmonary actinomycosis, likely from hematogenous spread from the lungs.

https://doi.org/10.1155/2018/4759807

Conflicts of Interest

The authors declare that there are no conflicts of interest regarding the publication of this paper.

References

[1] T. A. Russo, "Agents of actinomycosis," in Mandell, Douglas, and Bennett's Principles and Practice of Infectious Diseases, J. D. R. Bennett and M. Blaser, Eds., pp. 2863-2873, Elsevier Saunders, Toronto, ON, Canada, 2015.

[2] T. J. A. G. Sullivan and J. E. Wright, "Actinomycosis of the orbit," British Journal of Ophthalmology, vol. 76, no. 8, pp. 505-506, 1992.

[3] S. D. S. O. Nithyanandam, S. S. Rao, R. R. Battu, and S. George, "Rhinoorbitocerebral actinomycosis," Ophthalmic Plastic and Reconstructive Surgery, vol. 17, no. 2, pp. 134-136, 2001.

[4] L. C. L. Pagliani and G. M. Cavallini, "Orbital actinomycosis associated with painful ophthalmoplegia. Actinomycosis of the orbit," Ophthalmologica, vol. 220, no. 3, pp. 201-205, 2006.

[5] W. B. Ransom, "A case of actinomycosis of the orbit, with a summary of seven other cases of actinomycosis," BMJ, vol. 1, no. 1582, pp. 1553-1555, 1896.

[6] G. A. Leahey, "Orbital actinomycosis," Transactions of the American Ophthalmological Society, vol. 30, pp. 457-503, 1932.

[7] V. Hegde, N. Puthran, S. Mahesha, and B. Anupama, "A rare and an unusually delayed presentation of orbital actinomycosis following avulsion injury of the scalp," Indian Journal of Ophthalmology, vol. 58, no. 3, pp. 238-240, 2010.

[8] U. Dhaliwal, V. K. Arora, N. Singh, and A. Bhatia, "Clinical and cytopathologic correlation in chronic inflammations of the orbit and ocular adnexa: a review of 55 cases," Orbit, vol. 23, no. 4, pp. 219-225, 2004.

[9] J. Mandrioli, G. Frank, P. Sola et al., "Tolosa-hunt syndrome due to actinomycosis of the cavernous sinus: the infectious hypothesis revisited," Headache, vol. 44, no. 8, pp. 806-811, 2004.

[10] G. Har-El, D. A. Prager, F. De Soto La Paix, and M. Nash, "Actinomycotic granuloma masquerading as an infraorbital nerve neoplasm," Head & Neck, vol. 12, no. 3, pp. 261-263, 1990.

Paruspak Payoong (iD), Natcha Saetiew, Opass Putcharoen, and Chusana Suankratay (iD)

Division of Infectious Diseases, Department of Medicine, Faculty of Medicine, Chulalongkorn University, Bangkok, Thailand

Correspondence should be addressed to Chusana Suankratay; csuankratay@gmail.com

Received 15 May 2018; Accepted 9 July 2018; Published 26 July 2018

Academic Editor: Paul Horrocks

Caption: Figure 1: Computed tomography showing rim-enhancing hypodense lesion at the right orbital apex abutting to the optic nerve sheath complex and right medial rectus muscle.

Caption: Figure 2: (a) The pus Gram stain exhibiting Gram-positive filamentous bacilli. (b) Molar tooth-like colonies of Actinomyces israelii.
Table 1: A summary of all case reports of orbital actinomycosis.

                       Age/                  Published
Number    References    sex      Country        year

1            [2]       42/F      England        1992
2            [3]       45/M       India         2001
3            [4]       53/F       Italy         2006
4            [5]       32/F      England        1896
5            [6]       54/M    The United       1932
                                 States
6            [7]       20/M       India         2010
7            [8]        NA        India         2004
8            [9]       43/M       Italy         2004
9            [10]      71/M       Spain         1990
10         Our case    58/M       Thai          2017

              Underlying            Presentations,
Number          disease             duration (week)

1            Right macular               Total
                  scar            ophthalmoparesis, 3
2                  No                Proptosis and
                                       ptosis, 2
3             Tolosa-Hunt               Total
              syndrome on        ophthalmoparesis, 1
          methylprednisolone
           and carcinomas of
            the kidney and
                 breast
4                  No                   Total
                                   ophthalmoparesis
                                     progress to
                                    alteration of
                                   consciousness, 6
5                  No                Progressive
                                    right temporal
                                   and jaw pain, 12
6           History of deep        Right eyelid and
           scalp laceration        eyebrow swelling
               for 1 year             and pain, 2
7                  NA               Orbital abscess
8                  No                   Total
                                 ophthalmoparesis, 1
9                  No                Progressive
                                   swelling in the
                                     subcutaneous
                                   tissue overlying
                                 the right maxilla, 4
10                 No                   Total
                                   ophthalmoparesis
                                    and productive
                                       cough, 8

             Infection                      Treatment
Number          site         Surgery        Medication

1              IC (C)          Yes        Metronidazole
                                          and high-dose
                                          intramuscular
                                          penicillin V
                                         with probenecid
2             EC (ID)          Yes         Crystalline
                                            penicillin
                                           for 14 days
                                         followed by oral
                                          tetracycline
                                           for 4 weeks
3              IC (L)          Yes      Benzylpenicillin
                                          potassium and
                                        benzylpenicillin
                                       sodium intravenous
                                        for 42 days then
                                         amoxicillin oral
4             EC (ID)          Yes          Iodide of
                                            potassium
5             EC (ED)          Yes              NA

6             EC (ED)          Yes        IV penicillin
                                          G for 2 weeks
                                            then oral
                                         amoxicillin for
                                             6 months
7         Orbital abscess      NA               NA
8             EC (ID)          No         IV penicillin
                                           G then oral
                                           amoxicillin
9          Infraorbital        Yes              NA
             nerve mass
10             IC (C)          Yes        Penicillin IV
                                           for 6 weeks
                                           followed by
                                        amoxicillin oral
                                            12 months

Number        Outcome

1           Improvement
          after 3 months
            of follow-up
2           Improvement
           after 1 month
            of follow-up
3           Improvement
           after 1 month
            of follow-up
4              Death
5           Death after
            4 months of
              symptom
6          Full recovery
          at 4 months of
             follow-up
7                NA
8          Full recovery
          at 3 months of
             follow-up
9           Improvement
10        Improvement on
           12 months of
             follow-up

M: male, F: female, EC: extraconal, IC: intraconal,
ED: extradural, ID: intradural, C: central, L: lateral,
and IV: intravenous.
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Title Annotation:Case Report
Author:Payoong, Paruspak; Saetiew, Natcha; Putcharoen, Opass; Suankratay, Chusana
Publication:Case Reports in Infectious Diseases
Date:Jan 1, 2018
Words:1658
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