Newborn With Branchial Cleft Cyst Infection Due to Nocardia otitidiscaviarum.

OpenAlex İngilizce

To the Editors: Nocardia otitidiscaviarum was accounted for 0.3%–2.9% of all Nocardia infections reported.1,2 To the best of our knowledge, 54 cases diagnosed with N. otitidiscaviarum infection have been reported in the literature.3 Branchial cleft cysts are usually diagnosed in the 2nd to 3rd decade on average because of infection caused by streptococci and anaerobic strains.3 In our case, N. otitidiscaviarum was identified for the first time in the literature in a newborn with a branchial cleft cyst. A female baby was born 2600 g by cesarean section at 36 weeks of gestation from a 35-year-old mother. She presented with intermittent bloody stools at the age of 24 days. The patient, whose physical examination was normal, was hospitalized for further investigations with a preliminary diagnosis of cow’s milk allergy. A few days after hospitalization, rapidly growing swelling in the right mandible and hyperemia developed over time (Fig. 1). The patient never had a fever and the other system examination was normal. Laboratory values demonstrated a white blood cell count of 6.40 × 109 cells/L, neutrophil of 2.20 × 109 cells/L, platelets of 310 × 109 cells/L, and C-reactive protein of 30 mg/L. There was no growth in the blood culture. Ultrasonography demonstrated a dense loculation (abscess?) in the right submandibular region. Neck computed tomography revealed a 16 × 14 × 17 mm, well-circumscribed, thin-walled, contrast-enhancing, dense cystic lesion (infected 2nd branchial cleft cyst?) in the right submandibular region (Fig. 2). The patient was started intravenously ceftriaxone and teicoplanin for empiric. The fluctuating abscess developed 24 hours after the antibiotic was started and the abscess was drained (2 mL of purulent fluid). In a direct microscopic view, the initial aspiration showed abundant neutrophil and Gram-positive filamentous bacilli. N. otitidiscaviarum was grown in the aspirate culture and identified via matrix-assisted laser desorption ionization-time of flight mass spectrometry. Antibiotic susceptibility could not be tested. Chest X-ray, echocardiography, abdomen ultrasonography, transfontanelle ultrasonography, and lumbar puncture were performed for other organ spread and all of them were normal. Conditions causing immunodeficiency were investigated in the patient. Quantitative Ig assays demonstrated serum levels of IgA < 10, IgG = 104.7, and IgM < 20 mg/dL (compatible with hypogammaglobulinemia according to the standard deviations for age and our laboratory reference values ). Lymphocyte subgroups were normal. The human immunodeficiency virus test was negative. Swelling, redness, and fluctuating abscess recurred after the first aspiration, and the abscess was drained for the second time, 6 days apart. Treatment was switched to linezolid due to obtaining culture results and lacking clinical improvement. Linezolid was given intravenously for 8 days and subsequently, it was switched to oral trimethoprim/sulfamethoxazole (15 mg/kg/d). The patient, whose symptoms and signs regressed, was discharged on the 10th day. Serum IgM, IgG and IgA values measured at 6 months in the patient were within the normal range according to age and were diagnosed as transient hypogammaglobulinemia of infancy. No pathogenic mutation was detected in the whole exon scan performed for immunodeficiency. The antibiotic treatment was completed in 12 months. Follow-up examination was normal at 18 months after treatment was completed.FIGURE 1.: Swelling and hyperemia in the right mandible on physical examination.FIGURE 2.: A: Sagittal and (B) axial views of infected branchial cleft cyst showing on neck computed tomography image.The risk factors for Nocardia infections are malignancies, long-term corticosteroid use, and primary immunodeficiency states. It was also reported in immunocompetent patients.4 We thought that the fact that our patient had an anatomical disorder, was in the neonatal period, and was diagnosed with temporary hypogammaglobulinemia in infancy created a predisposition to this infection. Diagnosis (it grows slowly in culture) and management of nocardial infections are quite difficult.4 Treatment management is decided based on retrospective experience, case reports, and in vitro antimicrobial activity profiles. In case reports, recurrence was observed in some of the cases treated for 4–6 months. Deaths have been reported in a few patients with severe and disseminated disease. Considering these data, most N. otitidiscaviarum cases were administered 6–12 months of treatment.5,6 In conclusion, anatomical disorders may also be predisposing factors for Nocardia species, in addition to immunodeficiency states. Although it is more common in adults, it should be kept in mind that it can be a factor even in newborns. Difficulties in diagnosis and long treatment processes put clinicians in a difficult position.