A 50 year-old man with fever and necrotizing leg ulcers

Presented by Dr. Henry Foong
Ipoh, Malaysia

The patient is a 50-year-old chef who presented initially with blisters on the left leg. Within 3 days, the leg had swollen, painful with redness over the left leg.  He was admitted to the local hospital and was treated by an orthopedic surgeon for cellulitis for 10 days.  He requested AOR (at own risk) discharge and came in to seek for 2nd opinion.  His other medical history included COAD and hypertension.

Physical examination revealed an anxious man, breathless, tachyphniec with hight temperature of 38 deg C. His left leg appeared edematous, extensively inflamed from the foot to the knee, tender, with 3 large and deep necrotic with sloughy ulcerations on the left lateral malleolus, dorsum of left foot and ankle. There was no purplish or undermining edge. The peripheral pulses dorsals pedis was good.

Diagnosis: Necrotising cellulitis left leg

Swab was taken from the leg wound but did not grow any pathogens. He was started on IV ceftriazone 1 gm bd, wash with dermazine solution, bactigrass followed by gamjee dressing.  His blood counts and biochemistry was unremarkable except severe hypoalbuminemia. He was given IV albumin 50mg daily for 3 days.  A skin biopsy was done to exclude pyoderma gangrenosum.  His CXR showed hyperinflated lungs suggestive of emphysema.  His X ray of the left was unremarkable. His fever persisted. What kind of wound dressing would you recommend? What other empirical antibiotics would you recommend - the culture did not grow any pathogens. 

 

Reference:

1. Unna Boot Efficacy in Dermatologic Diseases

Gabriella Santa Lucia et. al.J Am Acad Dermatol. 2020 Nov 25;S0190-9622(20)33059-0.

Conclusion: Unna boot (UBs) are an inexpensive and noninvasive treatment strategy in which compression, antioxidants, physical restraint, and improved topical medication absorption enhance healing as well as quality of life measure. Even though the UB has been employed successfully by dermatologists for over a century, prior research regarding diseases treated, concurrent therapies used, and adverse events reported is minimal and UBs remain underutilized. Our findings suggest clinicians should consider using UB for a wide variety of dermatologic diseases when conservative management is a viable option. These results highlight that UBs are effective and well tolerated across a spectrum of pathologies, but also versatile in the locations where they can be applied.

Oral Hairy Leukoplakia in a Healthy Teen

The patient, a 14-year-old girl, who presented with a mildly painful process on the lateral borders of the tongue for about 4 months. She is a healthy child whose only medication is oral contraceptives for heavy menstrual cycles.  She has never used inhaled steroids or nose drops; but did have mononucleosis at 4 or 5 years old. There is no history of blood transfusions.  The affected areas are sensitive when she eats spicy or acidic foods.

OE: The examination shows rough, whitish papillae on the lateral margins of the tongue.  KOH prep was negative for Candida

Clinical Images:

October 2020 (taken by patients mother:

January 2021 (taken at dermatologist office)

Diagnosis: Oral Hairy Leukoplakia (OHL) must be considered. Although OHL was first described in association with HIV/AIDS, it has been reported in otherwise healthy individuals.

OHL was first described in 1984, and initially all OHL patients had HIV/AIDS.  Over the years, it has been seen in people with other immunedeficiencies and even in patients with normal immune systems.  This patient has no risk factors for immune compromise or HIV/AIDS. The question Dr. St. Pierre asks is how aggressively this 14 year-old should be worked up?  Florid OHL is much more impressive in its appearance.  This patient's lesions are subtle and appear to be resolving without treatment.

3.19.20 Addendum:
CBC and HIV tests are both normal.  We are considering the diagnosis of "oral frictional keratosis" in this otherwise healthy teenager.  Further history reveals that she is seeing an oral surgeon for bruxism.  Her tongue is symptomatic and she complains of pain when eating acidic foods. (See reference 5)

References:.

1. Oral Hairy Leukoplakia
Manu Rathee  1 , Prachi Jain  2
In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2020 Jan.
2020 Apr 22.  Free Full Text.

2.  Darling MR et al. Oral Hairy Leukoplakia in Patients With No Evidence of Immunosuppression: A Case Series and Review of the Literature. J Can Dent Assoc 2018 May;84:i4.
Abstract: Objectives: Oral hairy leukoplakia (OHL) is caused by Epstein-Barr virus (EBV) and is often associated with HIV and other immunosuppressive conditions. It is rare in HIV-negative patients, but has been reported in patients who use immune-modulating medications (e.g., cyclosporine).

Study design: A series of 7 new cases of OHL among HIV-negative patients is described. Langerhans cells were counted using an immunoperoxidase stain for CD1a and light microscopy.

Results: The 7 patients were male, ranging in age from 26 to 69 years. Clinically, all lesions were diagnosed as leukoplakia on the lateral border of the tongue. Microscopic examination revealed hyperparakeratosis and candidiasis in some cases, acanthosis and a band-like zone with clearing of cells in the upper spinous layer, which were EBV-positive by in-situ hybridization. There was a significant decrease in Langerhans cell counts in OHL patients.

Conclusion: OHL can occur in HIV-negative patients.

3.  Shanahan D et. al. Oral hairy leukoplakia in healthy immunocompetent patients: a small case series. Oral Maxillofac Surg. 2018 Sep;22(3):335-339.
Conclusion: Physicians must have a high index of suspicion for OHL when considering a differential diagnosis for white patches on the lateral borders of the tongue in apparently healthy immunocompetent patients. OHL should no longer be solely attributed to HIV infection, or immunosuppression. Greater awareness of OHL may lead to further cases in immunocompetent people being reported, particularly as our population ages.

4. Kyle Burke Jones, Richard Jordan. White lesions in the oral cavity: clinical presentation, diagnosis, and treatment. Semin Cutan Med Surg. 2015 Dec;34(4):161-70

Keywords: geographic tongue; hairy tongue; leukoedema; nicotine stomatitis; oral frictional hyperkeratosis; oral leukoplakia; oral lichen planus; oral lichenoid reaction; oral squamous cell carcinoma; smokeless tobacco keratosis; white sponge nevus. (see comment # 7)

5. Sook-Bin Woo, Dorothy Lin. Morsicatio mucosae oris--a chronic oral frictional keratosis, not a leukoplakia. J Oral Maxillofac Surg. 2009 Jan;67(1):140-6.
Conclusions: MMO is a form of chronic oral frictional keratosis that has no malignant potential, and should be signed out as such and not merely "hyperparakeratosis and acanthosis" so that it can be removed from the category of leukoplakia where it does not belong.