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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 9  |  Issue : 1  |  Page : 72-77

Bartonellosis in maxillofacial region


1 Maxillofacial Surgery Division, N. I. Pirogov University Multiprofile Hospital for Active Treatment and Emergency Medicine, Sofia, Bulgaria
2 Clinic of General and Clinical Pathology, N. I. Pirogov University Multiprofile Hospital for Active Treatment and Emergency Medicine, Sofia, Bulgaria

Date of Submission26-Jan-2022
Date of Acceptance21-Mar-2022
Date of Web Publication29-Apr-2022

Correspondence Address:
Dr. Bistra Blagova
Maxillofacial Surgery Division, University Multiprofile Hospital for Active Treatment and Emergency Medicine N. I. Pirogov, Gen. Totleben“ Blvd. . 21, 1606, Sofia
Bulgaria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/sjoralsci.sjoralsci_3_22

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  Abstract 


Cat-scratch disease (CSD) manifests as a painful regional lymphadenopathy that occurs after a cat scratch or bites transmitted by the bacterium Bartonella henselae. The diagnosis is made by biopsy or puncture of the affected lymph nodes, which must undergo histopathology, serology, or polymerase chain reaction. An 18-year-old woman presented with progressive, painful, right submandibular adenomegaly in the absence of systemic symptoms. She denied the previous contact with cats or any other animals. A computed tomography scan of the cervix showed nodular images corresponding to the affected lymph nodes. The patient underwent a lymph node biopsy, which suggested CSD as the primary diagnosis. The biopsy does not reveal specific diagnoses. B. henselae was confirmed serologically. This case illustrates a typical evolution of CSD in which a patient is affected by subacute lymphadenopathy, but with no epidemiological history. The only certain diagnostic mean was the positive serology of Bartonella. Good general condition, despite pronounced adenomegaly without signs of toxemia, is characteristic of CSD. The lack of more accurate laboratory tests, the difficulty in culturing the pathogen, and the need for histopathology prevent the rapid diagnosis of CSD and contribute to the nonrecognition of this disease. B. henselae testing should be considered when monitoring lymphadenopathy, especially if there is subacute evolution, as in the presented case.

Keywords: Bartonella henselae, bartonellosis, benign lymphadenopathy, cat-scratch disease, cat-scratch fever, felinosis, granulomatous disease, Warthin–Starry staining


How to cite this article:
Blagova B, Markov D, Sаrbianova N. Bartonellosis in maxillofacial region. Saudi J Oral Sci 2022;9:72-7

How to cite this URL:
Blagova B, Markov D, Sаrbianova N. Bartonellosis in maxillofacial region. Saudi J Oral Sci [serial online] 2022 [cited 2022 Dec 5];9:72-7. Available from: https://www.saudijos.org/text.asp?2022/9/1/72/344398




  Introduction Top


Bartonellosis/felinosis, including cat-scratch disease (CSD), is a group of infectious diseases that are rarely detected, so there are no statistics on their etiology and frequency. The causative agent is Batonella spp. Localize forms in Batonella infection, usually presented as self-limiting lymphadenitis, characterized by lymphadenopathy, which follows after contact with a cat and symptoms resolve within 2–4 months.[1],[2] Serology testing is the cornerstone of diagnosis. However, it may be necessary to use clinical samples for microbiological evaluation for diagnosis. Nevertheless, CSD often remains unrecognized in the case of cervicofacial lymph node enlargement, as it is often not considered in patients with cervical lymphadenopathy. The indicative diagnosis will usually be tuberculosis of the cervical lymph nodes, deep neck abscesses, infected bronchial cyst, and malignancies such as malignant lymphoma, oropharyngeal, or laryngeal carcinoma with lymph node metastases.[2] CSD is probably a more common cause of obscure masses in the head and neck than it is expected. In this report, we describe a case of localized Batonella infection with its clinical and pathological manifestations and a brief summary of its diagnosis clarity and management.


  Case Report Top


Patient's history and physical examination. An 18-year-old previously healthy woman presented to the maxillofacial surgery clinic with complaints of painful mass within her left submandibular region for ca. 3-week duration. There was no history of contact with infectious patients or any direct contact with animals. She denied any travel out of the area. She denied eating raw or undercooked foods as well as unpasteurized dairy products. She also denied using tobacco, alcohol, and recreational drugs. The swelling was sudden in onset, gradually progressive in nature, and not associated with rise in temperature. Following outpatient consultation, the patient was treated with antibiotics and saliva stimulating drugs for 2 weeks prior her presentation in our clinic. The therapy was without appreciable effect, as the swelling became larger. On administration, the swelling was approximately 3 cm × 5 cm, tender, rubbery, and nonfluctuant with limited mobility. The overlying skin was warm and reddish. The remainder of the head-and-neck examination was noncontributory. Her vital signs were stable. Her body temperature was not elevated, nor was there any other systematic signs or symptoms. On clinical and radiographic examination, no dental cause could be found [Figure 1]. The results of the complete blood count were not severely changed [Table 1].
Figure 1: Patient's orthopantomography revealed no dental pathology

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Table 1: Results of the laboratory tests on the date of the hospitalization

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The patient was admitted and treated with intravenous co-amoxicillin combined with metronidazole and nonsteroid anti-inflammatory drugs. She underwent an X-ray of the chest [Figure 2], ultrasound investigation of abdominal organs [Figure 3] and cervical structures [Figure 4], and a computed tomography (CT) scan of the neck and maxillofacial region [Figure 5].
Figure 2: X-ray of the chest – no pathology was detected

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Figure 3: Ultrasound investigation of abdominal organs – no pathologic findings

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Figure 4: Ultrasound investigation of cervical structures

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Figure 5: Computed tomography scan of the neck and maxillofacial region (coronal view) showing unhomogenic multiple enlarged lymph nodes within the left submandibular region, below the submandibular salivary gland, as well multiple subcentimeter cervical lymph nodes mainly on the left. The computed tomography showed hypodense lesions one of the submandibular lymph nodes

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Mycobacterium tuberculosis was included as a differential diagnosis. The QuantiFERON-TB Gold test was negative. The patient underwent fine-needle aspiration of the affected lymph node on the 3rd day of hospitalization for microbiological and histological examination. Tissue sample was tested for aerobic and anaerobic bacteria. The test results were negative. No pathologic agents were isolated. On histology, there were no certain findings [Figure 6].
Figure 6: Fine-needle aspiration cytology of the lymph node (H and E stain, ×100). Microscopic evaluation of clinical specimens of lymph node smears showed an aspecific granulomatous process, with a predominant macrophage-lymphoid infiltrate and sparse giant and epithelioid cells. There was an absence of recognizable bacteria, mycobacteria, fungi, and protozoa

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Due to the patient's worsening clinical course and a preliminary diagnosis, including node necrosis, the surgeon team consulted surgery for exploratory biopsy to obtain fresh tissue for histology and new mycobacteria culture. Hence, the patient underwent surgery under a general anesthetic. Although a submandibular incision, the enlarged firm necrotizing nodule was excised and sent for histopathologic examination. The incision was sutured. Healing was uneventful, and skin sutures were removed after 6 days. The patient was discharged 2 days later, for a total 7-day hospital stay.

Diagnosis of Bartonella henselae. Upon further questioning, the patient admitted to visiting a relative of her owner at a about 2 months before her illness. She denied any scratches or bites. While the patient was waiting for the biopsy result, the serology for B. henselae resulted in an IgM titer of 5.39. Histopathologic analysis confirmed the diagnosis. [Figure 7] Based on obtained results, the patient was referred to an infectious disease specialist following the discharge for follow-up.
Figure 7: Microscopic histopathological appearance of the lymph node showing histiocytic granuloma with central microabscess (H and E stain, ×100). Prominent necrotic, basophilic background, with lymphocytes, degenerated neutrophils, and histiocytes. Higher power view shows the focal area of atypical lymphoid cells with necrosis and granulomatous-type inflammation with polymorphous lymphocytes and histiocytes (×400). Cellblock revealed histiocytes, numerous neutrophils in a necrotic background and occasional atypical lymphoid and giant cells

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  Discussion Top


B. henselae is now considered a cosmopolitan emerging human pathogen.[2] CSD, also known as bartonellosis/felinosis, is a worldwide zoonosis caused by B. henselae and associated with a variety of manifestations ranging from lymphadenopathy alone to systemic disease. The main local form manifests as regional and painful lymph adenomegaly of subacute duration, often after a cat bite or scratch.[1] Enlarged tender lymph nodes are most often found in the head or neck area, as in the present case. Nodal enlargement usually lasts for 2–4 months and then resolves spontaneously.[3],[4],[5] The diagnosis of Bartonella infection is based on clinical syndromes, the history of animal exposure, and laboratory results from culture methods, immunoserological or immunocytochemical means, or DNA amplification.[5],[6] However, establishing the diagnosis of CSD can be challenging if the primary inoculation site has healed or is unapparent and no history of animal contact is elicited, like in our patient. Laboratory workup for CSD should include at least two modalities other than cytomorphological features.[7] A serological test can be very useful, but if the cause of lymphadenopathy remains uncertain, a biopsy should be considered to rule out benign or malignant neoplasms.[2] The serological test is expected to be performed as early as possible when the patient presents with such symptoms. The reported case meets the diagnostic criteria for CSD. In the case presented above, the diagnosis was made postoperatively after analysis of the sample and obtained serological results. Histopathological examinations and a review of the medical history confirmed the diagnosis.

Lymphadenopathy is the most common clinical manifestation of Bartonella infection and has been reported in more than 80% of all cases. The location of lymphadenopathy in CSD depends mainly on the site of inoculation. Approximately 10% of infected lymph nodes become fluctuant and develop an overlying cutaneous erythema with the potential for spontaneous suppuration. CSD should be considered in patients with chronic (>3 weeks) lymphadenopathy.[2] The primarily differential of granulomatous lymphadenopathy in the immunocompetent host is CSD, tuberculosis, and neoplasm, primarily lymphoma. Rarer infectious etiologies should also be considered (sarcoidosis, tularemia,  Brucellosis More Details, lymphogranuloma venereum, and sporotrichosis/other fungal infections).[8]

Although CSD has been recognized for decades, only with the recent application of molecular biology has the diagnostic criteria been clarified. MRI, scintigraphy, or CT are useful for demonstrating evidence of tissue involvement in the setting of CSD.[9],[10],[11],[12] Serological confirmation has now become the cornerstone of CSD diagnosis. Detection of antibodies against B. henselae by immunofluorescence analysis or enzyme immunoassay has high sensitivity (88%) and specificity (97%).[13],[14],[15],[16] False negatives may be due to a lack of detectable antibodies, infection with nonreactive bacteria (i.e., Bartonella clarridgeiae or Afipia felis), which are sometimes reported to cause CSD, or result from heterogeneous antigenicity among B. henselae species.[17],[18]

The clinical diagnosis can be made when three of the four criteria are met: 1) contact with a cat, with the presence of a scratch or primary lesion; 2) a positive skin test for CSD, in which the antigen of the skin test (0.1 ml) is injected intradermally on the flexor surface of the arm and a hardening of 5 mm or more is observed after 72 h; 3) negative laboratory results from serological tests, skin tests for purified protein derivatives, and cultures of aspirated pus or lymph nodes performed for other causes of lymphadenopathy; and 4) characteristic histopathological features in a biopsy specimen.[19]

Cytology of typical CSD samples is found to correlate closely with classic histology.[20] [Table 2] and [Table 3] Lymph node histology shows characteristic but nonspecific granulomatous lesions with central, sometimes stellar necrosis and neutrophils surrounded by palisade histiocytes. Granulomas in 77%, neutrophils in 62%, epithelioid histiocytes in 46%, and purulent granulomas in 38% were found in 13 fine-needle aspirates of CSD from the head and neck.[21] The clinical suspicion was of neoplasia in 38%, and the cytological difference was of bacterial abscess and lymphoproliferative disorder. Touch imprint from excised lymph nodes in eight cases diagnosed as CSD showed epithelioid histiocytes and monocytoid B cells.[22] The histopathology of both typical and atypical CSD was found to follow the same three stages of manifestation. Key cytomorphological findings are necrosis, multiple neutrophils, granulomatous changes (epithelioid histiocytes and giant Langhans cells), and monocytoid B cells in the lymph node. Microscopic findings should prompt consideration of mycobacterial infection and B. henselae infection among the less common infections in the immunocompetent host. The microscopic finding of suppurative necrosis and monocytoid B cells should prompt consideration of B. henselae. Early-stage infection lacks prominent neutrophils and suppurative necrosis, showing only increased histiocytes and follicular hyperplasia. Late-stage infection shows abundant neutrophils and abscess formation, as in this case.[8]
Table 2: Cytologic findings in fine-needle aspirates of cat-scratch disease[20]

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Table 3: Summary of histologic findings in various infective stages and in the resolving stage[8]

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The treatment of CSD remains a mystery as there are no prospective studies. The indications for antibiotic treatment and the type and duration of therapy are contradictory.[23] Although CSD is usually self-limiting and resolves spontaneously within 1–3 months, follow-up is performed on average up to 24 months for recurrences.[10],[24],[25] Antimicrobial therapy can shorten the period of symptomatic disease and may contribute to more rapid resolution of clinical abnormalities associated with infection.[1] The role of surgery in the management of CSD is limited. Needle aspiration of a suppurative node is the treatment of choice and may relieve local pain. Excision of the involved lymph nodes may be indicated when there is diagnostic uncertainty, upper airway obstruction, or for the management of a persistently draining sinus.[24] Suppurative lymph nodes that become tense, tender and painful should be drained, but incisions of nonpurulent lesions should be avoided as chronic drainage fistula or a very low tendency to heal may result.[10],[12] The patient with CSD does not need isolation or quarantine, as there is no evidence of human-to-human transmission.[26]


  Conclusion Top


Regional lymphadenopathy is sine qua non of Bartonella infection. Therefore, B. henselae should always be in the differential diagnosis of patients who present with a tender swollen lymph node. However, due to the wide range of systemic manifestations of this disease, it should not be neglected in patients with atypical manifestations. CSD occurs at all ages and should be included in the differential diagnosis of head-and-neck lymphadenopathy. Hence, it is likely to be seen by a general dentist and will undoubtedly be seen by oral and maxillofacial surgeons with increasing frequency over time. Biopsy is a useful method for diagnosing CSD and for excluding other etiologies. Despite the sequence of histological changes in the lymph nodes, they are not pathognomonic. Ultimately, no criterion should be considered the diagnostic gold standard and the diagnosis of B. henselae infection should rely on a combination of epidemiological, serological, clinical, histological, and bacteriological criteria.

Statement of informed consent

Written informed consent was obtained from the patient(s) for their anonymized information to be published in this article.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that their name and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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Giladi M, Kletter Y, Avidor B, Metzkor-Cotter E, Varon M, Golan Y, et al. Enzyme immunoassay for the diagnosis of cat-scratch disease defined by polymerase chain reaction. Clin Infect Dis 2001;33:1852-8.  Back to cited text no. 13
    
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Massei F, Messina F, Talini I, Massimetti M, Palla G, Macchia P, et al. Widening of the clinical spectrum of Bartonella henselae infection as recognized through serodiagnostics. Eur J Pediatr 2000;159:416-9.  Back to cited text no. 14
    
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Zbinden R. Bartonella henselae-based indirect fluorescence assays are useful for diagnosis of cat scratch disease. J Clin Microbiol 1998;36:3741-2.  Back to cited text no. 16
    
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Giladi M, Avidor B, Kletter Y, Abulafia S, Slater LN, Welch DF, et al. Cat scratch disease: The rare role of Afipia felis. J Clin Microbiol 1998;36:2499-502.  Back to cited text no. 17
    
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Sander A, Zagrosek A, Bredt W, Schiltz E, Piémont Y, Lanz C, et al. Characterization of Bartonella clarridgeiae flagellin (FlaA) and detection of antiflagellin antibodies in patients with lymphadenopathy. J Clin Microbiol 2000;38:2943-8.  Back to cited text no. 18
    
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Margileth AM. Cat-scratch disease; etiology, diagnosis and therapy. Infect Med 1993;10:38-46.  Back to cited text no. 19
    
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Silverman JF. Fine needle aspiration cytology of cat scratch disease. Acta Cytol 1985;29:542-7.  Back to cited text no. 20
    
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Donnelly A, Hendricks G, Martens S, Strovers C, Wiemerslage S, Thomas PA. Cytologic diagnosis of cat scratch disease (CSD) by fine-needle aspiration. Diagn Cytopathol 1995;13:103-6.  Back to cited text no. 21
    
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Kojima M, Nakamura S, Koshikawa T, Kurabayashi Y, Senpuku A, Yamane N, et al. Imprint cytology of cat scratch disease. A report of eight cases. APMIS 1996;104:389-94.  Back to cited text no. 22
    
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Jacomo V, Kelly PJ, Raoult D. Natural history of Bartonella infections (an exception to Koch's postulate). Clin Diagn Lab Immunol 2002;9:8-18.  Back to cited text no. 25
    
26.
Margileth AM, Rathore MH. Bortonella species; Cat Scratch Disease in The Neurological Manifestations of Pediatric Infectious Diseases and Immunodeficiency Syndromes. Humana Press 2008. p. 243-50.  Back to cited text no. 26
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]



 

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