Khan NAJ, Ullah S, Alkilani W, Zeb H, Tahir H, Suri J.
Sinking Skin Flap Syndrome: Phenomenon of Neurological Deterioration after
Decompressive Craniectomy. Case Rep Med. 2018 Oct 23;2018:9805395.
Abstract
Sinking skin flap syndrome is rare phenomenon that occurs in
patients with large craniectomies. Alteration in normal anatomy and
pathophysiology can result in wide variety of symptoms including altered mental
status, hemodynamic instability, and dysautonomias. Management is largely
conservative. We here present a case of a patient with large craniectomy who
was admitted to our hospital with pneumonia. Later on, he developed worsening
mental status and CT head revealed sinking skin flap with significant midline
shift. This is a very rare case of neurological deterioration after
craniectomies, commonly known as sinking skin flap syndrome. To our knowledge,
only few cases have been reported so far.
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From the article
An 18-year-old male, nursing home resident, presented to the
hospital with labored breathing and acute respiratory failure. The patient at
baseline was nonverbal with a Glasgow coma scale (GCS) of 10/15. Approximately
a year prior to presentation, the patient had suffered from traumatic brain
injury after a fall. He underwent large left-sided craniectomy with a bone flap
and placement of VP shunt at an outside hospital at the time. At initial
presentation to our hospital, the patient was started on broad-spectrum
intravenous antibiotics to cover for pneumonia. On day 3, the patient was noted
to have sinus bradycardia with heart rate in the 40 s and low blood pressure at
90/60. On examination, the patient had a GCS of 8/15 with unequal pupils.
Emergent CT head revealed sinking skin flap syndrome with paradoxical brain
herniation and 19.9 mm midline shift (Figure 1). The patient was placed in
Trendelenburg position and transferred to the intensive care unit for close
monitoring. An urgent neurosurgery consult was also obtained, and cranioplasty
was advised but the family deferred. The patient’s VP shunt was adjusted to
increase the intracranial pressure. A repeat CT scan after 5 days revealed
stable midline shift with no interval changes and improvement in mental status
(Figure 2).
The syndrome of trephined is an uncommon postoperative
complication that usually occurs after months of surgery in patients who
undergo large craniectomy for various reasons like traumatic brain injury,
malignant middle cerebral artery infarction, contusions, and subdural
hematomas. The first case of sinking skin flap syndrome was reported by Yamamura
et al. back in 1977. After that, sinking skin flap syndrome has been reported
fairly in the literature. In a study of 108 patients performed back in 2008 who
underwent decompressive crainectomy, syndrome of trephined was reported in 13%
of patients between 28 and 188 days after the surgery.
Patients often present with symptoms of neurologic
deterioration including new onset headache, dizziness, mood changes,
fatigability, or seizures. Some patients present with delayed dysautonomic
symptoms like postural hypotension and urinary and bowel dysfunction. Physical
examination often reveals a large skull defect with an overlying depressed skin
flap. The ubiquitous CT findings include paradoxical herniation, sunken skin
flap sign, and deviation of the midline structures. The CT head without
contrast of our patient revealed small slit-like ventricles and left-sided
craniectomy defect with a sunken brain/skin flap. In addition, it was noted
that the adjacent brain had an outwardly concave appearance with an associated
midline shift to the right by 19.9 mm (Figure 1).
The pathophysiology of this syndrome is not clear. Some
authors suggest that large craniectomies convert the cranium from a closed box
to an open cavity which alters the brain pathophysiology. The atmospheric
pressure and gravity overwhelms the intracranial pressure which leads to
paradoxical herniation and sunken skin flap syndrome. Langfitt TW theorized
that atmospheric pressure is directly transmitted to the intracranial cavity,
which increases inward shifting of the scalp over the surgery site.
The treatment options in this catastrophic syndrome are
limited and not clearly defined. The primary goal of treatment in syndrome of
trephined is the restoration of external pressure that is exerted by the
depression of the craniectomy site. The data supporting conservative management
in patients with neurological deterioration are lacking. However, trendelenberg
position largely reverses the paradoxical herniation in this syndrome. In some
cases, intrathecal saline infusion was found to be effective in reversing the
impending herniation.
The principal treatment option available currently is
cranioplasty. It corrects the abnormal CSF dynamics by lowering the local
intracranial pressure, thus improving postural blood flow and cerebral glucose
metabolism.
Park HY, Kim S, Kim JS, Lim SH, Kim YI, Lee DH, Hong BY.
Sinking Skin Flap Syndrome or Syndrome of the Trephined: A Report of Two Cases.
Ann Rehabil Med.2019 Feb;43(1):111-114.
Abstract
Decompressive craniectomy (DC) is commonly performed in
patients with intracranial hypertension or brain edema due to traumatic brain
injury. Infrequently, neurologic deteriorations accompanied by sunken scalp may
occur after DC. We report two patients with traumatic subdural hemorrhage who
had neurologic deteriorations accompanied by sunken scalp after DC. Neurologic
function improved dramatically in both patients after cranioplasty. Monitoring
for neurologic deterioration after craniectomy is advised. For patients showing
neurologic deficit with a sunken scalp, early cranioplasty should be
considered.
Dillen WL, Pittman TA, Grupke SL. Novel Temporary Treatment
for a Severe Case of Syndrome of Trephined. World Neurosurg. 2018
Dec;120:200-204.
Abstract
BACKGROUND:
Syndrome of the trephined is a unique neurosurgical condition
that is seen in patients that have undergone craniectomy. While the symptoms of
the condition range from mild to severe, the only definitive treatment for the
condition is replacement of the bone flap. This article presents a novel,
temporary treatment for syndrome of the trephined in a patient with severe
symptoms who was unable to undergo immediate cranioplasty due to infection.
CASE DESCRIPTION:
A 25-year-old gentleman with a history of trauma resulting
in hydrocephalus, craniectomy, and eventually ventriculoperitoneal shunt
placement presented with a cranial wound infection requiring removal of his
bone flap. While being treated with antibiotics, with his bone flap removed, he
developed severe syndrome of the trephined. An emergency bedside procedure was
developed and executed to treat his condition.
CONCLUSIONS:
Treating syndrome of the trephined with an external suction
device proved useful and lifesaving fort the patient presented. Such a device
can be made with common supplies found within any hospital. The technique used
to treat the patient is novel and may be useful for others to consider if ever
faced with a similar situation.
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