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Lipschütz ulcers (LU), also known as reactive non-sexually related acute genital ulcerations, are an uncommon condition that primarily affects sexually inactive girls under the age of 20 years. In this report, we describe a 13-year-old female with past medical history of periodic fever, aphthous stomatitis, pharyngitis, adenitis (PFAPA) in early childhood who presented to the emergency department (ED) for sudden onset of genital ulcerations and labial swelling. Based on her clinical presentation, negative primary infectious workup, unresponsiveness to broad-spectrum antimicrobials, and concurrent viral infection, she was diagnosed with LU. Her simultaneous presentation with aphthous stomatitis was unique and may indicate that PFAPA history potentially influences susceptibility to developing LU.

AMA Research Challenge 2024 

A case of Lipschütz ulcers in a 13-year-old with PFAPA syndrome

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Abstract Title
An Unusual Presentation of Erythrodermic Psoriasis
Background
Erythrodermic psoriasis is a rare form of psoriasis, occurring in less than 3% of patients with psoriasis. Limited information is known regarding diagnosis and treatment for this form of psoriasis, with most studies involving small groups or single patients. This unusual case of erythrodermic psoriasis outlines the presentation, diagnosis, and treatment that occurred in a 53-year-old patient who initially presented with dyspnea and a fever.
Case Presentation
The patient had a history of type 2 diabetes, psoriasis, COPD, atrial fibrillation, peripheral arterial disease, and DVT. Lab work showed evidence of leukocytosis and viral testing was done, which was negative for COVID and RSV. The patient suddenly developed a diffuse rash during the admission. A maculopapular rash developed diffusely on the upper extremities, lower extremities, trunk, and face. The rash then desquamated over the course of several days, appearing as exfoliating and scaling skin. The patient underwent two punch biopsies for diagnosis. She was treated with Hydrocortisone (2.5%) cream for the face and inguinal regions and Triamcinolone for the trunk. The patient continued this regimen daily for 2 weeks, and then alternated days for the following 2 weeks, and then stopped.
Discussion
Erythrodermic psoriasis can be challenging to diagnose and treat, and there is limited data to guide this treatment. A flare of the rash may be the initial presentation in some patients who have not had a prior diagnosis. There are many different causes and potential risk factors for a flare of erythrodermic psoriasis. The most common being a prior history of psoriasis, certain medications, and infections. A study of 50 patients with erythrodermic psoriasis found that common precipitating factors were the following: systemic corticosteroids and excessive use of topical steroids, phototherapy complications, severe emotional stress, and preceding illness.
The patient in this study had several precipitating factors worth noting, including recent diagnosis of colorectal cancer and infection with evidence of leukocytosis. She was also under a fair amount of emotional stress given her health issues and prolonged hospital stay. The patient was also receiving several medications that may have increased her risk of a flare, most notably being Vancomycin. Initial treatment is the use of topical corticosteroids and systemic therapy is considered if this does not improve symptoms. Prior case reports have shown that difficult to manage cases can be treated with Acitretin or systemic therapies such as Methotrexate.

An Unusual Presentation of Erythrodermic Psoriasis

Gout is an inflammatory disease characterized by hyperuricemia and monosodium urate crystal deposits, typically affecting peripheral joints at the extremities. We present an atypical case of the disease developing in the fingertip of a 56-year-old Middle Eastern female with a complex history of breast cancer, congestive heart failure, dilated cardiomyopathy, and left arm and hand lymphedema. After months of painful swelling and limited flexion in the left distal index finger, the lesion was initially diagnosed by her primary care provider as an abscess. The persistence of symptoms despite antibiotic treatment prompted further investigation. The patient received an excisional biopsy with histopathological examination, which revealed needle-like amphophilic crystals in the dermis accompanied by foreign body-type giant cells and histiocytes. These results, alongside elevated uric acid levels, confirmed the atypical presentation of tophaceous gout in the left index fingertip. The introduction of 300 mg daily of allopurinol significantly improved symptoms, decreasing joint pain, and the left fingertip lesion resolving. This unusual presentation of gout is secondary to a comprehensive breast cancer medication regimen and lymphedema of the left arm and hand, potentially resulting in the deposition of uric acid in this uncommon location. This unusual presentation of gout underscores the importance of considering gout in differential diagnoses, even in atypical locations.

Atypical Presentation of Gout in the Fingertip: A Case Report

Myelodysplastic syndrome (MDS) is a hematological disease manifesting as ineffective or nonfunctional hematopoiesis. It is characterized by a triad of cytopenia, morphologic dysplasia, and genetic evidence of clonality. Cutaneous manifestations have been associated with MDS in large numbers. We present a case of a MDS diagnosed through initial presentation as a lichenoid dermatitis.

Lichenoid Dermatitis Heralding Myelodysplastic Syndrome

Calciphylaxis is a disorder causing ischemic skin necrosis, typically associated with end-stage renal disease (ESRD) or those receiving dialysis. Occurrence is rare in those without ESRD, and treatment options are limited. This case report describes a patient with calciphylaxis without ESRD or history of dialysis. Treatment with sodium thiosulfate, a first line option, had to be stopped due to metabolic derangements, limiting the healing process. Diagnosis and treatment of this rare disorder is important to prevent further complications that may result.

Non-Chronic Kidney Disease-Induced Calciphylaxis: A Rare Case Report

Title: Pembrolizumab-Induced Epidermotropic CD8+ Dermatitis in a Patient with Multiple Squamous Cell Carcinomas

Background:
Pembrolizumab, a programmed death-1 (PD-1) inhibitor, has been increasingly associated with dermatologic immune-related adverse events (irAEs), varying from benign rashes to severe conditions such as Stevens-Johnson syndrome and toxic epidermal necrolysis. While the literature describes various dermatologic manifestations like lichenoid dermatitis and granuloma annulare as pembrolizumab side effects, we report a distinctive case of pembrolizumab-induced epidermotropic CD8+ dermatitis in a patient with a history of multiple squamous cell carcinomas (SCCs) and a keratin-positive malignant spindle cell neoplasm. This case adds to the evolving spectrum of pembrolizumab's dermatologic irAEs, underscoring the need for vigilance in patients receiving immune checkpoint inhibitors (ICIs).

A 69-year-old female with a history of multiple SCCs treated across various anatomical sites presented with complaints of a widespread rash. The patient reports that the rash had persisted for 20-30 years but had notably worsened following the initiation of pembrolizumab treatment two years prior. Pembrolizumab was administered as part of her treatment regimen for a keratin-positive malignant spindle cell neoplasm of the right proximal femur. She described the rash as intensely pruritic, worsening during winter months, with mild improvement during summer.

Methods:
Compiled through a retrospective review of medical records with patient consent. Diagnostic assessments were conducted using standard histological and immunohistochemical techniques, supplemented by PCR for T-cell receptor gene rearrangements to evaluate T-cell clonality.

Results
On skin examination, extensive coalescing, ill-defined erythematous patches with associated xerosis were observed. A biopsy of an eczematous lesion on the left lower back was performed to further investigate the rash's etiology. Histopathological analysis revealed hyperkeratosis, parakeratosis, and infiltration of epidermotropic lymphocytes within the epidermis. The infiltrate was predominantly composed of CD8+ T cells. Further molecular studies were negative for clonal T-cell receptor rearrangements.

Conclusion
Given the lack of T-cell clonality and the CD8+ cell-rich infiltrate, a diagnosis of pembrolizumab-induced epidermotropic CD8+ dermatitis was favored over other dermatologic conditions such as mycosis fungoides. The chronicity and the pre-existing nature of the rash, coupled with the exacerbation post-pembrolizumab therapy, underscore its drug-induced origin, distinct from typical dermatologic irAEs reported with ICIs. This case is particularly unique due to the uncommon histopathological feature of epidermotropism of CD8+ T cells, divergent from the more common CD4+ presentations seen in other cases of pembrolizumab-induced dermatitis.

The patient continued with pembrolizumab therapy for her underlying malignancy and her rash was managed with triamcinolone 0.1% topical ointment.This case adds to the literature on pembrolizumab-induced skin reactions, emphasizing the nuanced understanding needed for diagnosis and treatment, particularly in the context of a pre-existing dermatologic condition exacerbated by ICI therapy.

Pembrolizumab-Induced Epidermotropic CD8+ Dermatitis in a Patient with Multiple Squamous Cell Carcinomas

Hidradenitis suppurativa (HS), also known as acne inversa, is a chronic inflammatory disorder of the hair follicle that presents in apocrine gland-bearing areas of the body. It is characterized by nodules, abscesses, fistulae, scars, and sinus tracts in the perineal and perianal regions. The most commonly affected region is the axilla but can also present in the perineal/perianal site in 30-50% of cases. HS cannot be cured, but clinical management can control the disease. HS prevalence is unclear, estimates range from 0.00033% to 4.10% in the US population. Squamous cell carcinoma (SCC) is the second most common non-melanoma skin cancer, and accounts for 20% of skin cancer causes. SCC has been documented to be a rare complication of HS lesions. Early detection of SCC in HS patients is important for timely clinical management and treatment. The University of Southern California (USC) Institutional Review Board approved this study. Using de-identified electronic medical records on TriNetX from USC, we used ICM-10- CM codes to identify HS patients who developed skin cancer (basal cell carcinoma, squamous cell carcinoma, melanoma in situ, and malignant melanoma) from 1 day to present day post-HS diagnosis. Kaplan Meier analysis was conducted to compare survival probabilities of HS patients who developed the different skin cancers. Preliminary results show that out of 990 HS patients at USC, 20 patients develop some form of skin cancer in the HS regions (2.02%). SCC was the most common form of skin cancer arising in HS regions. The survival probability was lowest among HS patients who developed SCC (97.08%), compared to BCC (99.35%), melanoma in situ (99.41%), and malignant melanoma (99.60%). Clinicians should be aware of SCC as a complication of HS and be diligent in screening for SCC in management. SCC in HS patients is a rare, but serious complication and delays in diagnosis may lead to worse outcomes.

Squamous Cell Carcinoma in Hidradenitis Suppurativa Patients

Background: 
Scleroderma-associated digital ulcers (SSc-DUs) are seen in up to 50% of patients with scleroderma (SSc) and may precede complications including osteomyelitis and gangrene. The current literature regarding the risk factors for gangrene in SSc is scarce, as the presentation is becoming increasingly rare.
Risk factors for gangrene in unselected patients with SSc include previous digital ulcers (DUs), previous digital gangrene, and the diffuse-cutaneous subtype of SSc (dcSSc)3, which affects the visceral organs and the skin to a higher degree than the limited-cutaneous subtype of SSc (lcSSc). 
Traditional cardiovascular risk factors for peripheral arterial disease (PAD) (smoking, hypertension, dyslipidemia, diabetes mellitus, previous acute cardiovascular events) have not been consistently associated with gangrene in SSc.

Case Presentation: 
An 81-year-old female with Raynaud’s phenomenon, sick sinus syndrome status post pacemaker implantation, and well-controlled non-insulin-dependent diabetes mellitus presented to the emergency department complaining of two weeks of left foot pain.
She endorsed swelling and redness of the foot and ankle after having sustained minor trauma directly over a distal ulcer on the first digit of the affected foot. She denied prior digital ulcerations as well as a history of smoking, alcohol, and drug use. 
Laboratory analysis, including a complete blood count and a complete metabolic panel were within normal limits. Notably, the erythrocyte sedimentation rate and C-reactive protein suggested acute on chronic inflammation at 106 (normal range: 0-30 mm/hr) and 7.9 (0.1-0.9 mg/dL), respectively. Serology confirmed lcSSc with a positive anti-centromere antibody. Wound cultures obtained from purulent drainage grew Pseudomonas. Magnetic resonance imaging was contraindicated, given pacemaker incompatibility. Computed tomography and arterial ultrasonography of the extremity demonstrated no significant abnormalities. 
She was treated with six weeks of intravenous cefepime, given concerns of osteomyelitis. Soon after starting treatment, soft tissue edema subsided, and gangrenous changes were noted across the fifth digit of the left foot. Unfortunately, treatment further included digital amputation of the gangrenous digit.

Discussion: 
The currently available literature suggests that the development of gangrene would not be expected in the digital ulcers of the presented patient, given the absence of prior ulcerations and classically more aggressive subtype of SSc. The lack of established risk factors for both SSc-DU-associated gangrene and PAD in this patient suggests that SSc itself may have a unique disease course in the setting of trauma. A history of trauma may be a key element in helping clinicians categorize patients to determine which patients are likely to have increased disease burden and, therefore, benefit from more complex management. 

Conclusion: 
Due to the heterogeneity and small sample sizes in the currently available cohort studies on the topic, patients with SSc of all subtypes should be educated on the risk of complications that may follow injuries of ulcerated digits. Close follow-up and treatment after injuries may prevent digital amputation.

Patients should be educated on the risk of complications after injuries of ulcerated digits and be recommended close follow-up if these occur.

Trauma Leading to Gangrene in Digital Ulcers Affecting the Limited Cutaneous Subtype of Scleroderma

Background

Takotsubo Cardiomyopathy (TTC), or broken heart
syndrome, was first described in Japan in 1990. It is a
stress-induced cardiomyopathy in which the heart
resembles a Japanese “octopus pot”, or takotsubo.
TTC is characterized by the weakening of the left
ventricle, resulting in ballooning of the heart and left
ventricular apical akinesis, causing symptoms that
mimic acute coronary syndrome (ACS). Patients may
present with myocardial infarction (MI) - like
symptoms, show ST-segment elevation on
electrocardiogram (ECG), and have elevation in
ischemic cardiac enzymes. However, no coronary
artery blockage will be seen. Onset usually follows a
significant stressor, such as the death of a loved one,
coining the term “broken heart syndrome”. It is
hypothesized to be caused by the stunning of the
myocardium via catecholamine release. Treatment
involves ruling out ACS and MI, supportive care, and
follow up with a cardiologist to ensure resolution of
the syndrome. In the rare (<5%) case that a left
ventricular thrombus forms, systemic anticoagulation
is required.

Case Presentation

We present a case of a patient with a history
significant for chronic kidney disease and
hypertension presenting to the emergency
department with one-day history of unchanged acute
onset dyspnea and hypotension. She remarked that
she was “incredibly stressed'' recently. Labs were
significant for leukocytosis, increased troponins, and
lactic acidosis. Her ECG showed T-wave inversions in
the inferior and anterolateral leads.
Echocardiography was significant for findings
suggestive of TTC with an ejection fraction (EF) of 25-
30%. The patient was transferred to the intensive
care unit for cardiogenic shock. Following supportive
treatments, she was optimized based on her TTC,
and was discharged with cardiology follow up.
Currently, she is on goal-directed medical therapy,
wears a life-vest, and continues to have left
ventricular akinesis with an EF of 25-30%.

Discussion

Acute treatments are supportive, but vary based on
level of left ventricular outflow tract obstruction.
Traditional heart failure with reduced ejection
fraction (HFrEF) treatments have limited efficacy for
TTC-induced HFrEF. Some data indicates that TTC is
often underdiagnosed, and the true rate may be
closer to 5.7%. While most cases recover within 2
weeks, there is a significant recurrence rate of up to
4% per year. A consideration for prevention of
recurrence is screening and addressing mental health
comorbidities. It is important to note, that TTC is less
commonly also referred to as “Happy Heart
Syndrome,” so clinicians should be cognizant of TTC
patients presenting with positive symptoms.

A Heart-breaking Case of Takotsubo Presenting to a Community Emergency Department

Background:
We report two cases of seizure-like activity after accidental pediatric cannabis ingestion. As states continue to alter laws regarding sale and formulation of cannabis products, pediatric ingestion of these products remains a dynamic public health concern. Expansion of marijuana legalization has been paralleled by increasing availability of edible THC products, including ones that may be mistaken by children for snack foods like cookies and gummies. Diagnosis of cannabis intoxication in children has proven a difficult and costly endeavor. Many caregivers are reluctant to admit that a child could have been exposed to marijuana due to stigmas and fear of liability. On the other hand, physicians may fail to consider cannabis in children due to nonspecific symptoms (drowsiness, tremors, palpitations, numbness) and fear of a missed, more serious diagnosis. 

Case Presentation:
The first patient is an 8-year-old male who presented with focal shaking and abnormal behavior that progressed to generalized full body shaking. He reported muscle pain as well as auditory and visual hallucinations, a constellation of symptoms that have not been reported in the setting of pediatric cannabis ingestion. Presenting vitals were stable and bloodwork and head CT were negative, resulting in discharge home. Urine drug testing was positive for cannabinoids with a THC of 504 ng/mL. 
The second patient, a 2-year-old female, presented with agitation and neck and limb stiffening followed by multiple episodes of rhythmic truncal flexion without extremity shaking. Her vital signs were stable with normal blood work, blood cultures, urinalysis, viral testing, x-rays and head CT. Her urine drug testing was positive for cannabinoids with a THC level of 820 ng/mL. 

Discussion:
Both patients’ presentations led to extensive, expensive workups, placing patients at risk of harm. The only necessary interventions for either patient were intravenous normal saline, social services, and observation. Public health efforts must be taken to decrease pediatric exposures to cannabis, including, but not limited to, regulations on snack-food-like packaging. Additionally, more timely identification of cannabis intoxication through history and drug screening may decrease invasive and costly testing in pediatric populations. One such recommendation incudes delaying all testing other than urine drug screening in children who are stable and have bilaterally reactive pupils. Rapid, more specific urine toxin immunoassay may also facilitate more efficient diagnosis. Physician education regarding atypical case presentation, such as those addressed in this case report, may aid in more streamlined diagnosis as well as decreased cost and patient risk.


Accidental Pediatric Cannabis Ingestion Causing Seizure-Like Activity: Case Reports to Inform Diagnosis

Background
Atraumatic hemothorax presents a diagnostic challenge due to its rarity and the wide, diverse range of potential etiologies. We present a case of massive hemothorax without a clear cause, aiming to highlight the complexities of this pathology and the critical considerations necessary for accurate diagnosis.

Case Presentation
A 20-year-old male with a history of spine surgery presented to the ED following two syncopal episodes and a one-day history of nausea, vomiting, and right-sided back pain. The patient began practicing Jiujitsu two days prior but denies any significant injuries. Upon EMS arrival, systolic blood pressure was 90 mm Hg and the patient remained hypotensive after receiving two units of crystalloids. Chest X-ray revealed a large right-sided pleural effusion and thoracentesis with subsequent tube thoracostomy evacuated three liters of blood. The patient was transferred for higher level of care, and was transfused with four units of packed red blood cells, one unit of fresh frozen plasma, and a bolus of tranexamic acid. A FAST exam was negative. An additional 700-800 mL of sanguineous blood was evacuated upon a second thoracostomy with chest tubes left in place. CT angiography of the chest, abdomen, and chest did not reveal active contrast extravasation or traumatic injury of the aorta. Over the following five days, the patient’s symptoms and hemothorax resolved, chest tubes were removed, and the patient was discharged. There are no records of patient readmission.

Discussion
Massive hemothoraces, defined by blood drainage exceeding 1,000 mL after closed thoracostomy, are typically associated with high-impact traumas such as blunt or penetrating chest injuries. We present an unusual case with minimal evident trauma. Although our patient denied significant injury, it is possible that a minor incident may have been overlooked. We suspect our patient’s hemothorax was due to intercostal artery damage if he did suffer some type of injury. Regardless, minor injury alone would still not typically explain this massive hemothorax and therefore, must also consider the possibility of a massive, non-traumatic hemothorax due to coagulopathy, vascular malformations, connective tissue diseases, or autoimmune diseases. Given the unclear history, a broad differential diagnosis is crucial. This case underscores the importance of gathering a detailed history during the patient encounter and maintaining a high degree of suspicion for hemothorax in cases of refractory hypotension, even in the absence of a clear trauma history.

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A case of Lipschütz ulcers in a 13-year-old with PFAPA syndrome

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An Unusual Presentation of Erythrodermic Psoriasis

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