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"Journal of Clinical and Diagnostic Research is at present a well-known Indian originated scientific journal which started with a humble beginning. I have been associated with this journal since many years. I appreciate the Editor, Dr. Hemant Jain, for his constant effort in bringing up this journal to the present status right from the scratch. The journal is multidisciplinary. It encourages in publishing the scientific articles from postgraduates and also the beginners who start their career. At the same time the journal also caters for the high quality articles from specialty and super-specialty researchers. Hence it provides a platform for the scientist and researchers to publish. The other aspect of it is, the readers get the information regarding the most recent developments in science which can be used for teaching, research, treating patients and to some extent take preventive measures against certain diseases. The journal is contributing immensely to the society at national and international level."



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Lucknow
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On Aug 2018




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Dr. Arundhathi. S
MBBS, MD (Pathology),
Sanjay Gandhi institute of trauma and orthopedics,
Bengaluru.
On Aug 2018




Dr. Mamta Gupta,
"It gives me great pleasure to be associated with JCDR, since last 2-3 years. Since then I have authored, co-authored and reviewed about 25 articles in JCDR. I thank JCDR for giving me an opportunity to improve my own skills as an author and a reviewer.
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Reviewing articles is no less a pain staking process and requires in depth perception, knowledge about the topic for review. It requires time and concentration, yet I enjoy doing it. The JCDR website especially for the reviewers is quite user friendly. My suggestions for improving the journal is, more strict review process, so that only high quality articles are published. I find a a good number of articles in Obst. Gynae, hence, a new journal for this specialty titled JCDR-OG can be started. May be a bimonthly or quarterly publication to begin with. Only selected articles should find a place in it.
An yearly reward for the best article authored can also incentivize the authors. Though the process of finding the best article will be not be very easy. I do not know how reviewing process can be improved. If an article is being reviewed by two reviewers, then opinion of one can be communicated to the other or the final opinion of the editor can be communicated to the reviewer if requested for. This will help one’s reviewing skills.
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Dr. Mamta Gupta
Consultant
(Ex HOD Obs &Gynae, Hindu Rao Hospital and associated NDMC Medical College, Delhi)
Aug 2018




Dr. Rajendra Kumar Ghritlaharey

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Every one of us: authors, reviewers, editors, and publisher are responsible for enhancing the stature of the journal. I wish for a great success for JCDR."



Thanking you
With sincere regards
Dr. Rajendra Kumar Ghritlaharey, M.S., M. Ch., FAIS
Associate Professor,
Department of Paediatric Surgery, Gandhi Medical College & Associated
Kamla Nehru & Hamidia Hospitals Bhopal, Madhya Pradesh 462 001 (India)
E-mail: drrajendrak1@rediffmail.com
On May 11,2011




Dr. Shankar P.R.

"On looking back through my Gmail archives after being requested by the journal to write a short editorial about my experiences of publishing with the Journal of Clinical and Diagnostic Research (JCDR), I came across an e-mail from Dr. Hemant Jain, Editor, in March 2007, which introduced the new electronic journal. The main features of the journal which were outlined in the e-mail were extensive author support, cash rewards, the peer review process, and other salient features of the journal.
Over a span of over four years, we (I and my colleagues) have published around 25 articles in the journal. In this editorial, I plan to briefly discuss my experiences of publishing with JCDR and the strengths of the journal and to finally address the areas for improvement.
My experiences of publishing with JCDR: Overall, my experiences of publishing withJCDR have been positive. The best point about the journal is that it responds to queries from the author. This may seem to be simple and not too much to ask for, but unfortunately, many journals in the subcontinent and from many developing countries do not respond or they respond with a long delay to the queries from the authors 1. The reasons could be many, including lack of optimal secretarial and other support. Another problem with many journals is the slowness of the review process. Editorial processing and peer review can take anywhere between a year to two years with some journals. Also, some journals do not keep the contributors informed about the progress of the review process. Due to the long review process, the articles can lose their relevance and topicality. A major benefit with JCDR is the timeliness and promptness of its response. In Dr Jain's e-mail which was sent to me in 2007, before the introduction of the Pre-publishing system, he had stated that he had received my submission and that he would get back to me within seven days and he did!
Most of the manuscripts are published within 3 to 4 months of their submission if they are found to be suitable after the review process. JCDR is published bimonthly and the accepted articles were usually published in the next issue. Recently, due to the increased volume of the submissions, the review process has become slower and it ?? Section can take from 4 to 6 months for the articles to be reviewed. The journal has an extensive author support system and it has recently introduced a paid expedited review process. The journal also mentions the average time for processing the manuscript under different submission systems - regular submission and expedited review.
Strengths of the journal: The journal has an online first facility in which the accepted manuscripts may be published on the website before being included in a regular issue of the journal. This cuts down the time between their acceptance and the publication. The journal is indexed in many databases, though not in PubMed. The editorial board should now take steps to index the journal in PubMed. The journal has a system of notifying readers through e-mail when a new issue is released. Also, the articles are available in both the HTML and the PDF formats. I especially like the new and colorful page format of the journal. Also, the access statistics of the articles are available. The prepublication and the manuscript tracking system are also helpful for the authors.
Areas for improvement: In certain cases, I felt that the peer review process of the manuscripts was not up to international standards and that it should be strengthened. Also, the number of manuscripts in an issue is high and it may be difficult for readers to go through all of them. The journal can consider tightening of the peer review process and increasing the quality standards for the acceptance of the manuscripts. I faced occasional problems with the online manuscript submission (Pre-publishing) system, which have to be addressed.
Overall, the publishing process with JCDR has been smooth, quick and relatively hassle free and I can recommend other authors to consider the journal as an outlet for their work."



Dr. P. Ravi Shankar
KIST Medical College, P.O. Box 14142, Kathmandu, Nepal.
E-mail: ravi.dr.shankar@gmail.com
On April 2011
Anuradha

Dear team JCDR, I would like to thank you for the very professional and polite service provided by everyone at JCDR. While i have been in the field of writing and editing for sometime, this has been my first attempt in publishing a scientific paper.Thank you for hand-holding me through the process.


Dr. Anuradha
E-mail: anuradha2nittur@gmail.com
On Jan 2020

Important Notice

Case report
Year : 2022 | Month : May | Volume : 16 | Issue : 5 | Page : ZD07 - ZD11 Full Version

Recurrent Botryomycosis and Management Strategy with a Follow-up of Four Years: A Case Report


Published: May 1, 2022 | DOI: https://doi.org/10.7860/JCDR/2022/53186.16318
PS Gopinath Thilak, Sharanyamenon Radhakrishnan

1. Reader, Department of Oral and Maxillofacial Surgery, Nitte (Deemed to be University), Ab Shetty Memorial Institute of Dental Sciences (Absmids), Mangalore, Karnataka, India. 2. Postgraduate Student, Department of Oral and Maxillofacial Surgery, Nitte (Deemed to be University), Ab Shetty Memorial Institute of Dental Sciences (Absmids), Mangalore, Karnataka, India.

Correspondence Address :
Sharanya Menon Radhakrishnan,
NITTE (Deemed to be University) Department of Oral and Maxillofacial Surgery AB Shetty Memorial Institute of Dental Sciences, Mangalore, Karnataka, Karnataka, India.

Abstract

Cervicofacial botryomycosis is a chronic, granulomatous suppurative infection with varied presentations in the head and neck region. Since the discovery of this entity, treatment has been elusive without evidence of complete resolution of the lesion with a long period of follow-up. Here the authors report a case of cervicofacial botryomycosis with oral manifestations in a 37-year-old male patient with a history of trauma, for whom the authors devised a treatment plan tailored to the patient's microbiological profile after culture and sensitivity testing in addition to systemic and local antibiotic therapy and heat therapy which showed drastic regression of the lesion and an improved mouth opening. The patient has been under follow-up for the past four years without signs of recurrence. Botryomycosis is an aggressive lesion where a surgery-only approach is usually futile and requires further intervention due to recurrences. The present experience suggests surgical debridement along with antibiotic and local heat therapy to be effective in managing this usually recurring lesion.

Keywords

Bacterial pseudomycosis, Cutaneous botryomycosis, Granular bacteriosis, Granulomatous infections, Streptococci viridans

Case Report

A 37-year-old male patient reported to our Outpatient Department (OPD) with complaints of persistent cutaneous eruptions on the left side of the cheek for the past several years. There was no associated history of pain in the region; however, constant ooze from the region which was colourless and odourless with a progressive decrease in the mouth opening and difficulty in eating was noticed by the patient. On anamnesis, the patient gave a history of trauma to the same site about seven years ago in 2011. It was a road traffic accident following which he skidded off and fell on the road and sustained soft tissue injuries to the face for which he was treated conservatively at a local hospital. A month after the incident, he noticed a single pus-filled lesion on his left cheek which progressively increased to multiple lesions with a gradual reduction in mouth opening. The patient with these complaints consulted a hospital where he underwent wound debridement under general anaesthesia in 2014 along with intravenous antibiotic therapy (Vancomycin and Metronidazole) and there seemed to be no regression of the lesion. He underwent a second surgery of the same lesion with resection of the lesion to gain healthy wound margins under general anaesthesia in May 2017 at the same hospital. A sample tissue was subjected to a histopathological examination which was revealed to be botryomycosis, following which he was started on Injection (Inj.). Amoxicillin and Clavulanic acid 1.2gm TID, Inj. Amikacin 1gm OD, Inj. Metronidazole 100 ml BD for a week. The lesion recurred after three months with advanced borders. The agony and pain of the unresolved infection brought him to our hospital in the year 2017.

The patient’s medical history, drug allergy, and family history were non contributory. Extra oral examination revealed facial asymmetry with diffuse swelling on the left side of the face measuring around 5×3 cm in size; the lesion was seen extending from the left commissure of the mouth up to the ascending border of the mandibular ramus. The swelling had multiple draining sinuses with a yellowish exudate along with the presence of crusting (Table/Fig 1). Paraesthesia could not be elicited. Intra oral examination revealed an ill defined swelling extending from the distal aspect of 35 to the distal aspect of 38, with obliteration of the buccal vestibule, multiple sinus tracts, and purulent material was noticed along with a reduced mouth opening of about two finger breadths (Table/Fig 2).

The panoramic radiograph showed no abnormalities in the region of interest (Table/Fig 3). The patient was referred for the evaluation of immunocompromised status, and the results were negative.

Avascular margins were excised until fresh bleeding was observed from the extra oral lesion (Table/Fig 4) which was sent for histopathological analysis which revealed a diagnosis of botryomycosis (Table/Fig 5). To confirm this diagnosis, the surgically resected tissue (Table/Fig 6) from the hospital at which he initially consulted was also obtained and re-examined histopathologically that evidenced the presence of botryomycosis.

The photomicrograph shows the specimen in 10x magnification with Haematoxylin and Eosin (H&E) staining (Table/Fig 5).

The histopathology reveals epidermis and dermis. The dermis shows the presence of dense inflammatory aggregates that is composed of neutrophils, lymphocytes, plasma cells, histiocytes and foamy macrophages. Few haematoxyphilic colonies of cocci are seen surrounded by eosinophilic material in a fibrillar stroma suggestive of botryomycosis.

Pus from the lesion was sent for culture and sensitivity testing. The cultured micro-organisms were Streptococci viridans. A strategic conservative treatment protocol was devised specifically for the patient's microbial profile as previous surgical intervention led to repeated aggressive recurrence.

Treatment was planned as a triple mode therapy with intravenous and intraoral antibiotic lavage with a modified drain in place along with extra oral skin treatment. An infant feeding tube with a diameter of 3.03 mm was modified with multiple small holes made throughout the surface and was secured in place with the help of sutures in the buccal vestibule (Table/Fig 7). It was used in such a way that while irrigating the antibiotics in a saline medium, there was increased perfusion of this mixture into the intraoral lesion.

The patient was initiated on systemic antibiotic therapy based on culture sensitivity report (Table/Fig 8) along with local antibiotic therapy intra orally via irrigation through the modified drain this was followed by hot water fomentation for the skin lesion which was carried out during the first week of admission, after which the patient was subjected to excision of the avascular margins under local anaesthesia after which the antibiotic and heat therapy was continued postoperatively.

The antibiotic regimen was tailored to the patient’s microbial profile and the culture and sensitivity report. The treatment was devised by the author after the excision of the lesion. The protocol that was followed for the present case has been listed in (Table/Fig 9). This treatment protocol was devised by the author for the management of this lesion.

(Table/Fig 10) shows immediate postoperative results. The lesion visibly regressed within 15 days of rigorous local treatment (Table/Fig 11) with an increase in mouth opening. Our patient also reported better sensory function in the overlying skin during hot fomentation as treatment progressed. After 15 days of hospital stay and robust local management, the patient was discharged with oral antibiotics and was advised for topical colloidal silver gel (brand name: Hydroheal AM gel) application over the skin and was recalled for follow-up a month after discharge. On review at 3 months postoperative, there was a radical regression of the lesion with clear skin margins, and a mouth opening up to four fingers was recorded (Table/Fig 12). (Table/Fig 13) shows the complete resolution of the lesion 1-year postoperatively.

The patient has been under a long-term follow-up period of more than 4 years with no remissions (Table/Fig 14).

Discussion

Botryomycosis is a chronic, suppurative and granulomatous lesion with clinical presentation of fungus-like grains or granules within suppurative foci, caused by Staphylococcus aureus organisms in association with Pseudomonas aeroginosa, Proteus mirabilis, and Escherichia coli (1). The cutaneous manifestations are highly variable as they appear as fungating masses, blisters, nodules, papules, ulcers, and verrucous plaques with or without a sinus tract formation. It may also present with surrounding cellulitis with a superficial abscess which depending on the size and location of the lesion may involve underlying muscle, tendon, or bone. The diagnosis of botryomycosis is arrived at, after careful histopathological examination as it closely resembles an actinomycosis infection in its clinical and histological presentation (2). It was first reported by Bollinger O in 1870. There have been various treatment modalities that have been suggested for treating this lesion since its discovery (3),(4),(5). For the treatment of botryomycosis, a long course of antibiotics is required, depending on the pus culture and sensitivity. However, the most effective treatment for botryomycosis is antibiotics combined with surgical excision (6).

According to a systematic literature review published by Bailey P et al., the mean duration for antibiotic therapy ranges from 4.2 days to 40 weeks for complete resolution of the lesion without any adjuvant procedures (7).

Ellerbe DM et al., reported a case of botryomycosis that presented as cellulitis over the right cheek in an eight-month-old male child who was initially treated with Amoxicillin and Clavulanic acid which had a recurrence and the child was subjected to 19 dives of hyperbaric oxygen therapy along with Nafcillin which was the sensitive antibiotic for two weeks. They reported that the lesion recurred more aggressively and finally, the treatment was switched to medical management using prolonged therapy using Clindamycin for 10 months after which the lesion completely resolved (8).

Devi B et al., reported a case of cutaneous botryomycosis which was treated with surgical resection in addition to antimicrobial therapy for resolution of the lesion with a follow-up period of two months (4).

Eyer-Silva WD et al., in their paper reported a case of cutaneous botryomycosis over the right malar region in a 28-year-old Human Immunodeficiency Virus (HIV) infected woman. The patient underwent several courses of antimicrobial therapy that provided only partial and temporary remission. Complete clinical remission was only achieved five years later when a novel antiretroviral regimen consisting of Darunavir and Raltegravir was initiated. The authors stated that successful treatment of botryomycosis depends on several factors such as the immune status of the host, the causative agents, and the location and extent of the lesion. They concluded that the mainstay of therapy is appropriate antibiotic therapy guided according to bacterial resistance patterns and surgery should be reserved for diagnostic purposes or recalcitrant disease (9).

Lefell DJ et al., reported a case of botryomycosis over the dorsal surface of the foot which was treated with Carbon Dioxide (CO2) laser ablation therapy following failure of the use of broad-spectrum antibiotics in resolving the lesion with a follow-up period of five months (10). Topical heat therapy was first introduced for the treatment of sporotrichosis over the right dorsum of the hand byMackinnon JE and Conti-Diaz IA based on the thermosensitivity of Sporothrix schenckii (11). There are also sporadic reports of its effectiveness for chromoblastomycosis in literature (12),(13).

In addition to medical and surgical therapy, we inculcated the use of heat therapy as adjuvant therapy for the present patient after an extensive literature search on the effects of temperature for therapy-resistant cutaneous skin infections (12),(13),(14). In previously reported literature, there has only been one case report of cutaneous botryomycosis over the right foot which was treated with topical heat therapy along with antibiotic therapy reported by Ishibashi M et al., (14). In their study, the patient was initiated on antibiotic therapy using oral Minocycline at a dose of 200 mg once a day along with topical heat therapy using a commercially available pocket warmer which was constantly applied every day for as long as the patient could tolerate without causing a burn injury. The authors noted significant clinical improvement within four months and no signs of recurrence during an eight months follow-up period. They concluded the study by saying that heat therapy is an effective complementary therapy in the treatment of cutaneous botryomycosis.

The exact mechanism of how heat therapy works in botryomycosis is unknown because the germicidal effect of heat therapy on Staphylococcus aureus cannot be established as they are capable of growth at 48°C even after incubating for seven days (15). The literature reports that heat induces a phagocytic reaction as part of the host defence mechanism and exerts a cytostatic effect (14).

In this case report, instead of a constant heating device to provide heat therapy, we used hot water fomentation over the lesion for the desired effect. Of the cases of botryomycosis reported thus far (2),(3),(4),(16),(17) none have a follow-up period of more than one year with reports of relapse or remission of the disease. The extensive follow-up period without any recurrence in this case report reinforces the need for a multi-modality approach in the treatment of this disease.

Conclusion

Botryomycosis is a disease that requires aggressive management to prevent relapse in the long term. A surgery-only approach that was initially done for patient was futile and required further intervention. Hence, the authors would like to recommend adequate antibiotic therapy based on the patient's culture and sensitivity reports along with surgical debridement and heat therapy for acceptable regression of the lesion. It is also recommended that the same team of doctors should follow-up with the patient so that any changes that occur during the treatment of the lesion is immediately appreciable and further course of action can be determined accordingly.

References

1.
Bonifaz A, Carrasco E. Botryomycosis. International Journal of Dermatology. 1996;35(6):381-88. [crossref] [PubMed]
2.
Yencha MW, Walker CW, Karakla DW, Simko EJ. Cutaneous botryomycosis of the cervicofacial region. Head & Neck: Journal for the Sciences and Specialties of the Head and Neck. 2001;23(7):594-98. [crossref] [PubMed]
3.
Devaiah AK, Hoffman HT, Robinson RA, Carter K. Oral botryomycosis: a case report. The Journal of Laryngology & Otology. 1997;111(1):77-79. [crossref] [PubMed]
4.
Devi B, Behera B, Dash ML, Puhan MR, Pattnaik SS, Patro S. Botryomycosis. Indian Journal of Dermatology. 2013;58(5):406. [crossref] [PubMed]
5.
DeWitt JP, Stetson CL, Thomas KL, Carroll BJ. Extensive cutaneous botryomycosis with subsequent development of Nocardia-positive wound cultures. Journal of Cutaneous Medicine and Surgery. 2018;22(3):344-46. [crossref] [PubMed]
6.
Sirka CS, Dash G, Pradhan S, Naik S, Rout AN, Sahu K. Cutaneous botryomycosis in immunocompetent patients: A case series. Indian Dermatology Online Journal. 2019;10(3):311. [crossref] [PubMed]
7.
Bailey P, Raybould JE, Sastry S, Bearman G. Cutaneous botryomycosis and staphylococcus aureus: diagnosis, management, and a systemic literature review. Current Treatment Options in Infectious Diseases. 2018;10(3):347-61. [crossref]
8.
Ellerbe DM, Parsons DS, Cook PR. Botryomycosis: improved therapy for a difficult infection. International Journal of Pediatric Otorhinolaryngology. 1997;41(3):363-69. [crossref]
9.
Eyer-Silva WD, Silva GA, Ferry FR, Pinto JF. Facial botryomycosis-like pyoderma in an HIV-infected patient: remission after initiation of darunavir and raltegravir. Revista da Sociedade Brasileira de Medicina Tropical. 2017;50(02):277-79. [crossref] [PubMed]
10.
Leffell DJ, Brown MD, Swanson NA. Laser vaporization: a novel treatment of botryomycosis. The Journal of Dermatologic Surgery and Oncology. 1989;15(7):703-05. [crossref] [PubMed]
11.
Mackinnon JE, Conti-Diaz IA. The effect of temperature on sporotrichosis. Sabouraudia. 1963;2(2):56-59. [crossref]
12.
Yanase K. Yamada M. Pocket-warmer therapy of chromomycosis [letter]. Arch Dermatol. 1978;114(7):1095. [crossref] [PubMed]
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DOI and Others

DOI: 10.7860/JCDR/2022/53186.16318

Date of Submission: Jan 01, 2022
Date of Peer Review: Feb 02, 2022
Date of Acceptance: Mar 11, 2022
Date of Publishing: May 01, 2022

AUTHOR DECLARATION:
• Financial or Other Competing Interests: No
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. Yes

PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: Jan 13, 2022
• Manual Googling: Mar 09, 2022
• iThenticate Software: Mar 31, 2022 (8%)

ETYMOLOGY: Author Origin

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