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Presacral malakoplakia presenting as foot drop: a case report
Tom A Yates1,2, Katie Devlin3,4, Abed Arnaout5
1Clinical Infection Unit, Clinical Academic Group in Infection and Immunity, St George's University Hospitals NHS Foundation Trust, London, UK. t.yates@ucl.ac.uk.
Background:
Malakoplakia is a rare condition characterized by inflammatory masses with specific histological characteristics. These soft tissue masses can mimic tumors and tend to develop in association with chronic or recurrent infections, typically of the urinary tract. A specific defect in innate immunity has been described. In the absence of randomized controlled trials, management is based on an understanding of the biology and on case reports.
Case Presentation:
Here we describe a case of presacral malakoplakia in a British Indian woman in her late 30s, presenting with complex unilateral foot drop. Four years earlier, she had suffered a protracted episode of intrapelvic sepsis following a caesarean delivery. Resection of her presacral soft tissue mass was not possible. She received empiric antibiotics, a cholinergic agonist, and ascorbic acid. She responded well to medical management both when first treated and following a recurrence of symptoms after completing an initial 8 months of therapy. Whole exome sequencing of the patient and her parents was undertaken but no clear causal variant was identified.
Conclusions:
Malakoplakia is uncommon but the diagnosis should be considered where soft tissue masses develop at the site of chronic or recurrent infections. Obtaining tissue for histological examination is key to making the diagnosis. This case suggests that surgical resection is not always needed to achieve a good clinical and radiological outcome.
Insights
Malakoplakia, a rare inflammatory condition, can present as soft tissue masses mimicking tumors. This case highlights successful medical management, suggesting surgery isn't always necessary for presacral malakoplakia.
Area of Science:
- Pathology
- Immunology
- Genetics
Background:
- Malakoplakia is a rare inflammatory condition characterized by soft tissue masses, often associated with chronic infections and innate immunity defects.
- Management relies on case reports and biological understanding due to a lack of clinical trials.
Observation:
- A case of presacral malakoplakia in a woman presenting with unilateral foot drop after intrapelvic sepsis post-cesarean delivery.
- Surgical resection of the presacral mass was not feasible.
- The patient received antibiotics, a cholinergic agonist, and ascorbic acid, showing a good response.
Findings:
- The patient responded well to medical management for both initial treatment and recurrence.
- Whole exome sequencing did not identify a clear causal variant.
- Histological examination is crucial for diagnosing malakoplakia.
Implications:
- This case suggests that surgical intervention may not always be required for favorable clinical and radiological outcomes in presacral malakoplakia.
- The diagnosis of malakoplakia should be considered in cases of soft tissue masses near chronic infection sites.
- Effective medical management strategies can be employed for malakoplakia.
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