Congenital brucellosis presented with hypertrophic obstructive cardiomyopathy and persistent multidrug-resistant meningitis

Manish V Lakum; Prakash C Vaghela; Chirag A Gabani; Hiral D Mangukiya

doi:10.4103/jcn.jcn_141_20

CASE REPORT

Year : 2021 | Volume : 10 | Issue : 2 | Page : 133-134

Congenital brucellosis presented with hypertrophic obstructive cardiomyopathy and persistent multidrug-resistant meningitis

Manish V Lakum, Prakash C Vaghela, Chirag A Gabani, Hiral D Mangukiya
Department of Neonatal Intensive Care Unit and Neonatology, Nice Children Hospital, Bhavnagar, Gujarat, India

Date of Submission	29-Aug-2020
Date of Decision	12-Mar-2021
Date of Acceptance	25-Mar-2021
Date of Web Publication	15-May-2021

Correspondence Address:
Prakash C Vaghela
Nice Children Hospital, 1^st Floor, Sameep Complex, Opp. Kalubha, Bhavnagar, Gujrat
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jcn.jcn_141_20

Abstract

Congenital brucellosis is uncommon in neonates, and transplacental transmission is the most common etiology in such case. We reported such case with meningitis and hypertrophic obstructive cardiomyopathy successfully treated with oral rifampicin, trimethoprim + sulfamethoxazole, and injection ceftriaxone and gentamicin. This case illustrates that neonatal brucellosis behaves as a persistent multidrug-resistant sepsis.

Keywords: Congenital brucellosis, hypertrophic obstructive cardiomyopathy, meningitis, transplacental transmission

How to cite this article:
Lakum MV, Vaghela PC, Gabani CA, Mangukiya HD. Congenital brucellosis presented with hypertrophic obstructive cardiomyopathy and persistent multidrug-resistant meningitis. J Clin Neonatol 2021;10:133-4

How to cite this URL:
Lakum MV, Vaghela PC, Gabani CA, Mangukiya HD. Congenital brucellosis presented with hypertrophic obstructive cardiomyopathy and persistent multidrug-resistant meningitis. J Clin Neonatol [serial online] 2021 [cited 2023 Mar 21];10:133-4. Available from: https://www.jcnonweb.com/text.asp?2021/10/2/133/316171

Introduction

Brucellosis is a zoonotic disease,^[1] caused by Gram-negative coco bacillus, aerobic, nonmotile, nonspore-forming organism. It is a transmitted to human by contact with fluid from the infected animal and unpasteurized milk and milk products of sheep, goat, and cattle. Occupational exposure to laboratory worker, veterinarians, slaughter house workers has been seen in few cases. There are four types of brucellosis (1) Brucella abortus, (2) Brucella suis, (3) Brucella canis, (4) Brucella melitensis.^[2] Human-to-human transmission is rare^[3] but rarely transmitted by sexual intercourse, blood transfusion, and organ transplantation.^[4],[5] Breast milk to neonate and transplacental transmission is rare.^[6] Here, we discuss congenital brucellosis contracted from the transplacental route from serology-positive mother.

Case Report

A 32-year-old second gravida nondiabetic mother had premature rupture of membrane (PROM) at 32 weeks, so the baby was delivered by emergency lower segment cesarean section. Mother had a history of ingestion of unpasteurized milk. Mother has been diagnosed as having brucellosis at the third trimester as she was Brucella IgM positive, treated partially for Brucella fever by rifampicin for 6 weeks. At birth, the baby was intrauterine growth restriction (IUGR), 800 g required delivery room resuscitation in form of CPAP and transferred to the NICU for post resuscitation care. On examination, mild hepatomegaly and other examination findings were normal. Chest X-ray was done which was suggestive of moderate respiratory distress syndrome (RDS), so the baby was kept on nasal ventilator support.

Prothrombin time was prolonged so injection FFP was given. Blood culture was sent immediately; considering risk factor and clinical condition of the baby, intravenous antibiotics were started. On day 5, the baby was cyanosed, spo2 was 70% on cpap and so kept on invasive ventilator support, ABG was suggestive of respiratory acidosis with high lactate, blood reports were suggestive of persistent thrombocytopenia (90000/cumm), and high C reactive protein (CRP) 25 mg/dl, and B. abortus organism was isolated from the blood culture, so rifampicin^[7] at 15 mg/kg/day and trimethoprim + sulfamethoxazole at 25 mg/kg twice a day orally were started, and cerebrospinal fluid (CSF), protein: 200 mg/dl, sugar: 30 mg/dl, neutophils: 40%, which was done which was suggestive of meningitis. Two-dimensional echocardiography (2D ECHO) was done suggestive of poor myocardial contractility, moderate mitral regurgitation with hypertrophic obstructive cardiomyopathy [Figure 1]a and [Figure 1]b, and dilated left atrium, so injection dobutamine and milrinone infusion were started.

Figure 1: (a and b) Two dimensional echocardiography images showing hypertrophic obstructive cardiomyopathy

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In spite of oral rifampicin and trimethoprim + cotrimoxazole, the baby was not improving clinically. On day 21, still, low-grade sepsis and thrombocytopenia were persistent, CSF was suggestive of persistent pyogenic meningitis, so injection ceftriaxone and gentamicin was added and given for 3 weeks and repeat CSF was done which was normal, and blood report was suggestive of increased platelet count, with normal CRP. Follow-up 2D ECHO was normal. Finally, the baby was discharged on the 48^th day of life with intact neurology and normal growth. Oral rifampicin and trimethoprim + sulfamethoxazole were given for 6 weeks.

Discussion

In the present case, neonatal brucellosis was caused by B. abortus and B. melintesis. Untreated Brucella during pregnancy may result as PROM, IUGR, chorioamnionitis, which can be further prevented by timely diagnosis and treatment. Before the diagnosis of congenital brucellosis,^[8] other forms of bacterial sepsis should be ruled out. In this patient, congenital brucellosis presented with persistent high CRP, persistent thrombocytopenia, and hypertrophic obstructive cardiomyopathy. In preterm neonates Congenital brucellosis mostly present at the end of first week where as in term neonate it present late, but the incubation period may vary from 1 week to 1 month. Other symptoms include pyrexia, palpable liver and spleen, and RDS in the chest X-ray. Diagnosis of brucellosis is confirmed by the presence of Brucella by polymerase chain reaction analysis of newborn's blood sample. In preterm neonates immune system is immature so serology test comes positive as delayed response. In our case, parents were not living in Brucella endemic area, though mother had a history of ingestion of unpasteurized raw milk. Congenital Brucella was best treated with rifampicin (10–15 mg/kg/day) and trimethoprim + sulfamethoxazole 25 mg/kg/day twice for 6 weeks. Meningitis associated with Brucella best was treated with ceftriaxone and gentamicin.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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