Primary Actinomycosis of the Hand: a Very Rare Case Report · tb 2×1 for about 5 years of this 8...
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MEDICINE Primary Actinomycosis of the Hand: a Very Rare Case Report Sabahattin Yüzkan 1 & İpek Tamsel 1 & Duygu Cengiz 1 & Meltem Taşbakan 2 & Banu Yaman 3 & Mehmet Argın 1 Accepted: 29 July 2019 /Published online: 3 August 2019 # Springer Nature Switzerland AG 2019 Introduction Actinomycosis is a slowly progressive, chronic suppura- tive infection caused by the gram-positive branching bac- terium of the genus Actinomyces. It is an anaerobic or microaerophilic bacterium which normally located in the microflora of mouth, vagina, and colon [1] that usually causes infection in diabetic and immunosuppressed pa- tients. The three major encountered forms of actinomyco- sis are cervicofacial (65%), abdominal/pelvic (20%), and thoracic (15%) [2, 3]. Primary involvement of hand by actinomycosis is very rare. In this case report, we aimed to present primary actinomycosis of hand in which bones, muscles, and subcutaneous soft tissues were affected. Case Report Sixty-year-old male patient was referred to our hospital be- cause of left hand swelling, pain, and limitation of movements for a long time. Complete blood count, biochemical laboratory values, erythrocyte sedimentation rate, and C-reactive protein values do not change significantly. He did not remember any trauma history. There is no clinical history of any chronic disease, malignancy, or immunosuppression. On examination, the patient was afebrile. Musculoskeletal examination showed a moderately swollen left hand. The swelling of the hand started from the carpal areas extended to metacarpal bones. In the inspection of the hand, the skin was hard, and few fistulous tracks were seen with some purulent material coming from fistulas. There was no palpable lymphadenopathy. He said that he did not achieve the cure of the hand for 8 years. He used cefazolin sodium (Iespor) 1 g IM 2 × 1 and amoxicillin-clavulanate potassium (Augmentin) 875/125 mg tb 2 × 1 for about 5 years of this 8 year period, but he did not get response from these antibiotics. He started ampicillin (ampicid) 1 g IM 4 × 2 and sulfamethoxazole-trimethoprim (Bactrim forte) 800/160 mg tb 2 × 1 treatment and used these for about following 3 years; however, he could not get any improvement. Also during this time, he received 40 sessions of hyperbaric oxygen therapy, and various exercises has been performed at different times to decrease the limitation of movements. His complaints did not regress despite the various beta-lactam antibiotics, keratolytics, and physiotherapy. Magnetic resonance imaging showed that inflammation of the skin-subcutan soft tissues and osteomyelitis with involve- ment of the carpal and metacarpal bones of the left hand. These changes were confirmed on CT and X-ray images too (Figs. 1, 2 and 3). During this time, a biopsy of the left hand was done. The biopsy material was stained with hematoxylin and eosin showed soft tissue abscess formation, with many of the abscesses containing purple filamentous sulfur granules This article is part of the Topical Collection on Medicine * Sabahattin Yüzkan [email protected] İpek Tamsel [email protected] Duygu Cengiz [email protected] Meltem Taşbakan [email protected] Banu Yaman [email protected] Mehmet Argın [email protected] 1 Department of Radiology, Ege University Faculty of Medicine, Izmir, Turkey 2 Department of Infectious Diseases and Clinical Microbiology, Ege University Faculty of Medicine, Izmir, Turkey 3 Department of Pathology, Ege University Faculty of Medicine, Izmir, Turkey SN Comprehensive Clinical Medicine (2019) 1:687–691 https://doi.org/10.1007/s42399-019-00119-9
Transcript of Primary Actinomycosis of the Hand: a Very Rare Case Report · tb 2×1 for about 5 years of this 8...
MEDICINE
Primary Actinomycosis of the Hand: a Very Rare Case Report
Sabahattin Yüzkan1& İpek Tamsel1 & Duygu Cengiz1 & Meltem Taşbakan2
& Banu Yaman3& Mehmet Argın1
Accepted: 29 July 2019 /Published online: 3 August 2019# Springer Nature Switzerland AG 2019
Introduction
Actinomycosis is a slowly progressive, chronic suppura-tive infection caused by the gram-positive branching bac-terium of the genus Actinomyces. It is an anaerobic ormicroaerophilic bacterium which normally located in themicroflora of mouth, vagina, and colon [1] that usuallycauses infection in diabetic and immunosuppressed pa-tients. The three major encountered forms of actinomyco-sis are cervicofacial (65%), abdominal/pelvic (20%), andthoracic (15%) [2, 3]. Primary involvement of hand byactinomycosis is very rare. In this case report, we aimedto present primary actinomycosis of hand in which bones,muscles, and subcutaneous soft tissues were affected.
Case Report
Sixty-year-old male patient was referred to our hospital be-cause of left hand swelling, pain, and limitation of movementsfor a long time. Complete blood count, biochemical laboratoryvalues, erythrocyte sedimentation rate, and C-reactive proteinvalues do not change significantly. He did not remember anytrauma history. There is no clinical history of any chronicdisease, malignancy, or immunosuppression. On examination,the patient was afebrile. Musculoskeletal examination showed
a moderately swollen left hand. The swelling of the handstarted from the carpal areas extended to metacarpal bones.In the inspection of the hand, the skin was hard, and fewfistulous tracks were seen with some purulent material comingfrom fistulas. There was no palpable lymphadenopathy. Hesaid that he did not achieve the cure of the hand for 8 years.He used cefazolin sodium (Iespor) 1 g IM 2 × 1 andamoxicillin-clavulanate potassium (Augmentin) 875/125 mgtb 2 × 1 for about 5 years of this 8 year period, but he did notget response from these antibiotics. He started ampicillin(ampicid) 1 g IM 4 × 2 and sulfamethoxazole-trimethoprim(Bactrim forte) 800/160 mg tb 2 × 1 treatment and used thesefor about following 3 years; however, he could not get anyimprovement. Also during this time, he received 40 sessionsof hyperbaric oxygen therapy, and various exercises has beenperformed at different times to decrease the limitation ofmovements. His complaints did not regress despite the variousbeta-lactam antibiotics, keratolytics, and physiotherapy.Magnetic resonance imaging showed that inflammation ofthe skin-subcutan soft tissues and osteomyelitis with involve-ment of the carpal and metacarpal bones of the left hand.These changes were confirmed on CT and X-ray images too(Figs. 1, 2 and 3). During this time, a biopsy of the left handwas done. The biopsy material was stained with hematoxylinand eosin showed soft tissue abscess formation, with many ofthe abscesses containing purple filamentous sulfur granules
This article is part of the Topical Collection on Medicine
* Sabahattin Yü[email protected]
İpek [email protected]
Duygu [email protected]
Meltem Taş[email protected]
Banu [email protected]
Mehmet Argı[email protected]
1 Department of Radiology, Ege University Faculty of Medicine,Izmir, Turkey
2 Department of Infectious Diseases and Clinical Microbiology, EgeUniversity Faculty of Medicine, Izmir, Turkey
3 Department of Pathology, Ege University Faculty of Medicine,Izmir, Turkey
SN Comprehensive Clinical Medicine (2019) 1:687–691https://doi.org/10.1007/s42399-019-00119-9
(Figs. 4 and 5). With tissue biopsy, fungal infection (Mannantest and Grocott’s GMS negative), and tuberculosis (ARBnegative) were excluded. Because of extensive osteomyelitis,surgical debridement was advised. However, the patient re-fused for 8 years. Finally, in 2019, the patient accepted surgi-cal debridement. The carpal and metacarpal bones were ex-cised together with the surrounding debris and necrotic tis-sues. The wrist is fixed with Kirschner wires placed on theproximal phalanges from the radius (Figs. 6 and 7). The wrist
distance was maintained by placing spacer with antibiotics tothe excision area. After surgery, the patient used ciprofloxacin(Cipro) 500 mg tb 2 × 1 and sulfamethoxazole-trimethoprim(Bactrim forte) 800/160 mg tb 2 × 1 for 2 months.While usingthese drugs, he did not have any new complaints like fever andcefoperazone sodium (Sulzon) 1 g IV 4 × 1 was added to thetreatment after 2 months. These drugs will be continued untilcomplete resolution of his symptoms that will be regularlychecked at the orthopedic polyclinic monthly.
Fig. 1 (a) AP hand X-Ray (b)Coronal CT (c) 3D volume ren-dering images from the studydone in 2011 show extensiveosteolysis in metacarpal and car-pal bones
Fig. 2 Fat-saturated T2 weightedimages from the study done in2011 (a and b) showshyperintense ring surrounding thecentral hypointense point which iscalled as dot in circlesign.(showed with black arrow)Fat-saturated postcontrast T1weighted images (c and d) showdiffuse enhancement of bonemarrow and adjacent soft tissuesdue to current infection
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Discussion
Actinomycosis is a rare, chronic, and slowly progressiveinfection that can affect various parts of the body [4]. Itis an anaerobic or microaerophilic bacterium which nor-mally located in the microflora of mouth, vagina, andcolon. The most common subtype is Actinomycesisraelii that usually affects diabetic and immunosup-pressed patients. It is also seen in patients with long-term usage of steroids or chronic renal failure. In our
case, there was no chronic disease, drug use, or immu-nosuppression. Actinomycosis was not found outside ofliving creatures [5]. The cultures are positive in only24% of cases; therefore, the gold standard diagnosis ishistopathological examination [6]. In the microscopicexamination, abscess collections with sulfur granulesare diagnostic [7].
The three major encountered forms of actinomycosis arecervicofacial (65%), abdominal/pelvic (20%), and thoracic(15%) [2, 3]. Primary actinomycosis of extremity is extremelyrare. Most of the infections occurred by direct extension after
Fig. 3 AP hand X ray imagestaken in 2017 (a), 2018 (b) and2019 (c) show severe progressionin osteolysis. At fat saturatedpostcontrast T1 weighted image(d) shows persistence in activeinfection signs in most recentimaging of the patient
Fig. 4 Suppurative inflamation with formation of multiple abscessescontains characteristic granules (sulfur granules) seen with H&E stain Fig. 5 Characteristic granules (sulfur granules) seen with gram stain
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the trauma or hematogenous spread. Lymphadenopathy is notseen because lymphatic spread of infection is not common [7].The infection generally starts in the skin-subcutaneous softtissues then spreads towards the muscles and bones. The acti-nomycosis infection usually manifests with sinus tracts andabscess collections in skin and subcutaneous soft tissues. Insome cases, it may also present as a mass or nodule. X-Rayand contrast-enhancedMRI findings are diagnostic. Extensiveosteolysis due to osteomyelitis in bone structures anddiffuse edema-sinus tracts in the soft tissues are mostcommon findings. The hyperintense ring surroundingthe central hypointense point on T2-weighted images isan important finding which is defined for fungal infec-tions (dot in circle sign).
Actinomycosis is often misdiagnosed due to its chronic andsuppurative course resembling fungal infection, cutaneous tu-berculosis, and malignancy [8]. Because of this, patients gen-erally get diagnosed at late stage. To achieve early and accu-rate diagnosis, it is necessary to be highly suspicious.
Conclusion
Actinomycosis should be kept in mind in chronic and sub-acute progressive infectious processes. In this way, it is possi-ble to successfully treat actinomycosis in early stage by sur-gical debridement and antibiotics.
Fig. 7 The sinus tracts opening tothe skin are seen in the photosafter surgery: One of the sinustract is pointed with black arrow
Fig. 6 AP (a) and oblique (b) lefthand X-Ray images after surgicaldebridement
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Authors Contributions 1 Sabahattin YüzkanConception and design, acquisition of data, analysis, and interpretation
of data. Drafting the article. Final approval of the version to be published.Agree to be accountable for all aspects of the work if questions ariserelated to its accuracy or integrity.
2 İpek TamselAnalysis and interpretation of data. Revising the article critically for
important intellectual content. Final approval of the version to be pub-lished. Agree to be accountable for all aspects of the work if questionsarise related to itsaccuracy or integrity.
3 Duygu CengizAnalysis and interpretation of data. Revising the article critically for
important intellectual content. Final approval of the version to be pub-lished. Agree to be accountable for all aspects of the work if questionsarise related to its accuracy or integrity.
4 Meltem TaşbakanAnalysis and interpretation of data. Revising the article critically for
important intellectual content. Final approval of the version to be pub-lished. Agree to be accountable for all aspects of the work if questionsarise related to its accuracy or integrity.
5 Banu YamanAnalysis and interpretation of data. Revising the article critically for
important intellectual content. Final approval of the version to be pub-lished. Agree to be accountable for all aspects of the work if questionsarise related to its accuracy or integrity.
6 Mehmet ArgınAnalysis and interpretation of data. Revising the article critically for
important intellectual content. Final approval of the version to be pub-lished. Agree to be accountable for all aspects of the work if questionsarise related to its accuracy or integrity.
Compliance with Ethical Standard
Conflict of Interest The authors declare that they have no conflict ofinterest.
Research Involving Human Participants and/or Animals Not applicable.
Informed Consent The patient gave his consent for publication of thiscase report and accompanying images.
References
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5. Aypak C, Gokce H, Altunsoy A, Koc S, Kaplan S. Primary actino-mycosis of hand: a rare soft tissue infection. J Dermatol. 2012;39(8):741–2.
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