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after back neck a host full 9 past 4 was was also non and and soft . p l e of of r d 2 e . PARIPEX - INDIAN JOURNAL OF RESEARCH Volume-7 | Issue-6 | June-2018 | PRINT ISSN No 2250-1991 showed remarkable results in his recovery. He was briefed about multiple basophilic granules, that represent lobulated micro- the need of long term treatment and was followed up every 2 colonies of Actinomyces3. Less invasive diagnostic techniques, months. A year after the therapy the patient has shown fine-needle aspiration (FNAC) is not very useful as it shows only considerable improvement with significant reduction in size of the inflammatory cells. The radiological investigations does not swelling and healing of ulcerations. There is no evidence of provide a specific diagnosis but can help us to asses the pattern recurrent lesion since last one year. and spread of the disease4.

DISCUSSION Actinomycosis requires prolonged treatment with high doses of Cervical Actinomycosis is an uncommon but fascinating infection antimicrobials and Penicillin is the drug of choice for prolonged of the head and neck. It has been called by Russo 'the most treatment. Although therapy must be individualised , the intra- misdiagnosed disease', and it has been said that no disease is so venous administration of 18 - 24 million units of penicillin daily often missed by experienced clinicians1. During the 1970s, an for 2-6 weeks, followed by oral therapy with penicillin or annual incidence of 1 case per 300 000 was found in Cleveland amoxycillin for a total duration of 6- 12 months is considered as area2. During same period, Desai et al reviewed 40 cases of the standard therapy1. For penicillin-allergic patients, , mycetoma infection in India, in which there were 2 cases of minocycline, tetracycline, clindamycin, erythromycin and Actinomycosis israelii infection of the cervicofacial region5. Recent cephalosporins have been found to be effective1. In the present reports are however rare. A review of literature, as described in case, coming from a far off village and being the only breadwinner Volante et al study, revealed 48 cases of cervicofacial in the family, patient was not willing for a long term hospital stay. 3 Actinomycosis reported over the last 25 years . Improved dental So we treated him with high dose injectable penicillin (4 lakh IU) hygiene and the widespread use of antibiotic treatment have every 3 weekly along with oral penicillin (800mg per day) and the contributed to a decrease in this incidence. result of our therapy was equivocal to the of the standard therapy described in literature. As seen in the present case study, the disease is more commonly seen in middle aged men, usually endemic in farmers, gardeners or Surgical treatment may include incision and drainage of the stable workers in whom the causative organisms, Actinomyces sp. , resection of necrotic tissue, and curettage of bone1. An (most commonly A. israelii), are often present as commensals in early diagnosis can often avoid the need for surgery and prevent oral cavity, until the tissue-lining becomes damaged by injury or unnecessary morbidity and mortality. Hyperbaric oxygen therapy disease. The bacteria can then penetrate deeper into the body and can also be used to treat Actinomycosis by increasing oxygen produce disease with the 'copathogens'. Sometimes, the tension which often proves lethal to this anaerobic organism. antecedent history of oral trauma or may not be elicited. No underlying disease or immunosuppression is found in most Regular and meticulous follow up every 2 to 3 months should be 2 cases . Classically, Cervical Actinomycosis is characterised in the ensured to assess the response to the therapy. Monitoring the initial stages by soft- tissue swelling of the perimandibular area. impact of therapy with CT and MRI is advisable when appropriate1. Direct spread into the adjacent tissues occurs over time along with formation of abscesses, fibrosis and woody induration of tissues, The preventive measure include maintenance of good oral hygiene and draining sinuses that discharge purulent material. However, and adequate regular dental care, in particular removal of dental according to some authors, this classic formation appearance of plaque, may reduce the density if not the incidence of colonisation woody swelling with sinus tracts draining purulent material is and low-grade periodontal infection with Actinomyces species. observed in approximately 40% of cases, and, when present, may 3 be helpful in the differential diagnosis . Symptoms like pain is rare The prognosis for the treated Actinomycosis infection is excellent if however slight fever may be associated in around 50 % patients3. recognised early. The cure rates are as high as 90 % and neither deformity nor death is common. The overall mortality from The classic clinical presentation that should prompt consideration Actinomycosis ranges from 0-28% depending mainly on the site of this unique infection are1: of infection and the time of diagnosis; CNS involvement being • the combination of chronicity, progression across tissue associated with greatest mortality2. boundaries and mass like features. • the development of sinus tract which may spontaneously CONCLUSION resolve and recur Based on our case report, it could be concluded that Actinomycosis • the remission and exacerbation of symptoms occurring in poses a great diagnostic challenge as it can mimic many other parallel sequence with initiation and cessation of antibiotic disease because of its insidious course and non specific symptoms. administration Since medical therapy alone can frequently cure the disease, challenge lies in considering the possibility of Actinomycosis, In the present case, when our patient presented with recurrent diagnosing it in the least invasive way and avoid unnecessary neck swelling with non healing skin ulcers and change of voice, our surgery. A greater index of clinical suspicion, early diagnosis and differential diagnosis included , chronic , other appropriate treatment can ensure high cure rates and reduced chronic granulomatous lesions, fungal infection or even morbidity and mortality. malignancy. Since Actinomycosis infection is rare, insidious, chronic, has nonspecific symptoms and imitates more common conditions such as malignancy and TB, the diagnosis was a challenge.

The diagnosis of Actinomycosis is suspected clinically and is confirmed on microscopy, culture and imaging. The diagnosis may be established only by a positive culture, however, Actinomyces growth is very difficult even on appropriate anaerobic media. Thus, Figure 1 : Neck swelling was seen with multiple non healing microbiological identification of this organism is often impossible. ulcerations. Whitish discharge was noted. The macroscopic presence of the classic granules with a yellow sulfur like appearance, termed as 'sulfur granules', in tissue specimens or drainage may be of some help when making diagnosis even if these features are not pathognomic, since may also present with sulfur granules3. However, according to Lancella et al study, the characteristic sulphur granules, in the specimens, are present in only 35-55% of cases and in these cases, the diagnosis is confirmative6.

The histopathological examination of the tissue biopsy is the cornerstone for definitive diagnosis of the disease. The microscopic examination reveals a typical finding of an outer zone Figure 2 : CECT neck showing the extent of the disease and of granulation and a central zone of necrosis which contains narrowing of supraglottic airway 10 www.worldwidejournals.com PARIPEX - INDIAN JOURNAL OF RESEARCH Volume-7 | Issue-6 | June-2018 | PRINT ISSN No 2250-1991

Figure 3 : Histopathological examination showing Actinomycosis israelii and its confirmation on special stains (PAS and ZN stain)

BIBLIOGRAPHY 1. Russo T. Actinomycosis and Whipple's disease. Harrison principle of internal medicine. 2015; 200: 1088-1091. 2. Acevedo F, Baudrand R, Letelier L, Gaete P. Actinomycosis: a great pretender. Case reports of unusual presentations and a review of the literature. International Journal of Infectious Diseases. 2008;12(4):358-362. 3. Volante M, Fantoni A, Ricci R, Galli. Cervicofacial actinomycosis: still a differential diagnosis. J Acta Otorhinolatryngol Ital. 2005;25(2):116-9. 4. Brook I. Abscesses from actinomyces infection: why so difficult to diagnose?.Expert Review of Anti-infective Therapy. 2011;9(12):1097-99. 5. Nagral S, Patel C, Pathare P, Pandit A, Mittal B. Actinomycotic Pseudotumor of the mid cervical region ( A case report). J Postgrad Med. 1991;37:62-4. 6. Lancella A, Abbate G, Foscolo A, Dosdegani R. Two unusual presentations of cervicofacial Actinomycosis and review of literature. J Acta Otorhinolaryngology Ital.2008;28(2):89-93.

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