December 2007

A 4-year-old white boy was transferred from a local community hospital emergency room for evaluation and treatment of a complicated pneumonia with hypoxia (SpO₂ 84% on room air).

The history of the chief complaint began five days prior to admission when he presented to his primary care physician with fever and vomiting. He was diagnosed with a viral syndrome. The vomiting resolved but the fever persisted, reaching a temperature maximum of 104ºF. On the day of admission, he awoke complaining of right upper quadrant pain and actually requested to go to the hospital.

The evaluation at the outside ER included a normal computed tomography scan of the abdomen and pelvis secondary to concerns for appendicitis; however, the upper cuts of the scan revealed right lower lobe pneumonia with effusion. Basic laboratory tests demonstrated a bandemia and an anion gap metabolic acidosis. A blood culture was also obtained. A chest radiograph revealed bilateral pneumonia with an effusion on the right side, and treatment was initiated with vancomycin, azithromycin (Zithromax, Pfizer) and ceftriaxone (Rocephin, Roche Pharmaceuticals).

His past medical history was significant only for allergic rhinitis, eczema and molluscum contagiosum, and his immunizations were up-to-date. There had been no recent travel, animal exposure or known sick contacts. His family and social history was unremarkable.

Upon arrival to the referral center, the patient was in mild respiratory distress with a respiratory rate of 44, SpO₂ 94% to 98% on 2L O₂ via nasal cannula and right-sided splinting. Lung examination revealed decreased breath sounds in lower lung fields bilaterally with the right side greater than the left side. He had no retractions or nasal flaring. Chest radiograph on arrival again demonstrated right-sided consolidation with effusion (Figure 1). A CT scan of the chest showed right middle lobe, right lower lobe and left lower lobe consolidations with estimation of a necrotizing pneumonia with a large amount of pleural fluid on the right (Figure 2).

He was continued on intravenous vancomycin and Rocephin for possible streptococcal or staphylococcal infection, and interventional radiology was consulted for placement of a chest tube (pigtail) using CT guidance (Figures 3, 4). The small amount of pleural fluid that was obtained was Gram stain-negative and culture subsequently grew coagulase-negative Staphylococcus, which was thought to be a contaminate. He continued to have fever and respiratory distress; therefore, a repeat CT scan was done, again revealing possible necrotizing pneumonia. The pediatric surgery department was consulted for thoracotomy and chest tube placement.

On hospital day seven, he underwent a right-sided muscle sparing mini-thoracotomy with debridement and decortication. Two 20-French chest tubes were placed in an anterior and posterior position, directed toward the apex of the lung (Figure 5). Empyema fluid was again negative and had no growth.

Postoperatively, the patient developed Horner syndrome (Figure 6), with miosis and ptosis of the right eye.

What is the most likely cause of this case of Horner syndrome?

1. Pneumonia with empyema
2. Chest tube placement
3. Pigtail catheter placement
4. Thoracotomy

Answer

The most likely cause of Horner syndrome in this case is, the high placement of the chest tubes (Figure 5). The tip of the chest tube in the apex of the lung could possibly damage the sympathetic ganglion located in this area. The sympathetic ganglion in the thorax is only separated from the parietal pleura by the endothoracic fascia, which is a thin membranous layer.

A French scientist first described Horner syndrome in animal experiments as early as 1727. François Pourfour du Petit found that cutting intercostal nerves in the necks of dogs would cause ocular and facial changes on the ipsilateral side. Edward Sellick Hare, a British physician, described similar changes in a man with a neck tumor in 1838. Claude Bernard more thoroughly described the symptoms in 1852. Three American physicians, one of whom had been influenced by Bernard, described the syndrome in a man shot through the neck in 1864. However, the Swiss ophthalmologist, Johann Friedrich Horner, described the classic symptoms of miosis, ptosis and anhidrosis in a 40-year-old woman in 1869 (Whonamedit.com).

Classic Horner syndrome consists of the triad of miosis, ptosis and anhidrosis due to interruption of the sympathetic innervation of the eye. First order sympathetic nerve fibers originate in the posterolateral hypothalamus and descend uncrossed through the midbrain and pons to terminate in the intermediolateral cell column of the spinal cord at the level of T8-C1. Second order preganglionic pupillomotor fibers exit the spinal cord at T1 which places them in close proximity to the lung apex. They ascend and synapse in the superior cervical ganglion located near the bifurcation of the carotid artery.

Postganglionic pupillomotor fibers leave the ganglion and ascend with the internal carotid artery. Vasomotor and sudomotor branches are given off immediately after leaving the ganglion, and these fibers travel along the external carotid artery to supply the vessels and sweat glands of the face. The pupillomotor fibers enter the cavernous sinus, join with the fibers of the abducens nerve and enter the orbit with the fibers of the ophthalmic branch of the trigeminal nerve. They innervate the Muller muscle and pupilatory dilator via the long ciliary nerves (Bardos E, emedicine.com).

Horner syndrome is more commonly found in adults with tumors compressing this area. Other causes include surgery or other injury to this structure. Tube thoracostomy is a surprisingly rare cause of Horner syndrome. After reviewing the literature on Horner syndrome as a complication of chest tube placement, only two pediatric case reports can be found. Turkish physicians describe the case of a 3-year-old girl who developed Horner syndrome after repair of a diaphragmatic hernia. She had almost total improvement of symptoms one month after her operation.

Regarding the other choices listed, pneumonia with empyema was reported in one paper as a cause of Horner syndrome. The paper described a 3-month-old baby girl who developed Horner syndrome as a complication of pneumonia with empyema. No mechanism was proposed to explain this finding. She also had a chest tube placed for treatment of the pneumonia, but the authors did not address this as a potential cause of the findings. She was discharged home without resolution of her symptoms.

Thoracotomy with debridement is also an unlikely cause of Horner syndrome. These procedures are usually limited to the lateral, lower thoracic cavity (Figure 7). Debridement does not involve the area of the chest near the postganglionic pupillomotor fibers.

As can be seen from the chest radiographs, the pigtail catheter is also an unlikely cause. The pigtail catheter was not placed anywhere close to the apex of the lung, and even if it was, it is so flexible that it would not likely be able to cause injury to these nerve fibers.

The patient presented in this case completely recovered from the pneumonia (Figure 8), and upon follow-up with his primary care physician two weeks after discharge, his symptoms of Horner syndrome had completely resolved.

I would like to thank Denise Pautler, MD, for her research and preparation of this case. Dr. Pautler, a 2006 graduate of the Texas A&M College of Medicine, is currently a second-year pediatric resident here at Scott & White. I would also like to thank Dan McAllister, MD, Pediatric Hospitalist at The Children’s Hospital at Scott & White for contributing to this case.

For the fifth December in a row, I am asking for holiday support for our uniformed personnel, especially those separated from their families and those in harm’s way. I know first-hand that soldiers can find all sorts of ways to lift their spirits during this time of year, but support from home, even from those they do not know, makes a huge difference. If you are interested in helping a soldier this holiday season, I recommend that you check out the “Adopt-a-Unit” program at America Supporting Americans website at: www.asa-usa.org/site/PageServer?pagename=AdoptaUnitInfo.

From my family to yours, we hope you have a safe, healthy and happy holiday season. — Dr. Brien

For more information:

• Bertino RE, Wesbey GE, Johnson RJ. Horner syndrome occurring as a complication of chest tube placement. Radiology. 1987;164:745.
• Özel SK, Kazez A. Horner’s syndrome secondary to tube thoracostomy. The Turkish Journal of Pediatrics. 2004;46:189-190.
• Bhaskar G, Lodha R, Kabra SK. Unusual complications of empyema thoracis: diaphragmatic palsy and Horner’s Syndrome. The Indian Journal of Pediatrics. 2006;73:941-943.