Cerebral thrombophlebitis observed in a 10-year-old child with measles: A case report

Abstract

Context

Measles is a highly contagious disease. According to recent estimates by the WHO in 2023, over 300,000 cases of measles were recorded worldwide. Cerebral thrombophlebitis in children is a rare condition, with a wide range of clinical manifestations that can be misleading. Severe sequelae can occur if not treated early.

Case presentation

we describe a possible association between measles infection and cerebral thrombophlebitis in a 10-year-old child who was not vaccinated against measles and developed cerebral thrombophlebitis in the context of a measles infection. Diagnosis is based on clinical evaluation and imaging data. Our patient responded well to anticoagulant treatment.

Conclusion

This case underscores the potential for measles infection, particularly in unvaccinated children, to be associated with serious neurological issues such as cerebral thrombophlebitis. However, it is crucial to interpret this association with caution, as more research is needed to fully understand the connection.

Introduction

Measles is a viral illness that can lead to serious complications. Not only can it affect immunocompromised individuals, but it can also impact otherwise healthy children. In some cases, measles can even be associated with neurological complications [1]. From 2000–2023, global efforts led to a substantial decline in the measles burden. Estimated measles cases plummeted by 72 %, dropping from 36.94 million to 10.34 million, while annual measles deaths decreased by an impressive 87 %, from 800,000 to 107,500 [2]. Currently, most patients recover without any treatment, but about one-fifth require hospitalization, and approximately 30 % experience complications [3]. Furthermore, it is estimated that about 1–3 out of every 1000 people may develop measles encephalitis, which occurs more frequently in adults than in children [4]. Neurological sequelae of measles are rare but well recognized and include: primary measles encephalitis, acute post-measles encephalitis, measles inclusion body encephalitis, and subacute sclerosing panencephalitis [5]. In this article, we describe a rare case of measles infection associated with cerebral thrombophlebitis.

Case report

We report the case of a 10-year-old child who was not vaccinated against measles and had no notable medical history, the family investigation revealed no history of thrombophilia or any known predisposition to thrombosis. He was initially admitted to the pediatric emergency department of the Mother-Child Hospital in Marrakech with impaired consciousness in the context of a measles infection. The initial symptoms, appearing 15 days before presentation, included a generalized morbilliform erythema and a dry cough. This clinical picture was later complicated, nine days thereafter and in the absence of fever, by an alteration in consciousness. Upon admission, the patient was unconscious, with a Glasgow Coma Scale score of 10. His pupils were in miosis, he was afebrile at 36.4°C,without clinical signs of dehydration, capillary blood glucose level was at 104 mg/dL. The patient was tachypneic at 46 breaths per minute, with an oxygen saturation of 92 % on room air, and normal lung auscultation. He protected his airway and was tachycardic at 136 beats per minute, with a blood pressure of 123/77 mmHg and a capillary refill time of less than 3 s. Urine output was preserved. The patient was stabilized and transferred to the pediatric intensive care unit for further management.

The biological workup in intensive care revealed acute renal failure, hyperkalemia, and metabolic acidosis, along with elevated inflammatory markers, liver function tests were normal (Table 1).

Table 1.

Biological Workup at admission.

Parameter	Patient Result	Normal Values	Unit
Renal Function
Urea	2.27	2.5 – 6.7	mmol/L
Creatinine	81.75	62 – 106	mg/L
eGFR (estimated GFR)	9.72	≥ 90	mL/min/1.73 m²
Electrolytes
Potassium (K+)	7.7	3.5 – 5.0	mmol/L
Arterial Blood Gas (ABG)
pH	7.20	7.35 – 7.45
PCO2	32.9	35 – 45	mmHg
PO2	133	75 – 100	mmHg
HCO3-	13	22 – 28	mmol/L
BE	−15	−2 to + 2	mmol/L
SaO2	98 %	95 – 100 %	%
Inflammatory Markers / Hematology
White Blood Cells	12,000	4000 – 11,000	/mm³
Hemoglobin	12.8	13.5 – 17.5 (M) / 12.0 – 15.5 (F)	g/dL
Hematocrit	38.4 %	39 – 50 % (M) / 35 – 45 % (F)	%
Platelets	200,000	150,000 – 450,000	/mm³
C-reactive protein	112.60	< 5	mg/L
Lymphocytes	0.87	1.0 – 4.8	/mm³
Coagulation Profile
Prothrombin Time (PT)	80 %	70 – 100 %	%
aPTT ratio	1	0.8 – 1.2	Ratio
Liver Function Tests
ASAT ALAT	29 30	< 35–40 < 30–45	UI/L
Cerebrospinal Fluid
Appearance	Clear, colorless
White Blood Cells	2	0–5	cells/mm³
Lymphocytes	100		%
Polymorphonuclear cells	0		%
Red Blood Cells	0		%
Proteinorachia	0.59	< 0.45	g/l
Glycorachia	0.60	0.40–0.70	g/l
Blood Glucose	0.75
Culture	Sterile

The chest X-ray revealed the presence of ill-defined, bilateral opacities, more pronounced on the right side, suggesting a diffuse interstitial involvement without signs of superinfection. (Fig. 1), the diagnosis of measles was made clinically and not by serology due to the high number of patients we received during the pandemic period, contrast-enhanced brain CT scan (Fig. 2) revealed cerebral thrombophlebitis of the superior sagittal sinus, right transverse sinus, and sigmoid sinus, extending to the proximal portion of the right internal jugular vein.

Fig. 1.

Chest X-ray showing diffuse interstitial involvement, more pronounced on the right side, without signs of superinfection.

Fig. 2.

Axial view of a contrast-enhanced brain CT scan, with the red arrow pointing to a spontaneously hyperdense appearance suggestive of cerebral thrombophlebitis of the superior sagittal sinus.

The infectious workup was unremarkable (lumbar puncture, urinalysis), as well as the renal ultrasound. An MRI scan (Fig. 3) was ordered to investigate any underlying cause of the thrombophlebitis, confirming the CT scan findings of thrombophlebitis of the superior sagittal sinus extending to the lateral sinus and right jugular vein, with signal anomalies in the frontoparietal-occipital regions suggestive of edema. Fabry disease panel, and urine amino acid electrophoresis were all negative.

Fig. 3.

Cerebral venous MRI angiogram, coronal section, TIR sequence, the white arrow shows thrombophlebitis of the superior longitudinal sinus extending to the right jugular vein (green arrow).

The patient received 8 days of intravenous third-generation cephalosporins (50 mg/kg/day) probabilistically targeting a pulmonary superinfection and unfractionated heparin (300 UI/kg/day) for 4 days, monitored by APTT, followed by a switch to rivaroxaban at 10 mg/day, administered as 5 mg every 12 h.

The day 3 inflammatory workup showed an improved CRP of 76 mg/L, though leukocytosis at 15,250 /mm³ persisted. Given the ongoing renal failure, the patient received hemodialysis sessions. Neurologically, the patient showed improvement, achieving a Glasgow Coma Scale (GCS) score of 15 at discharge. Renal function also improved, allowing the cessation of dialysis after three sessions without compromising urine output. This suggests that the renal insufficiency was likely secondary to the systemic inflammatory component associated with measles. The patient was discharged on the 11th day of hospitalization with follow-up appointments initially scheduled every 15 days. During the 3-month follow-up consultation, thrombophilia and renal function tests were conducted, both yielding normal results. A follow-up Cerebral venous MRI angiogram has been planned at the 6-month mark to monitor progress.

Discussion

In this case report, we describe a 10-year-old unvaccinated child who presented with cerebral thrombophlebitis (CTP) in the context of a measles infection. This outcome highlights a rare but severe association that warrants careful consideration, especially given the rising global incidence of the disease.

A key question in our patient's presentation is whether the CTP arose primarily as a localized complication of measles-induced CNS inflammation (encephalitis) or as a sequela of a systemic hypercoagulable state driven by the severe measles infection.

While measles is primarily known for its respiratory and dermatological manifestations, the virus is also recognized for its capacity to invade the central nervous system (CNS), potentially leading to various neurological sequelae. This CNS involvement, whether through direct viral effect or secondary inflammatory processes, may predispose to complications [6].

The appearance of central nervous system (CNS) symptoms during the prodromal period or the early stages of the rash suggests a direct viral effect. CNS involvement is evidenced by clinical symptoms, histopathological changes, EEG abnormalities, and pleocytosis in the cerebrospinal fluid [7]. However, regardless of the pathogenic pathway, measles-associated encephalitis is a severe complication, with a high rate of permanent sequelae and mortality [8].

Measles-associated encephalitis is well-documented in the literature, as is cerebral thrombophlebitis as a potential consequence of infections in general. However, the specific link between measles and cerebral thrombophlebitis as a complication is not extensively present in the existing literature. This association must therefore be interpreted with caution, and further studies are needed to better understand and establish this potential connection [9].

Cerebral thrombophlebitis is a rare condition, with its annual incidence likely underestimated due to variable anatomy, rapid recanalization of the venous sinuses, and clinical polymorphism [10]. The most frequently reported symptoms in the literature include seizures, headaches, and altered consciousness [11]. As in our case, the lateral sinuses and the superior sagittal sinus are the preferred sites of cerebral thrombophlebitis [12].

The causes of cerebral thrombophlebitis are diverse. General causes are dominated by infections and anemia, alongside chronic conditions predisposing to cerebral thrombophlebitis, such as nephrotic syndrome, congenital heart disease, and systemic lupus erythematosus [13]. A retrospective study of 12 cases by Naima Baddouh et al. identified infection as the predominant cause of cerebral venous thrombosis [10],. According to Bousser et al., 6–12 % of cerebral venous thromboses have an infectious origin; systemic infections, as well as meningitis or meningoencephalitis, play a pathophysiological role in their occurrence [14]. Furthermore, Rosenstingl et al. reported that a hemostasis abnormality was found in 41 % of cases of cerebral venous thrombosis [15]. Acute kidney injury, although a rare cause, has been associated with cerebral venous thrombosis and may play a role in the pathophysiology of cerebral thrombosis [13].

However, the association between measles and cerebral thrombophlebitis remains rare and must be interpreted with caution. In our case, no preexisting risk factors or known underlying conditions, such as a hemostasis abnormality or thrombophilia, were identified, further supporting the hypothesis of a potential complication of measles. Nevertheless, other pathophysiological mechanisms, such as an excessive inflammatory response or transient coagulation abnormalities induced by the viral infection, could also be involved.

This case highlights the importance of considering neurological complications of measles. Although the available data do not allow for the establishment of a direct causal relationship, this case illustrates the need for heightened surveillance of patients with measles, particularly those presenting with neurological symptoms, and the importance of considering cerebral thrombophlebitis as a potential complication. Further studies are necessary to better understand the underlying mechanisms and confirm a potential causal link between measles and cerebral thrombophlebitis.

Conclusion

Our article explores a possible association between measles viral infection and cerebral thrombophlebitis. As such, any alteration in consciousness following measles infection should encourage healthcare providers to consider not only primary measles encephalitis, acute post-measles encephalitis, and measles inclusion body encephalitis, but also cerebral thrombophlebitis as a potential complication. However, it is important to approach this connection with caution, as the evidence remains limited, and further studies are needed to better understand and confirm this potential relationship.

Author agreement statement

We the undersigned declare that this manuscript is original, has not been published before and is not currently being considered for publication elsewhere.

We confirm that the manuscript has been read and approved by all named authors and that there are no other persons who satisfied the criteria for authorship but are not listed. We further confirm that the order of authors listed in the manuscript has been approved by all of us.

We understand that the Corresponding Author is the sole contact for the Editorial process. He/she is responsible for communicating with the other authors about progress, submissions of revisions and final approval of proofs

CRediT authorship contribution statement

Houssam Eddine SARHAOUI: Supervision. Manal MERBOUH: Writing – review & editing. Mouaffak Youssef: Supervision. Monteiro Antolivio: Writing – original draft. Younous Said: Validation.

Patient consent

Written informed consent has been obtained from the patient's parent for publication of this case report and accompanying images.

Registration of research studies

This is not an original research project involving human participants in an interventional or observational study but a case report. This registration was not required.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Ethical approval

Due to the type of article (case report), ethical approval was not required.

Funding

No fundings.