Intracranial hypotension as a unique complication of the rupture of an anterior sacral meningocele into a recurrent pilonidal cyst

Background: Intracranial hypotension (IH) is an underdiagnosed, but important cause of new-onset, daily persistent headache, especially among the young- and middle-aged population. It results from a cerebrospinal fluid (CSF) leak with subsequent lowered CSF pressure. Case Description: A 37-year-old female presented to the emergency department with sudden onset severe headaches. Two years earlier, she had undergone surgery for resection of a pilonidal cyst (PC). The night before admission, she had watery discharge from the recurrent PC and severe diffuse positional headaches associated with photophobia and neck pain. The head computed tomography showed pneumocephalus in the posterior fossa and a spine magnetic resonance imaging revealed an anterior sacral meningocele (ASM) in close contact with the recurrent PC. A final diagnosis was made of headaches due to IH. The leakage site was the rupture of the ASM in the PC. The surgical repair of the ASM was achieved suturing two overlapping dural flaps. There was no more CSF leakage from the PC and the headaches disappeared. Conclusion: This is a unique case of IH due to the rupture of an ASM into a recurrent PC. The association of an ASM and PC, at the best of our knowledge, is unique. Moreover, the fistulation of the ASM to the PC is exceptional. ASM can be successfully closed with a posterior approach, using two overlapping dural flaps.

Anterior sacral meningocele repair assisted by intraoperative intrathecal fluorescence and 3D printing model: illustrative case

BACKGROUND Marfan syndrome is rarely accompanied by anterior sacral meningocele (ASM) resulting from erosion of the sacrum by dural ectasia. ASM may induce symptoms due to severe mass effects. ASM may also mimic ovarian cysts, and the risk of cerebrospinal fluid (CSF) leakage is high if spontaneous rupture of the cyst occurs. In this study, the authors presented a rare case of ASM with iatrogenic CSF leakage in a 34-year-old woman with suspected Marfan syndrome. OBSERVATIONS The patient initially presented with a giant ASM that was first misdiagnosed as an ovarian cyst. Previously, it had been partially resected, which was followed by iatrogenic CSF leakage. Symptoms of intracranial hypotension, including postural headache and dizziness, developed within 1 month. Brain magnetic resonance imaging (MRI) showed pituitary enlargement, bilateral subdural effusion, and tonsillar herniation. Preoperative computed tomography myelography provided three-dimensional (3D) examination of the deformed sacrum and CSF leakage site. Transabdominal approaches led to primary repair, and repair of the meningocele was achieved by intraoperative fluorescein fluorescence and 3D printed model–guided polymethyl methacrylate bone cement reconstruction. No CSF leakage or recurrent ASM was found at the 1.5-year follow-up visit. LESSONS Intraoperative intrathecal fluorescence and 3D-printed models are useful for ASM repair. Preoperative MRI is helpful for differentiating ASM from other causes of a huge pelvic mass, including ovarian cyst.

Currarino syndrome presenting as a cerebrospinal fluid leak from the dermal sinus tract: case report

Currarino syndrome is an autosomal dominant condition with variable expressivity and penetrance that is associated with several classic features: sacral dysgenesis, presacral mass, and/or anorectal anomalies. The authors present a unique case in which the patient’s initial presentation was a CSF leak from a sinus tract. The sinus tract was identified and disconnected from the thecal sac, obliterating the anterior sacral meningocele. This case represents a unique scenario in which Currarino syndrome manifested as a CSF leak from a dermal sinus tract.

Anterior sacral meningocele complicated by rectothecal fistula and rectorrhea: A Case report and review of the literature

Background: Anterior sacral meningocele (ASM) is a rare congenital anomaly. It is characterized by herniation of the dura through a defect in the anterior sacrum. Rarely, however, it may extend to the rectal area through a rectothecal fistula with or without rectorrhea. Case Description: Here, we present a case of ASM associated with a rectothecal fistula and rectorrhea. Surgical closure of the ostium of the cyst through a posterior approach resulted in long-term improvement Conclusion: An ASM with both rectothecal fistula and rectorrhea is extremely rare.