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07 March 2023: Articles

AIDS-Related Kaposi Sarcoma Associated with Steroid-Unresponsive Periorbital Lymphedema that Responded to Chemotherapy

Challenging differential diagnosis, Diagnostic / therapeutic accidents, Unusual setting of medical care, Rare disease, Educational Purpose (only if useful for a systematic review or synthesis)

Yuhao Zeng * , Rahul Prasad A , Rachel D. King * , Rajshri Joshi E , Jason Lane B , Sterling Shriber E , Faris Shweikeh E , Yani Zhang A*

DOI: 10.12659/AJCR.938801

Am J Case Rep 2023; 24:e938801

Table 1. Case reports on periorbital edema in AIDS-KS.

SourceAgeSexDescription of KS-associated lymphoedemaCD4 (cells/µL)Viral load (copies/mL)TIS stagingTherapy (duration)DiagnosisOutcome
Frans et al (1994) []25 56MGeneralized non-pitting edema, especially in the face (periorbital region)538T1, I0, S0INF-αLymphangio-matous variant of KS“Complete regression of the edema”
Bossuyt et al (1995) []26 57MPronounced non-pitting edema, initial on the feet and lower leg and later spreading gradually to the hand and face528T1, I0, S0INF-αLymphangioma-Like variant of AIDS-KS“Progressive regression of edema”
Zidar et al (1995) []6 27MSevere periorbital and facial edemaT1, Ix, SxDoxorubicin, vincristine, bleomycin (every 2 weeks ×4 months)AIDS-KS“Resolved rapidly”
Schwartz et al (1995) []27 32MEdematous face and periorbital ecchymosis below each eyeT1, Ix, S0Ecchymotic KS
Hengge et al (2000) []28 29MSevere lymphoedema of lower extremities, anasarca, periorbital edema, and multiple sarcomatous hyperkeratotic and erosive lesions on the trunk, both legs, and feet15T1, I1, S0Bleomycin, vincristine and doxorubicinAIDS-related hyperkeratotic KSPartial remission of 1 year duration
Bautista et al (2001) []29 29MFacial and periorbital edema110T1, I1, S0Doxorubicin (6 weeks)AIDS-KS“Substantially improved 6 weeks later”
Connick et al (2004) []30 35MFace (periorbital) and neck swelling, starting during the 2 week of ART81>750,000T1, I1, S1Stavudine, lamivudine, lopinavir-ritonavir + paclitaxel (Start at 2 week for 10 weeks)AIDS-KS“At 10 weeks of ART, facial edema had resolved”
Feller et al (2008) []12 28MDevelopment of severe edema of the face after 3-month followup of his first clinic visitT1, Ix, SxRefused any TxAIDS-KSDeceased (within 3 weeks of onset of rapid progressive facial lymphoedema)
55MPronounced edema of the face and the lower extremities12T1, I1, S1AIDS-KSDeceased (7 days after presentation)
Volkow et al (2008) []31 19MAfter 2 weeks of prednisone for IRIS, growing facial edema in a matter of hours18221,000T1, I1, S1Lamivudine, stavudine, indinavir and ritonavir + vincristine and bleomycin (when facial edema developed)AIDS-KSResolved
Feller et al (2010) []32 11MRapid progressive facial swelling/lymphoedema14T1, I1, S1ARTAIDS-KSDeceased (4 days after admission, a few hours after starting ART)
Patel et al (2012) []33 37MFacial puffiness with periorbital edema with minimal skin findings227159,000T1, I0, S1AIDS-KS
Chabria et al (2016)[]34 51MEdema 2 weeks after initiation of prednisone (20 mg daily) for KS-IRIS476< 10T1, I0, S0Tenofovir, emtricitabine, and raltegravir + liposomal doxorubin (At onset of facial edema)Steroid-exacerbated AIDS cutaneous KS-IRISImproved
Call et al (2022)[]35 21MPersistent, non-tender, bilateral periorbital edema with right eye swollen shut, bilateral lower extremity non-pitting edema (received multiple corticosteroid regimens from various hospitals due to rash)154522T1, I1, S0ART + pegylated liposomal doxorubicinDisseminated AIDS-KS
KS – Kaposi sarcoma; AIDS-KS – acquired autoimmune deficiency syndrome-Kaposi sarcoma; ART – anti-retroviral therapy; IRIS – immune reconstitution inflammatory syndrome; TIS – Tumor, Immune System, Systemic Illness staging system.

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923