Neurological complications, including Guillain-Barre syndrome and acute transverse myelitis [1,2] following typhoid fever have been reported from typhoid endemic settings. We report a rare complication of subsultus tendinum (an involuntary twitching of the muscles of the limbs) in a young boy with blood culture-confirmed typhoid fever.

A 5-year-old boy, residing in a semi-urban settlement of Vellore town, presented to the community clinic in the area with a history of three days of fever associated with sore throat, malaise, cough, nausea, and headache. The highest temperature recorded during the episode was 103.8 °F. As a part of the SEFI (Surveillance for Enteric Fever in India) protocol, a blood culture is performed for all study children who present with three or more days of fever, and hence the child’s blood culture was sent to the laboratory [3]. Blood culture grew Salmonella enterica serovar Typhi. Following the culture result, oral azithromycin (20 mg/kg body weight) was initiated and continued for 10 days.

Fever abated on the third day following the initiation of azithromycin. Four days following fever defervescence, the child was brought back with pain in the left side of the neck and shoulder spreading down to the scapular region, with no swelling, warmth or tenderness. Two days later, the child developed twitching movements over the left shoulder and scapular region which increased over the next few days and was observed even while the child was asleep. There was an associated worsening of pain in the left neck and shoulder. There was no history of seizures, abnormal movements or loss of consciousness. There was no history of seizures in the family. His general and systemic examination including a complete neurological examination was normal except for the persistent twitching over his left shoulder and scapular regions. His investigations showed serum creatinine (0.39 mg/dL) and sodium (138 mmol/L) to be within the normal range. Given the persistent twitching, he was suspected to have epilepsia partialis continua. However, MRI of the brain with contrast and EEG were normal. He was evaluated by a pediatric neurologist and was started on clonazepam at a dosage of 0.25 mg twice daily. The twitching movements continued to persist during sleep; however, with decreased intensity and frequency. The twitching persisted for five weeks from the onset of his symptoms and then subsided. He was followed up again at eight weeks and 20 weeks after discharge, and was asymptomatic. In view of this clinical course, a diagnosis of subsultus tendinum complicating typhoid fever was made.

The burden of typhoid fever continues to remain high in India, especially in the pediatric population [4]. Complications that ensue following an episode of enteric fever are protean including neurological conditions [5]. These neurological complications can present as delirium, drowsiness, seizures, tremors, chorea, cranial nerve palsies, and even blindness. However, sparse literature mentions subsultus tendinum as a complication of typhoid fever in children. It is defined as the involuntary twitching of muscles, typically of fingers and wrists, and is classically described as one of the components of ‘typhoid state’, that occurs rarely in association with typhoid fever and occasionally with typhus fever and other bacteremias. Typhoid state is defined as a febrile state of semi-consciousness accompanied by delirium [6]. Typhoid state can be associated with carphology, which is the picking of clothes and floccillation, the state of picking at imaginary objects with the patient often found motionless and exhausted [6]. Subsultus tendinum has also been described along with the typhoid state. In this child, the twitching was not associated with delirium and it involved the muscles around the shoulder girdle rather than the fingers or wrists. Hence, we propose that subsultus tendinum need not present with the typical ‘typhoid state’, but can rarely present as localized twitching of the skeletal muscles around the limb-girdle, even without delirium. The condition may persist for a few weeks, and abates gradually with the treatment of typhoid fever. It is not clear if anti-seizure medication is mandated in this situation; however, it can be supportive in relieving the symptoms temporarily, with timely and appropriate anti-microbial therapy being the mainstay of management.

Contributors: KNS,MS: designed the concept and overviewed literature, collected the case details and prepared the initial draft; PDM,MT: managed the patient and supervised the draft; JJ: supervised and reviewed the draft; WR: supervised the draft and overall guarantor of the authenticity of the case. The final manuscript was approved by all the authors.

Funding: None; Competing interest: None stated.

REFERENCES

1. Kapoor K, Jain S, Jajoo M, Talukdar B. A rare neurological complication of typhoid fever: Guillain-Barre’ syndrome. J Pediatr Neurosci. 2014;9:148-9.

2. Mishra K, Kaur S, Basu S, Gulati P, Parakh A. Acute transverse myelitis: An unusual complication of typhoid fever. Paediatr Int Child Health. 2012;32:174-6.

3. John J, Bavdekar A, Rongsen-Chandola T, Dutta S; NSSEFI Collaborators, Kang G. Estimating the incidence of enteric fever in children in India: A multi-site, active fever surveillance of pediatric cohorts. BMC Public Health. 2018;18:594.

4. John J, Van Aart CJ, Grassly NC. The burden of typhoid and paratyphoid in India: Systematic review and meta-analysis. PLoS Negl Trop Dis. 2016;10:e0004616.

5. Udani PM, Purohit V, Desai P. Typhoid fever in children in the past and present multi-drug resistant type with special reference to neurological complications. Bombay Hospital Journal. 1999;4102.

6. Verghese A. The “typhoid state” revisited. Am J Med. 1985;79:370-2.