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| CASE REPORT |
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| Year : 2023 | Volume
: 3
| Issue : 2 | Page : 405-408 |
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Rare presentation of bilateral congenital toxoplasmosis in newborns
Mahak Bhandari1, Anubhav Goyal2, T Lekha3, Tony Mampilly4, Sindhu T Stephan5, Anantharaman Giridhar6
1 Department of Vitreo – Retina, Giridhar Eye Institute, Kochi, Kerala, India
2 Consultant Surgical Vitreo-Retina and ROP Services, Giridhar Eye Institute, Ernakulum, Kerala, India
3 Retina and Uvea Consultant, Department of Retina, Giridhar Eye Institute, Kochi, India
4 Department and Chief of Neonatology, Lisie Hospital, Kochi, Kerala, India
5 Department of Pediatrics, Government Medical College, Ernakulum, Kerala, India
6 Department of Vitreo – Retina Services, Giridhar Eye Institute Kochi, Kerala, India
| Date of Submission | 25-Oct-2022 |
| Date of Acceptance | 02-Jan-2023 |
| Date of Web Publication | 28-Apr-2023 |
Correspondence Address:
Anubhav Goyal
Consultant Surgical Vitreo-Retina and ROP Services, Giridhar Eye Institute, Ernakulum, Kerala
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/IJO.IJO_2577_22
Congenital toxoplasmosis is asymptomatic in 85% of the cases and can flee detection due to a lack of a standard screening protocol. Management of foveal-threatening Toxoplasma retinochoroiditis is critical and involves pyrimethamine and sulfadiazine as the first-line treatment, but safer drugs like clindamycin and azithromycin are also available. We report two cases of bilateral congenital Toxoplasma retinochoroiditis in infants who were screened in the newborn intensive care unit (NICU), to highlight the importance of timely treatment with a combination of steroids and antibiotics in order to prevent vision-threatening sequelae.
Keywords: Clindamycin, methylprednisolone, retinochoroiditis, Toxoplasma, ventriculo-peritoneal (VP) shunt, right eye (RE), left eye (LE)
How to cite this article:
Bhandari M, Goyal A, Lekha T, Mampilly T, Stephan ST, Giridhar A. Rare presentation of bilateral congenital toxoplasmosis in newborns. Indian J Ophthalmol Case Rep 2023;3:405-8 |
How to cite this URL:
Bhandari M, Goyal A, Lekha T, Mampilly T, Stephan ST, Giridhar A. Rare presentation of bilateral congenital toxoplasmosis in newborns. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Jun 2];3:405-8. Available from: https://www.ijoreports.in/text.asp?2023/3/2/405/374957 |
Congenital toxoplasmosis is one of the most common types of asymptomatic intrauterine infection caused by the protozoan parasite Toxoplasma gondii via vertical transmission during pregnancy.[1] In India, 56,737–176,882 children per year are born with a probable risk of congenital toxoplasmosis. A prevalence rate of 22.4% (8.8%–37.3%) has been reported with an overall IgM positivity of 1.43% in the Indian scenario.[2] The classical triad of congenital toxoplasmosis is chorioretinitis, intracranial calcification, and hydrocephalus. Timely detection and management of congenital Toxoplasma retinochoroiditis is the main concern. The earliest detection can be made by performing polymerase chain reaction (PCR) on the amniotic fluid, but the most practical method is by serological testing of maternal and fetal blood, both of which are cumbersome due to the requirement of amniocentesis and the false-positive rates, respectively.[3] Testing is usually done only on the presentation of clinical symptoms, due to which a large number of asymptomatic patients go undetected only to present to us during adulthood. Management of symptomatic congenital toxoplasmosis is done by a combination of pyrimethamine–sulfadiazine along with steroids (classical triple therapy).[4] The differential diagnoses of Toxoplasma consist of other perinatal infections such as rubella, cytomegalovirus, syphilis, and herpes. We present two cases of bilateral congenital toxoplasmosis at birth to emphasize on early diagnosis and timely management with safer antibiotics in combination with steroids for better outcome.
| Case Series | |  |
Case 1
A 2-day-old full-term asymptomatic infant born with 3200 g birth weight was advised random screening eye examination by a neonatologist for thrombocytopenia (platelet count- 30,000/μL) detected on routine blood tests done before discharge. Mother was also asymptomatic with no significant antenatal history of fever or infection but was a pork meat lover. The infant was diagnosed to have organized macular scar in the right eye (RE) and fovea-threatening active retinochoroiditis lesion in the left eye (LE) [Figure 1]. The baby did not have any other systemic involvement at that time in the neonatal intensive care unit (NICU). Computed tomography (CT) scan brain showed diffuse intracranial calcification in the frontoparietal area [Figure 1]. On performing a TORCH panel (Toxoplasma, others like syphilis, rubella, cytomegalovirus, and herpes), the newborn was found to be positive for both IgG and IgM Toxoplasma titers. Infant's cerebrospinal fluid (CSF) PCR testing for Toxoplasma was also positive. The newborn was started on intravenous clindamycin (20 mg/kg/day) in three divided doses for 2 weeks, followed by oral clindamycin (20 mg/kg/day) for 6 weeks duration. Intravenous pulse methylprednisolone (10 mg/kg body weight) was started after 2 days of initiation of clindamycin and was given for 5 days, followed by oral prednisolone (1 mg/kg/day) for 4 weeks with tapering doses and stopped. Subsequent second week follow-up showed significant resolution of retinochoroiditis lesion with scar formation and sparing of fovea in the OS [Figure 2]. Since the infant had intracranial calcification on CT, he was kept on serial monitoring for the development of hydrocephalus. At 2 weeks, when the infant developed hydrocephalus, he was referred to the neurology department where they advised ventriculo-peritoneal shunt. The patient was lost to follow-up after that. | Figure 1: (a) Color fundus photo of the right eye showing dark pigmented macular scar (white arrow) at birth. (b) Color fundus photo of the left eye showing fresh foveal-threatening chorioretinitis lesion at birth (blue arrow). (c) CT scan showing multiple calcified hyperreflective lesions in the frontoparietal region. CT = computed tomography
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 | Figure 2: Color fundus photo of the left eye showing healed scar in the juxtafoveal area (just sparing fovea) after 2 weeks of treatment of the newborn with intravenous pulse and oral steroids with oral clindamycin
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Case 2
A premature infant of 31 weeks gestation with 1700 g birth weight was admitted in the NICU from day 1 of birth for multiple systemic manifestations of respiratory distress, shock, hepatosplenomegaly, and jaundice. Antenatal scan revealed pleural effusion and ascites. Suspecting intrauterine infection in the mother, the infant was tested for TORCH panel, which revealed positive IgG and negative IgM Toxoplasma titers. The newborn was advised early eye screening at 1 week of birth. Fundus examination showed active retinochoroiditis lesion in fovea with signs of grade 1 vitritis in OD and macular scar in OS [Figure 3]. CT scan brain showed diffuse intracranial calcification in the frontoparietal area, suggestive of TORCH infection [Figure 3]. The newborn was planned to be put on oral pyrimethamine 1 mg/kg twice a day loading dose for 2 days, followed by 1 mg/kg once a day for 6 months and the same dose three times a week till the age of 1 year. Fifty mg/kg BD dose of sulfadiazine was advised till the age of 1 year and folinic acid 10 mg three times a week till 1 week after stopping pyrimethamine. Topical prednisolone treatment was also given, but the patient was lost to follow-up after the loading dose of pyrimethamine and came to us after 1 month with complete resolution and scar formation in RE, finally resulting into macular scar in both eyes [Figure 4]. | Figure 3: (a) Color fundus photo of the right eye showing fresh foveal chorioretinitis lesion at birth (blue arrow). (b) Color fundus photo of the left eye showing dark pigmented macular scar at birth (white arrow). (c) CT scan showing multiple calcified hyperreflective lesions in the frontoparietal region. CT = computed tomography
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 | Figure 4: Color fundus photo of the right eye showing healed foveal scar after 1 month follow-up
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| Discussion | | |
We are reporting case series in which the two cases managed differently, signifying the importance of early diagnosis, timely treatment, and follow-up for treatment of bilateral congenital toxoplasmosis.
The first case was a completely asymptomatic infant who was diagnosed after the pediatrician had a keen suspicion based on the nonspecific finding of thrombocytopenia and requested for an ophthalmic evaluation. Based on Toxoplasma positive titers and the classical clinical triad, the treatment was started, which saved the infant's vision. The recommendation to repeat the serology testing after 10 days to rule out false-positive patients was not required to be made as the CSF (gold standard test) had a PCR positivity for toxoplasmosis. The systemic manifestations of toxoplasmosis came later. Randomized clinical trials have studied the efficacy of intravitreal clindamycin,[4],[5] but the intravenous route is usually followed for administration along with the classical triple therapy.[6] Clindamycin and azithromycin are safe and effective alternative anti-microbials in cases showing atopy or severe intolerance to sulphonamides.[7]
Approximately 60%–80% of the infected infants are completely asymptomatic at birth but may gradually develop ocular and neurological symptoms in the first few months of life.[6] Contrarily, both our cases had active lesion at birth and were diagnosed within the first week of birth. The presence of necrotizing retinitis in brush fire pattern, along with immunocompromised state and presence of positive serology would help to differentiate the diagnosis from cytomegalovirus. The classical triad of rubella includes cataract, cardiac abnormalities, and deafness, which was not present in either of our patients. An infant with congenital syphilis would have facial features such as saddle nose malformation and snuffles. A complete TORCH panel would help to direct us to the perinatal infection that is being dealt with.
Diagnosis of Toxoplasma infection during pregnancy depends mainly on serological screening or the detection of fetal ultrasonic abnormalities.[8],[9] Along with the classical triad, other features such as jaundice, hepatosplenomegaly, anemia, and thrombocytopenia are also classic manifestations of congenital toxoplasmosis.[10] Our second case presented with all the typical features and was diagnosed early but was lost to follow-up and presented with poor visual sequalae, suggesting the need for a complete treatment for better visual outcomes. The authors would like to state that till date, there is no reference method for IgM detection and only 49% of the IgM-positive samples are detected to be positive. In this scenario, a complete systemic and ocular examination can help the clinician conclude the diagnosis.[11]
Monitoring of infants either on clindamycin or pyrimethamine should include complete blood count weekly for the first 2–3 weeks and then every 2 weeks. Liver and renal function tests are monitored every 3–6 months. Steroids can cause side effects such as hyperglycemia, hypertension, sepsis, and growth retardation if given for a long term, which was not the case in our series. Multidisciplinary approach along with the neonatologist is required when dealing with infants.
| Conclusion | | |
In conclusion, to the best of our knowledge, our case series seems to be the first one to document the complete resolution of ocular toxoplasmosis by intravenous clindamycin along with steroids at birth. We would recommend the intravenous route for steroids along with antibiotics in cases where there is sight-threatening ocular lesion along with inflammatory hydrocephalus.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names 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.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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