Primary male factor infertility due to asthenospermia in maturity-onset diabetes of the young type 5 (MODY 5): uncommon presentation of an uncommon disease

Mutations in hepatocyte nuclear factor-1β gene result in a multisystemic syndrome where a monogenic form of diabetes (maturity-onset diabetes of young type 5; MODY 5) and renal anomalies, usually bilateral multiple cysts are the most characteristic findings. Many of them have pancreatic structural abnormalities as well. A plethora of extrapancreatic manifestations like altered liver function tests, hypomagnesaemia, hyperuricaemia with/without gout and urogenital malformations, particularly in females are also components of the syndrome. Structural malformation of male urogenital tract is rare in MODY 5, even rarer is asthenospermia. We encountered a young non-obese individual having insulin-requiring diabetes following secondary oral agent failure with primary male factor infertility secondary to asthenospermia. A suggestive family history, lack of acanthosis, negative pancreatic autoimmunity, hypomagnesaemia, bilateral renal and epididymal cysts, and absence of body and tail of pancreas pointed towards underlying MODY 5.

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Superficial temporal artery pseudoaneurysm following facial trauma

A 68-year-old man presented with rapid swelling of the right forehead 11 days after sustaining a laceration secondary to a fall. Presumed to be an abscess due to retained foreign body, needle aspiration was performed and arterial blood obtained. Doppler ultrasound revealed a 3 cm mixed echogenicity lesion with 'see-sawing' internal Doppler flow arising from the superficial temporal artery (STA), in keeping with a pseudoaneurysm. Treatment options including interventional radiology and open surgery were considered. Open operative intervention with direct surgical ligation provided an excellent outcome. Delayed pseudoaneurysm of the STA is a rare complication of trauma but should be considered in the differential of a traumatic lateral forehead swelling to prevent complications and inappropriate investigations and management.

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Septic presentation of a giant fibroepithelial polyp of the vulva

Fibroepithelial stromal polyps are mesenchymal lesions occurring typically in reproductive age women with a predilection for the vulvo-vaginal region. Malignancy may mimic this polyp in morphology, rendering further investigations including detailed histopathology mandatory. Histologically its characteristic features are stellate and multinucleate stromal cells identified near the epithelial–stromal interface. This case report discusses incidental finding of largest fibroepithelial polyp presented in a 31-year-old nulliparous woman. She was initially admitted with sepsis and detailed physical examination revealed a right-sided infected pedunculated labial mass measuring 20x21 cm. After initial resuscitation for sepsis, she was further investigated for the mass. Transabdominal ultrasound was essentially normal apart from a small fibroid 3x2 cm in the anterior wall of the uterus. The vulval mass was removed under local and regional anaesthesia and was confirmed to be a giant fibroepithelial stromal polyp on histopathology. The woman recovered well and was followed up until 1 year.

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Herpes zoster ophthalmicus evolving into headache characterised as hemicrania continua

Postherpetic neuralgia (PHN) is the most common chronic complication of herpes zoster infection. However, a few patients may develop different types of pain after herpetic lesions. We are reporting two patients who developed postherpetic hemicrania continua (HC). Case 1: a 54-year-old woman had a 10-month history of continuous left-sided pain with superimposed exacerbations. The pain started with the onset of herpetic lesions in the ophthalmic division. The lesions subsided in a few weeks. However, the pain persisted and it responded exclusively to indomethacin. Case 2: a 61-year-old woman developed clinical features pertinent to PHN. However, later on, the pattern and associated clinical features changed. The patient fulfilled the criteria of HC and showed a complete response to indomethacin. We suggest that every patient with PHN should be asked for cranial autonomic features and a trial of indomethacin should be given in refractory herpes zoster neuropathy.

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Thyrotoxic crisis as an acute clinical presentation in a child

A previously well, 4-year-old girl presented with a 4–6 weeks' history of increased appetite, weight loss, tiredness, sleep difficulty, excessive sweating, swelling in the neck and new-onset 'prominent, protruding eyes.' Family history revealed paternal grandmother receiving treatment for hyperthyroidism. Clinical assessment demonstrated features of thyrotoxicosis (tachycardia, warm peripheries, small smooth goitre with no nodules, exophthalmos). TFT (Free T4=101 pmol/L, thyroid-stimulating hormone <0.05 mIU/L) with raised thyroid peroxidase antibody levels (TPO=541 IU/mL) confirmed autoimmune hyperthyroidism. Observation on the ward showed features of thyrotoxic crisis with persistent severe tachycardia on ECG (sinus tachycardia with left ventricular hypertrophy (LVH)) and hypertension. Ultrasound thyroid showed diffuse thyroiditis with no focal lesion. Echocardiogram confirmed the above findings. A diagnosis of Graves' disease with thyrotoxic crisis was made. Antithyroid treatment (carbimazole) and beta-blocker (propranolol) was commenced. Thyrotoxic crisis resolved over 2 weeks and the child has continued to respond to carbimazole treatment at 1-year follow-up.

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Worm in anterior chamber of the eye

We report a case of a 42-year-old female patient who presented to the ophthalmology outpatient department with painful red eye for 1 month. Slit-lamp microscopy showed a live worm in the anterior chamber of left eye. The worm was surgically removed under topical anaesthesia. It was sent to the microbiology department for further identification and was found to be adult female Loaloa.

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Placenta accreta complicated with peripartum cardiomyopathy

A 33-year-old G2P1 was referred to our hospital due to placenta accreta. During perioperative preparations, the patient was diagnosed with having a peripartum cardiomyopathy. The patient underwent caesarean hysterectomy at 36 weeks with an associated 2 L blood loss. Haemodynamic maintenance and stabilisation during the operation were challenging, with the combinations of fluid therapy, blood transfusions as well as vasoactive, antifibrinolytic and haemostatic drug. Postoperatively, the patient was managed in the intensive care unit and was subsequently transferred to intermediate care after less than 24 hours' observation. She was stable enough to be moved to the obstetrics ward the next day.

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