The patient was a 40-year-old female with a 5-month history of recurrent painful labial ulcers in December 2013. Investigations by a private physician revealed a negative VDRL (Venereal Disease Research Laboratory) test and vulval swab culture grew commensals only. The ulcers, however, failed to heal following treatment with antibiotics.

Vulval biopsy was performed at her first visit and histopathology report revealed only a non-specific ulcer with negative stains for fungus, bacteria, acid-fast bacilli, and herpes simplex virus. Papanicolaou smear showed atypical squamous cells of unknown significance (ASCUS).

In the absence of any obvious pathology for the non-healing ulcer, colposcopy was arranged: four ulcers were identified within the vagina, scattered over the right and left vaginal walls (Fig 1). A left lower vulval ulcer measuring 3 cm with slightly raised edges was also seen (Fig 2). Biopsies were taken over the cervix, vaginal, and vulval ulcers. Histopathology of these biopsies confirmed the presence of cervicitis and ulcers, with no evidence of malignancy.

Upon systemic review, the patient revealed that she had been suffering from recurrent mouth ulcers and bruising over both shins. Subsequent examination revealed multiple aphthous mouth ulcers, as well as erythema nodosum over both shins.

Based on the clinical signs, a diagnosis of Behçet’s disease was made. The patient was commenced on prednisolone, and all ulcers had healed at subsequent follow-up.

Behçet’s disease is a rare cause of genital ulceration. It is a multisystem vasculitic disorder and the diagnosis is based on clinical criteria.1 2 3 4 Our patient fulfilled the criteria of recurrent oral and genital ulcerations as well as a cutaneous manifestation.

Although there is little published evidence to support the value of colposcopy and biopsy in the diagnosis of Behçet’s disease, colposcopy enables a more detailed evaluation of the genital tract, and can be used to exclude the possibility of malignancy. Histopathological results in Behçet’s ulcer are typically non-specific with chronic active inflammation.

Papanicolaou smear in our patient showed ASCUS, but histopathology result for the cervix was normal. In a prospective study by Özdemir et al,2 abnormal cervical cytology, acetowhite epithelium, and iodine-negative epithelium on colposcopy were more common in Behçet’s patients. Nonetheless, the majority of ASCUS results revealed a normal finding for cervical histopathology. It was suggested that this was due to the benign inflammatory changes in the cervical epithelium.2

As in this case, diagnosis for recurrent genital ulcers can be difficult although when information about oral ulcers became available, a diagnosis of Behçet’s became more obvious. A complete history and physical examination that pays particular attention to signs or symptoms of an underlying associated systemic condition are essential when evaluating patients with recurrent genital ulcers.

1. Patel S, Prime K. Recurrent vulval ulceration: could it be Behçet’s disease? Int J STD AIDS 2012;23:683-4. Crossref

2. Özdemir S, Özdemir M, Celik C, Balevi A, Toy H, Kamış U. Evaluation of patients with Behçet’s disease by cervical cytology and colposcopic examination. Arch Gynecol Obstet 2012;285:1363-8. Crossref

3. Keogan MT. Clinical Immunology Review Series: an approach to the patient with recurrent orogenital ulceration, including Behçet’s syndrome. Clin Exp Immunol 2009;156:1-11. Crossref

4. Bandow GD. Diagnosis and management of vulvar ulcers. Dermatol Clin 2010;28:753-63. Crossref

A 90-year-old Caucasian female was admitted to the hospital following a fall preceded by left thigh pain for 2 weeks in May 2013. Her medical history included postmenopausal osteoporosis, depression, ischaemic heart disease, and atrial fibrillation. She was on alendronate for 11 years, as well as aspirin, bisoprolol, digoxin, calcium, vitamin D, frusemide, metformin, paracetamol, pantoprazole, and sertraline for around 14 years prior to hospital admission.

The Figure shows an atypical diaphyseal fracture of the femur commonly associated with long-term bisphosphonate therapy. These fractures usually occur in patients taking bisphosphonates for more than 5 years although it is known to occur with shorter duration of usage and in bisphosphonate-naïve patients (10%).1 This patient fulfilled all the ASBMR (American Society for Bone and Mineral Research) task force major and minor criteria for atypical fractures.2 The mandatory major criterion is fracture located along the femoral diaphysis from just distal to the lesser trochanter to just proximal to the supracondylar flare. In addition, at least four of five major features must be present: (1) The fracture is associated with minimal or no trauma, as in a fall from a standing height or less. (2) The fracture line originates at the lateral cortex and is substantially transverse in its orientation, although it may become oblique as it progresses medially across the femur. (3) Complete fractures extend through both cortices and may be associated with a medial spike; incomplete fractures involve only the lateral cortex. (4) The fracture is non-comminuted or minimally comminuted. (5) There is localised periosteal or endosteal thickening of the lateral cortex at the fracture site (“beaking” or “flaring”). The minor features not essential for diagnosis include: generalised increase in cortical thickness of the femoral diaphysis, unilateral or bilateral prodromal symptoms such as dull or aching pain in the groin or thigh, bilateral incomplete or complete femoral diaphysis fracture, and delayed fracture healing. Differential diagnoses in these patients include femoral fractures with subtrochanteric extension, pathological fractures associated with tumours, and periprosthetic fractures.2

Management strategies include cessation of bisphosphonates, protected weight-bearing, prophylactic intramedullary rod insertion, and use of anabolic bone agents like teriparatide or strontium.3 It is unclear whether a drug holiday is useful to prevent these fractures.4 Our patient was treated with intramedullary nailing and commenced on strontium after cessation of bisphosphonates. Greater awareness of this condition would prevent misdiagnosis especially in frail older patients and facilitate proper management.

Dr PK Shibu has received educational grants and honorarium from Novartis Pty Australia for Osteoporosis and Fracture Liaison related clinical research projects or lectures in the past and received honorarium from Amgen Ltd for presenting at educational meetings in the past.

1. Dell RM, Adams AL, Greene DF, et al. Incidence of atypical nontraumatic diaphyseal fractures of the femur. J Bone Miner Res 2012;27:2544-50. Crossref

2. Shane E, Burr D, Abrahamsen B, et al. Atypical subtrochanteric and diaphyseal femoral fractures: second report of a task force of the American Society for Bone and Mineral Research. J Bone Miner Res 2014;29:1-23. Crossref

3. Carvalho NN, Voss LA, Almeida MO, Salgado CL, Bandeira F. Atypical femoral fractures during prolonged use of bisphosphonates: short-term responses to strontium ranelate and teriparatide. J Clin Endocrinol Metab 2011;96:2675-80. Crossref

4. Diab DL, Watts NB. Bisphosphonate drug holiday: who, when and how long. Ther Adv Musculoskelet Dis 2013;5:107-11. Crossref