title: Facial disfigurement due to osteitis fibrosa cystica or brown tumor from secondary hyperparathyroidism in patients on dialysis: A systematic review and an illustrative case report.
abstract: Osteitis fibrosa cystica (OFC) is the most frequent type of osseous change in renal osteodystrophy affecting the majority of dialysis patients. Brown tumors are a severe form of OFC. The involvement of the craniofacial skeleton causing facial disfigurement in patients on dialysis appears to be limited to case reports. After searching PubMed, we performed a systematic review of 127 cases with a severe form of OFC resulting in a facial disfigurement to understand possible determinants for this condition. We found that since the first published case in 1974, and after a peak in 1996, there appears to be an increase in published reported cases. Only 27.6% of these cases were published in nephrology journals. The most common region for reported cases was North America. Mean age of these patients was 31.2 years with a mean dialysis duration of 7 years. Almost 67% were women, and almost all were on hemodialysis. The disease tended to most commonly localize to the maxilla (73.2%) and mandible (57.5%). As part of the treatment, 59% of patients had a parathyroidectomy. More than one-third (35.4%) had symptomatic improvement at follow-up. Mean follow-up was 1.6 years. Clinicians should be aware of this clinical presentation of a severe form of OFC and/or brown tumors. Timely diagnosis and intervention may help to prevent or decrease destructive bone changes and reduce negative psychological consequences of facial disfigurement.
authors.affiliation: Division of Nephrology and Hypertension, Nassau University Medical Center, East Meadow, NY, USA.
authors.forename: Rafael M
authors.initials: RM
authors.lastname: Baracaldo
authors.affiliation: Division of Nephrology and Hypertension, Nassau University Medical Center, East Meadow, NY, USA.
authors.forename: Dashi
authors.initials: D
authors.lastname: Bao
authors.affiliation: Division of Nephrology and Hypertension, Nassau University Medical Center, East Meadow, NY, USA.
authors.forename: Pichet
authors.initials: P
authors.lastname: Iampornpipopchai
authors.affiliation: Department of Finance and Business Management, Brooklyn College, Brooklyn, NY, USA.
authors.forename: Joshua
authors.initials: J
authors.lastname: Fogel
authors.affiliation: Division of Nephrology and Hypertension, Nassau University Medical Center, East Meadow, NY, USA.
authors.forename: Sofia
authors.initials: S
authors.lastname: Rubinstein
mesh_terms: D000328:Adult; D005151:Facial Injuries / Q000209:etiology*; D005260:Female; D006801:Humans; D006962:Hyperparathyroidism, Secondary / Q000150:complications*; D008297:Male; D010002:Osteitis Fibrosa Cystica / Q000150:complications*; D006435:Renal Dialysis / Q000009:adverse effects*
keywords: Brown tumors; craniofacial skeleton; dialysis; facial disfigurement; osteitis fibrosa cystica; secondary hyperparathyroidism