Gingival swelling associated with hypoplasminogenemia

A middle-aged woman presented for evaluation of gingival swelling. She was previously diagnosed with hypoplasminogenemia and current plasminogen level was 29% (reference value: 78-130%). Intraoral examination revealed swelling of the right maxillary gingiva (Figure 1). Biopsy with routine histopathologic analysis revealed fibrinoid deposits and a mixed inflammatory infiltrate within the lamina propria, consistent with hypoplasminogenemia (Figure 2).

Figure 1
Swelling of the right anterior maxillary gingiva.

Figure 2
Amorphous fibrinoid deposits (*) in the gingival lamina propria. (A) Hematoxylin-eosin (magnification ×100), (B) trichrome (magnification ×40).

Hypoplasminogenemia (type 1 plasminogen deficiency) is commonly associated with ligneous conjunctivitis and ligneous gingivitis.^1,2and3 An oral mucosal biopsy can aid in the diagnosis of this rare condition as histopathologic evidence of fibrin deposition is highly suggestive of hypoplasminogenemia.^2and3

Acknowledgements

The authors thank Dr. Sophia Elmuradi for assistance with clinical photography.

References

• ¹
  Schuster V, Hugle B, Tefs K. Plasminogen deficiency. J Thromb Haemost. 2007;5(12):2315-22.
• ²
  Scully C, Gokbuget AY, Allen C, Bagan JV, Efeoglu A, Erseven G, et al. Oral lesions indicative of plasminogen deficiency (hypoplasminogenemia). Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2001;91(3):334-7.
• ³
  Scully C, Gokbuget A, Kurtulus I. Hypoplasminogenaemia, gingival swelling and ulceration. Oral Dis. 2007;13(6):515-8.

Publication Dates

• Publication in this collection
  Jul-Sep 2016

History

• Received
  24 Mar 2016
• Reviewed
  14 Apr 2016
• Accepted
  03 May 2016