Letters to the Editor—Brief Communication / European Journal of Obstetrics & Gynecology and Reproductive Biology 172 (2014) 136–139
those in the pregnancy samples (approximately 12% and 13% respectively in the non-pregnant state compared with approximately 16% during pregnancy) but much higher than in most normal adults. Residual low-level HbF continues to be synthesized during adult life [4]. Using specialized techniques, F-cells can be detected in up to 7% of normal adults [5]. Awareness of the possibility of elevated maternal haemoglobin F ensures that samples can be sent quickly to appropriate reference laboratories. These patients should continue to receive anti-D following potentially sensitizing events and at delivery, and FMH investigated when indicated by both AE and flow cytometry techniques. Finally, a suspected false positive AE test should be a diagnosis of exclusion. In summary, these two cases highlight a challenging clinical dilemma requiring multidisciplinary input. Contributions JCD, FNiA, BK, AD wrote the paper; FNiA and BK edited the paper; BK coordinated investigations performed in Bristol Institute for Transfusion Sciences, DC and DM coordinated investigations performed in the Rotunda Hospital, Dublin, FNiA, JCD, SMC were involved in the clinical management of the patient. Conflicts of interests The authors report no conflicts of interest
Vulval oedema: how many doctors does it take to make a diagnosis? Dear Editor, We would like to highlight the case of a 17-year-old woman with vulval oedema in whom it took multiple doctors in multiple specialities 2 years to confirm the underlying diagnosis. We feel lessons on improving the diagnostic pathway for vulval oedema have been learnt locally and we wish to share our findings with your readers. She presented initially with a 1-year history of ‘vulval lumps’. Molluscum contagiosum was suspected and she was referred to the Genitourinary Medicine Department. The lumps were itchy and intermittently discharged pus. She also gave a long history of swelling to her vulva and, in her early teens, mouth ulcers and intermittent swelling of the cheek and lower lip. Systemic enquiry revealed no other symptoms. On examination she had a body mass index (BMI) of 18 and her labia were diffusely oedematous with multiple pedunculated lesions, typical of lymphangiectasia (Fig. 1). Abdominal and pelvic examination was otherwise normal. Investigations revealed a hypochromic microcytic anaemia (haemoglobin 8.4 g/dl). Chest X-ray, serum angiotensinogenconverting enzyme (ACE), renal and liver function and acute phase proteins were all normal. Imaging of her pelvis (ultrasound then MRI) reported only vulval oedema and polycystic ovaries. She was reviewed by the gastroenterology team and the vulval clinic. A radioactive white cell scan was arranged which was normal. She
References [1] Whittle MJ. Rhesus haemolytic disease. Arch Dis Child 1992;67:65–8. [2] Zipursky A, Hull A, White FD, Israels LG. Foetal erythrocytes in the maternal circulation. Lancet 1959;1:451–2. [3] Chambers E, Davies L, Evans S, Birchall J, Kumpel B. Comparison of haemoglobin F detection by the acid elution test, flow cytometry and high-performance liquid chromatography in maternal blood samples analysed for fetomaternal haemorrhage. Transfus Med 2012;22:199–204. [4] Thein SL, Menzel S, Lathrop M, Garner C. Control of fetal hemoglobin: new insights emerging from genomics and clinical implications. Hum Mol Genet 2009;18(R2):R216–23. [5] Wood WG, Stamatoyannopoulos G, Lim G, Nute PE. F-cells in the adult: normal values and levels in individuals with hereditary and acquired elevations of Hb F. Blood 1975;671–82.
Jennifer C. Donnelly Sharon M. Cooley Aoife Doyle Department of Obstetrics, Rotunda Hospital, Parnell Street, Dublin 1, Ireland Deirdre Murphy Deirdre Corcoran Department of Haematology and Blood Transfusion, Rotunda Hospital, Parnell Street, Dublin 1, Ireland Belinda Kumpel Bristol Institute for Transfusion Sciences, NHS Blood and Transplant, Bristol, United Kingdom Fionnuala Nı´ A´inle* Department of Haematology and Blood Transfusion, Rotunda Hospital, Parnell Street, Dublin 1, Ireland *Corresponding author. Tel.: +353 1 8730700; fax: +353 1 8733760 E-mail address: [email protected] (F.N. A´inle) 14 June 2013 http://dx.doi.org/10.1016/j.ejogrb.2013.10.012
137
Fig. 1. Patients vulva at presentation.
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Letters to the Editor—Brief Communication / European Journal of Obstetrics & Gynecology and Reproductive Biology 172 (2014) 136–139
was offered a vulval biopsy but declined. Over the next year her problems persisted and she went on to develop perianal oedema and lymphangiectasia. A colonoscopy with terminal ileal biopsies was performed (the colon and terminal ileum were macroscopically normal) along with a vulval biopsy. On histology microscopic foci of inflammation with early aphthous ulceration and granulomas were seen in the ileum with inflammation and granulomas on the vulval sample too, consistent with a diagnosis of Crohn’s disease. She remains under gastroenterology follow-up and is currently taking azathioprine. A delayed diagnosis, at such a vulnerable age, also contributed to negative feelings and embarrassment for her. Crohn’s disease is a chronic granulomatous inflammatory bowel disorder. It has several well-recognised extra-intestinal manifestations such as erythema nodosum, uveitis and arthritis. Vulval involvement is much rarer but is possibly underdiagnosed [1]. It occurs most commonly by direct extension of gastrointestinal disease but more rarely as ‘metastatic Crohn’s disease’ (MCD), where cutaneous granulomatous reactions occur in areas separated from the gastrointestinal tract by normal skin. Metastatic vulval involvement manifests most commonly as labial swelling or induration, but erythema, nodules, papules, plaques, pustules or ulceration have all been seen [2]. It is worth remembering that in 25% of cases of cutaneous involvement, skin changes predate the development of gastrointestinal symptoms by years. This can lead to diagnostic difficulty, which is confounded by the fact that MCD is characterised histologically by granulomatous inflammation, which carries a broad differential diagnosis (including mycobacterial infections, actinomycosis, lymphogranuloma venereum, syphilis, donovanosis, sarcoidosis and hidradenitis suppurativa) [2]. It has been noted that particularly in the paediatric cohort, vulval oedema caused by MCD is classically non-tender and painless, which may be of some help diagnostically. Even in the absence of gastrointestinal symptoms, both colonoscopy with biopsy (demonstrating non-caseating granulomata) [3] and the use of MRI (with demonstration of skin thickening, subcutaneous oedema, abscess formation or fistulae) [4] may be useful. MRI also has a role in excluding other potential causes of vulval oedema, such as vascular malformations or malignancies. Radiolabelled leucocyte scans have significant limitations in the initial diagnosis of Crohn’s disease as histology is not obtained, and faecal biomarkers may now provide a better non-invasive test for bowel inflammation [5]. This case illustrates the difficulty in early recognition of Crohn’s disease when women present with cutaneous vulvar changes, in absence of active gastrointestinal symptoms. Ultimately a multidisciplinary approach, along with skin and intestinal biopsies secured the diagnosis of this multisystem disease. References [1] Foo WC, Papalas JA, Robboy SJ, Selim MA. Vulvar manifestations of Crohn’s disease. Am J Dermatopathol 2011;33:588–93. [2] Keiler S, Tyson P, Tamburro J. Metastatic cutaneous Crohn’s disease in children: case report and review of the literature. Pediatr Dermatol 2009;26:604–9. [3] Mun JH, Kim SH, Jung DS, Ko HC, Kim MB, Kwon KS. Unilateral, non-tender, vulvar swelling as the presenting sign of Crohn’s disease: a case report and our suggestion for early diagnosis. J Dermatol 2011;38:303–7. [4] Pai D, Dillman JR, Mahani MG, Strouse PJ, Adler J. MRI of vulvar Crohn’s disease. Pediatr Radiol 2011;41:537–41. [5] Lamb CA, Mansfield JC. Measurement of faecal calprotectin and lactoferrin in inflammatory bowel disease. Frontline Gastroenterol 2011;2:13–8.
L. Mitchell Genitourinary Medicine Department, Newcastle Hospitals NHS Foundation Trust, New Croft Centre, Market Street East, Newcastle upon Tyne NE1 6ND, United Kingdom
J. Hussey* Genitourinary Medicine Department, City Hospitals Sunderland NHS Foundation Trust, Kayll Road, Sunderland SR4 7TP, United Kingdom J. Mansfield M. Gunn Gastroenterology Department, Newcastle Hospitals NHS Foundation Trust, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne NE1 4LP, United Kingdom J. McLelland Dermatology Department, Newcastle Hospitals NHS Foundation Trust, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne NE1 4LP, United Kingdom S. Needham Histopathology Department, Newcastle Hospitals NHS Foundation Trust, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne NE1 4LP, United Kingdom *Corresponding author. Tel.: +44 0191 5699021; fax: +44 0191 5699244 E-mail addresses: [email protected] [email protected] (J. Hussey) 19 March 2013 http://dx.doi.org/10.1016/j.ejogrb.2013.10.020
Ureaplasma parvum peritonitis after oocyte retrieval for in vitro fertilization Dear Editor, Ultrasound-guided transvaginal oocyte retrieval (TVOR) during in vitro fertilization (IVF) is a simple procedure, but not without risk. Complications are rare but can be serious. The most frequent ones are vaginal bleeding and pelvic infection. We report here an unusual case of peritonitis caused by Ureaplasma parvum after oocyte retrieval for IVF. In March 2010, 72 h after oocyte retrieval for IVF was performed, a 34-year-old woman was hospitalized for severe pelvic pain mainly in the right iliac fossa, with fever of 39 8C for the last 24 h. The couple had undergone oocyte retrieval for male factor infertility. The patient received antimicrobial prophylaxis at the time of TVOR (cefoxitin 2 g, one intravenous injection). On admission, physical examination revealed guarding in the right iliac fossa, predominant right lumbar pain and the presence of leukocytes and nitrites on the urine dipstick test. Laboratory testing showed a biological inflammatory syndrome with a leukocytosis at 9350 neutrophils/mm3 and C-reactive protein at 202 mg/l. The patient was treated with cefixime 200 mg 2 per day. Pelvic ultrasound performed on the following day showed a pelvic fluid collection in the pouch of Douglas and a periappendiceal collection suggestive of acute appendicitis. As her clinical condition failed to improve, an exploratory laparoscopy was performed in the digestive surgery department at the Bordeaux University Hospital. A retro-uterine pelvic collection of pus was found without inflammation of the appendix. The abscess was drained and bacteriological specimens were taken (deep pus and false membranes).
those in the pregnancy samples (approximately 12% and 13% respectively in the non-pregnant state compared with approximately 16% during pregnancy) but much higher than in most normal adults. Residual low-level HbF continues to be synthesized during adult life [4]. Using specialized techniques, F-cells can be detected in up to 7% of normal adults [5]. Awareness of the possibility of elevated maternal haemoglobin F ensures that samples can be sent quickly to appropriate reference laboratories. These patients should continue to receive anti-D following potentially sensitizing events and at delivery, and FMH investigated when indicated by both AE and flow cytometry techniques. Finally, a suspected false positive AE test should be a diagnosis of exclusion. In summary, these two cases highlight a challenging clinical dilemma requiring multidisciplinary input. Contributions JCD, FNiA, BK, AD wrote the paper; FNiA and BK edited the paper; BK coordinated investigations performed in Bristol Institute for Transfusion Sciences, DC and DM coordinated investigations performed in the Rotunda Hospital, Dublin, FNiA, JCD, SMC were involved in the clinical management of the patient. Conflicts of interests The authors report no conflicts of interest
Vulval oedema: how many doctors does it take to make a diagnosis? Dear Editor, We would like to highlight the case of a 17-year-old woman with vulval oedema in whom it took multiple doctors in multiple specialities 2 years to confirm the underlying diagnosis. We feel lessons on improving the diagnostic pathway for vulval oedema have been learnt locally and we wish to share our findings with your readers. She presented initially with a 1-year history of ‘vulval lumps’. Molluscum contagiosum was suspected and she was referred to the Genitourinary Medicine Department. The lumps were itchy and intermittently discharged pus. She also gave a long history of swelling to her vulva and, in her early teens, mouth ulcers and intermittent swelling of the cheek and lower lip. Systemic enquiry revealed no other symptoms. On examination she had a body mass index (BMI) of 18 and her labia were diffusely oedematous with multiple pedunculated lesions, typical of lymphangiectasia (Fig. 1). Abdominal and pelvic examination was otherwise normal. Investigations revealed a hypochromic microcytic anaemia (haemoglobin 8.4 g/dl). Chest X-ray, serum angiotensinogenconverting enzyme (ACE), renal and liver function and acute phase proteins were all normal. Imaging of her pelvis (ultrasound then MRI) reported only vulval oedema and polycystic ovaries. She was reviewed by the gastroenterology team and the vulval clinic. A radioactive white cell scan was arranged which was normal. She
References [1] Whittle MJ. Rhesus haemolytic disease. Arch Dis Child 1992;67:65–8. [2] Zipursky A, Hull A, White FD, Israels LG. Foetal erythrocytes in the maternal circulation. Lancet 1959;1:451–2. [3] Chambers E, Davies L, Evans S, Birchall J, Kumpel B. Comparison of haemoglobin F detection by the acid elution test, flow cytometry and high-performance liquid chromatography in maternal blood samples analysed for fetomaternal haemorrhage. Transfus Med 2012;22:199–204. [4] Thein SL, Menzel S, Lathrop M, Garner C. Control of fetal hemoglobin: new insights emerging from genomics and clinical implications. Hum Mol Genet 2009;18(R2):R216–23. [5] Wood WG, Stamatoyannopoulos G, Lim G, Nute PE. F-cells in the adult: normal values and levels in individuals with hereditary and acquired elevations of Hb F. Blood 1975;671–82.
Jennifer C. Donnelly Sharon M. Cooley Aoife Doyle Department of Obstetrics, Rotunda Hospital, Parnell Street, Dublin 1, Ireland Deirdre Murphy Deirdre Corcoran Department of Haematology and Blood Transfusion, Rotunda Hospital, Parnell Street, Dublin 1, Ireland Belinda Kumpel Bristol Institute for Transfusion Sciences, NHS Blood and Transplant, Bristol, United Kingdom Fionnuala Nı´ A´inle* Department of Haematology and Blood Transfusion, Rotunda Hospital, Parnell Street, Dublin 1, Ireland *Corresponding author. Tel.: +353 1 8730700; fax: +353 1 8733760 E-mail address: [email protected] (F.N. A´inle) 14 June 2013 http://dx.doi.org/10.1016/j.ejogrb.2013.10.012
137
Fig. 1. Patients vulva at presentation.
138
Letters to the Editor—Brief Communication / European Journal of Obstetrics & Gynecology and Reproductive Biology 172 (2014) 136–139
was offered a vulval biopsy but declined. Over the next year her problems persisted and she went on to develop perianal oedema and lymphangiectasia. A colonoscopy with terminal ileal biopsies was performed (the colon and terminal ileum were macroscopically normal) along with a vulval biopsy. On histology microscopic foci of inflammation with early aphthous ulceration and granulomas were seen in the ileum with inflammation and granulomas on the vulval sample too, consistent with a diagnosis of Crohn’s disease. She remains under gastroenterology follow-up and is currently taking azathioprine. A delayed diagnosis, at such a vulnerable age, also contributed to negative feelings and embarrassment for her. Crohn’s disease is a chronic granulomatous inflammatory bowel disorder. It has several well-recognised extra-intestinal manifestations such as erythema nodosum, uveitis and arthritis. Vulval involvement is much rarer but is possibly underdiagnosed [1]. It occurs most commonly by direct extension of gastrointestinal disease but more rarely as ‘metastatic Crohn’s disease’ (MCD), where cutaneous granulomatous reactions occur in areas separated from the gastrointestinal tract by normal skin. Metastatic vulval involvement manifests most commonly as labial swelling or induration, but erythema, nodules, papules, plaques, pustules or ulceration have all been seen [2]. It is worth remembering that in 25% of cases of cutaneous involvement, skin changes predate the development of gastrointestinal symptoms by years. This can lead to diagnostic difficulty, which is confounded by the fact that MCD is characterised histologically by granulomatous inflammation, which carries a broad differential diagnosis (including mycobacterial infections, actinomycosis, lymphogranuloma venereum, syphilis, donovanosis, sarcoidosis and hidradenitis suppurativa) [2]. It has been noted that particularly in the paediatric cohort, vulval oedema caused by MCD is classically non-tender and painless, which may be of some help diagnostically. Even in the absence of gastrointestinal symptoms, both colonoscopy with biopsy (demonstrating non-caseating granulomata) [3] and the use of MRI (with demonstration of skin thickening, subcutaneous oedema, abscess formation or fistulae) [4] may be useful. MRI also has a role in excluding other potential causes of vulval oedema, such as vascular malformations or malignancies. Radiolabelled leucocyte scans have significant limitations in the initial diagnosis of Crohn’s disease as histology is not obtained, and faecal biomarkers may now provide a better non-invasive test for bowel inflammation [5]. This case illustrates the difficulty in early recognition of Crohn’s disease when women present with cutaneous vulvar changes, in absence of active gastrointestinal symptoms. Ultimately a multidisciplinary approach, along with skin and intestinal biopsies secured the diagnosis of this multisystem disease. References [1] Foo WC, Papalas JA, Robboy SJ, Selim MA. Vulvar manifestations of Crohn’s disease. Am J Dermatopathol 2011;33:588–93. [2] Keiler S, Tyson P, Tamburro J. Metastatic cutaneous Crohn’s disease in children: case report and review of the literature. Pediatr Dermatol 2009;26:604–9. [3] Mun JH, Kim SH, Jung DS, Ko HC, Kim MB, Kwon KS. Unilateral, non-tender, vulvar swelling as the presenting sign of Crohn’s disease: a case report and our suggestion for early diagnosis. J Dermatol 2011;38:303–7. [4] Pai D, Dillman JR, Mahani MG, Strouse PJ, Adler J. MRI of vulvar Crohn’s disease. Pediatr Radiol 2011;41:537–41. [5] Lamb CA, Mansfield JC. Measurement of faecal calprotectin and lactoferrin in inflammatory bowel disease. Frontline Gastroenterol 2011;2:13–8.
L. Mitchell Genitourinary Medicine Department, Newcastle Hospitals NHS Foundation Trust, New Croft Centre, Market Street East, Newcastle upon Tyne NE1 6ND, United Kingdom
J. Hussey* Genitourinary Medicine Department, City Hospitals Sunderland NHS Foundation Trust, Kayll Road, Sunderland SR4 7TP, United Kingdom J. Mansfield M. Gunn Gastroenterology Department, Newcastle Hospitals NHS Foundation Trust, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne NE1 4LP, United Kingdom J. McLelland Dermatology Department, Newcastle Hospitals NHS Foundation Trust, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne NE1 4LP, United Kingdom S. Needham Histopathology Department, Newcastle Hospitals NHS Foundation Trust, Royal Victoria Infirmary, Queen Victoria Road, Newcastle upon Tyne NE1 4LP, United Kingdom *Corresponding author. Tel.: +44 0191 5699021; fax: +44 0191 5699244 E-mail addresses: [email protected] [email protected] (J. Hussey) 19 March 2013 http://dx.doi.org/10.1016/j.ejogrb.2013.10.020
Ureaplasma parvum peritonitis after oocyte retrieval for in vitro fertilization Dear Editor, Ultrasound-guided transvaginal oocyte retrieval (TVOR) during in vitro fertilization (IVF) is a simple procedure, but not without risk. Complications are rare but can be serious. The most frequent ones are vaginal bleeding and pelvic infection. We report here an unusual case of peritonitis caused by Ureaplasma parvum after oocyte retrieval for IVF. In March 2010, 72 h after oocyte retrieval for IVF was performed, a 34-year-old woman was hospitalized for severe pelvic pain mainly in the right iliac fossa, with fever of 39 8C for the last 24 h. The couple had undergone oocyte retrieval for male factor infertility. The patient received antimicrobial prophylaxis at the time of TVOR (cefoxitin 2 g, one intravenous injection). On admission, physical examination revealed guarding in the right iliac fossa, predominant right lumbar pain and the presence of leukocytes and nitrites on the urine dipstick test. Laboratory testing showed a biological inflammatory syndrome with a leukocytosis at 9350 neutrophils/mm3 and C-reactive protein at 202 mg/l. The patient was treated with cefixime 200 mg 2 per day. Pelvic ultrasound performed on the following day showed a pelvic fluid collection in the pouch of Douglas and a periappendiceal collection suggestive of acute appendicitis. As her clinical condition failed to improve, an exploratory laparoscopy was performed in the digestive surgery department at the Bordeaux University Hospital. A retro-uterine pelvic collection of pus was found without inflammation of the appendix. The abscess was drained and bacteriological specimens were taken (deep pus and false membranes).
