Baastrup’s disease  :  A rare case report
A 50 years old female farmer presented to us with insidious onset, gradually progressive low backache of one year’s duration.  The low backache was activity related. There was history suggestive of neurogenic claudication with the claudication distance being restricted to less than 100 meters.  Non-operative treatment taken during this period had not been effective.

On examination, palpable steps were present at L3-4 and L4-5 levels.  The steps reduced on flexion and increased on extension.  Local spinal tenderness and paraspinal muscle spasm was present.  Extension of the spine was significantly restricted and painful.  No significant tension or localizing signs were present.  No neurological deficit was noted.
 
X-rays (fig. A&B) of the lumbosacral spine in the anteroposterior and lateral views showed the presence of grade I anterolisthesis of L3 over L4 and grade II anterolisthesis of L4 over L5.  There was loss of L4-5 disc space with sclerosis of the adjacent endplates.  Significant facetal hypertrophy was noted at both the levels.  MRI (T1 & T2 images) showed the presence of neoarthrosis between the spinous processes of L3 & L4 with associated interspinous bursitis (fig:C, yellow arrow).  There was also a cyst in the midline in the posterior epidural space at the same level as the interspinous cyst, with resultant canal compromise (fig: C, red arrow).  There were minimal disc bulges at L3-L4 and L4-5 levels. The presence of the interspinous cyst was suggestive of Baastrup’s disease.

Since the beginning of this century, Baastrup’s disease has been a source of controversy as regards its painful nature and its appropriate treatment. Bywaters and Evans noted pseudojoints, chondroid metaplasia, enchondral ossification and gross osteoarthritic bone erosion with eburnation in their autopsy study on these patients. Clinically, these may produce pain on extension especially if there is lordosis as with obesity, hip limitation and in champion swimmers. Early reports indicated relief in pain on injection of local anaesthetic agents.  Bywaters suggested that since there is gross osteoarthritic bone erosion in the approximating spinous processes and since bone is supplied with sensory nerve endings; this must be a common cause of localized backache, episodic in nature.  Beckers incriminated the accompanying hyperlordosis as the cause of pain.  Similar views were expressed by Resnick.  Haig et al reported that diffuse fatty replacement of the paraspinal muscles, perhaps due to a compartment syndrome or other vascular event, might have a role in the pathogenesis of Baastrup’s disease.  Macnab felt that “kissing spines” cannot occur in the absence of an unstable disc segment and in the balance of probabilities; it is the associated disc degeneration rather than the bony opposition of the spinous processes that is the cause of the patient’s symptoms. Goto et al reported cervical myelopathy due to osteophytic lipping and pseudarthrosis between the posterior tubercle of C1 and the spinous process of C2.  Hazlett reported the association of spondylolisthesis and Baastrup’s disease.



A                                            B                                              C

D                                            E                                                F



Surgical treatment was undertaken for our patient.  Through the posterior midline approach, careful dissection was done to identify and dissect out the interspinous cyst.  Attempt at excision of the intact cyst was made. This revealed the presence of a communicating extension between this cyst and the second cyst in the posterior epidural space, through the midline cleft in the ligamentum flavum.  The entire cyst was excised. The kissing spinous processes were then excised. Thorough decompression of the spine was done. Stabilization of both the segments (L3-4 & L4-5) was done with posterior Moss Miami fixation, along with intertransverse fusion (fig. E & F).


Subsequent to the surgery, the patient noted significant subjective improvement.

To the best of our knowledge, this is the first case report of Baastrup’s disease causing a cyst formation of an hourglass configuration leading to epidural extension and compression of the neural elements.  It is our contention that there could be a dynamic element of compression involved in such cases.  The epidural part of the cyst could be increasing in size on extension with a corresponding decrease in size in the interspinous part; the phenomenon being reversed on flexion.  We however, have not been able to objectively demonstrate this in the present case report.  We feel that a dynamic MRI would be the best way to demonstrate this and it should be an essential investigation in future studies of a similar kind.