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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 46  |  Issue : 1  |  Page : 44-49

A confirmed case of menstrual cup-associated toxic shock syndrome: A Philippine perspective


Department of Obstetrics and Gynecology, ManilaMed, Manila, Philippines

Date of Submission24-Jan-2022
Date of Acceptance24-Jan-2022
Date of Web Publication15-Apr-2022

Correspondence Address:
Andrea Francesca I. Santos
MD, Department of Obstetrics and Gynecology, Medical Center Manila, 850 United Nations Ave, Paco, Manila
Philippines
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/pjog.pjog_8_22

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  Abstract 


Menstrual toxic shock syndrome (TSS) is a severe, fatal, superantigen toxin-mediated illness, which leads to multiorgan system failure early in its course. At the time of writing, there are no local data available on menstrual cup-associated TSS. Reported is a 30-year-old healthy Filipino, diagnosed as a case of menstrual cup-associated TSS. Diagnosis was confirmed by case definition criteria and supported by vaginal discharge culture of methicillin-resistant Staphylococcus aureus. The patient was treated empirically with antibiotics that led to successful treatment outcomes with no recurrence. At present, when women empowerment is of utmost importance, we support women's decisions pertaining to their health, including their choice of menstrual hygiene products. This case is reported to raise awareness, promote wellness and safety among menstrual cup users and to educate clinicians on the course and management of menstrual cup associated toxic shock syndrome to prevent its catastrophic sequelae.

Keywords: Menstrual cup, methicillin-resistant Staphylococcus aureus, toxic shock syndrome


How to cite this article:
Santos AF, Montinola-Morales MM. A confirmed case of menstrual cup-associated toxic shock syndrome: A Philippine perspective. Philipp J Obstet Gynecol 2022;46:44-9

How to cite this URL:
Santos AF, Montinola-Morales MM. A confirmed case of menstrual cup-associated toxic shock syndrome: A Philippine perspective. Philipp J Obstet Gynecol [serial online] 2022 [cited 2022 May 27];46:44-9. Available from: https://www.pogsjournal.org/text.asp?2022/46/1/44/343237




  Introduction Top


Toxic shock syndrome (TSS) was first introduced in 1978 in a Lancet publication describing the disease to occur in children 8–17 years of age presenting as an acute fatal febrile illness.[1] It was in 1980 when TSS was associated with young menstruating women; thus, it was further categorized into two: menstrual and nonmenstrual TSS.[2]

Menstrual TSS is a severe, superantigen toxin-mediated, multiorgan system disease, whose sequelae are shock and multiorgan system failure early in its clinical course. It is highly associated with the use of highly absorbent tampons among young healthy women, with more than 90% having Staphylococcus aureus isolates in their vaginal cultures. It was postulated that the root cause of disease is the inflammatory response to the toxin produced by S. aureus which is the TSS toxin 1.[3]

The incidence of menstrual and nonmenstrual TSS is estimated to be around 0.8–3.4 per 100,000 with menstrual cases accounting for 74% of total cases in the United States. However, at present, there are no local statistics available with no published case at the time of writing.[4]

In lieu of advancements of menstrual hygiene products, the menstrual cup was invented and slowly introduced worldwide including the Philippines. The menstrual cup is a menstrual hygiene device, made of silicone, rubber, or thermoplastic isomer in the shape of a cup or bell whose main purpose is to collect menstrual blood. It is found to be more convenient, ergonomic, and cost-effective than the traditional napkin pads and tampons.[5] It is also said to be safer than tampons due to the very rare incidence of menstrual TSS associated with its use.[6],[7]

Reported here is a case of a 30-year-old, nulligravid diagnosed as a confirmed case of menstrual cup-associated TSS treated with clindamycin and meropenem, which led to successful treatment outcome.


  Case Report Top


This is a case of a 30-year-old Filipino, nulligravid, with no known present comorbidities. She is allergic to ranitidine and cotton napkin pads. No allergies to food, latex, rubber, and other medications were noted. She is a resident physician, a nonsmoker, and nonalcoholic beverage drinker. The patient menstruates regularly and has been using a 25cc silicone menstrual cup for 6 years, following manufacturer's recommendations of using it for less than 12 h, washing the cup with tap water in between use, and sterilizing it by boiling for 5 min daily. She also replaces the cup yearly.

The patient consulted our emergency room due to high-grade fever. History started 1-month prior; on her 2nd day of menses, she noted macular rashes on bilateral palms, arms, and shoulders, which were erythematous and pruritic. The patient self-medicated with cetirizine 10 mg/tablet which afforded relief of symptoms and clearing of rashes; no consult was done.

Three days before consult, the patient experienced headache, body malaise, and anorexia. This was on her 3rd day of menses where she also reported unintentional prolonged use of menstrual cup, of more than 12 h [Figure 1]. No consult was done and no medications were taken. The following day, symptoms persisted, now with associated arthralgia, myalgia, and high-grade fever Tmax of 39.3°C. Now, the pruritic macular rashes in her palms were noted to desquamate as well. She self-medicated with paracetamol 500 mg/tablet every 4 h which only provided temporary relief of symptoms.

Two hours before consult, on her 5th day of menses, symptoms persisted now with associated myalgia, vomiting, and erythematous tongue; thus, she consulted the emergency room.
Figure 1: Actual photograph of menstrual cup used by the index patient

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The patient was seen conscious and coherent but hypotensive and febrile with initial blood pressure range of 80–90/60 mmHg and temperature of 39°C, nontachypneic, and nontachycardic. She was initially managed with fluid bolus hydration with crystalloids and intravenous (IV) antipyretics, which elevated her blood pressure to 100/60 mmHg and decreased her temperature to 37°C. On physical examination, her skin was warm and supple, with note of bilateral erythematous palms with desquamation. There were no other rashes on the rest of her body [Figure 2]a. Her tongue was noted to be erythematous with prominent papules [Figure 2]b. Bilateral lung fields were clear, and cardiovascular findings were unremarkable. Her abdomen was flat, soft with no areas of tenderness. Pelvic examination was done revealing normal external genitalia. Speculum examination showed a hyperemic vaginal canal with rugae, noted copious amount of whitish-yellow foul-smelling vaginal discharge within the canal. The cervix was hyperemic, smooth with no lesions. On internal examination, the cervix was firm and smooth with cervical motion tenderness, uterus was small, retroverted not enlarged, and no bilateral adnexal masses or tenderness was noted. On rectovaginal examination, her anal sphincter tone was intact, parametria were smooth and pliable, and the cul-de-sac was not full.
Figure 2: (a) Bilateral erythematous palms, with desquamation. (b) Erythematous tongue with prominent papules

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Her blood picture revealed septicemia with neutrophilic predominance. Inflammatory markers of erythrocyte sedimentation rate and procalcitonin levels were elevated. Electrolytes, blood chemistry, bleeding parameters, 12-lead electrocardiogram, 2D echocardiogram, COVID-19 swab, transvaginal sonogram, and chest X-ray all revealed normal findings [Table 1]. Urine, blood, and vaginal cultures were also collected at the emergency room. At this time, her working impression is to consider menstrual cup-associated TSS, with possible pelvic inflammatory disease. With above findings, the patient was referred to an infectious disease specialist for comanagement. She was immediately started on clindamycin 600 mg and meropenem 1 g IV every 8 hours, as well as paracetamol 300 mg and metoclopramide 10 mg IV every 8 h for fever and vomiting. Hydration was maintained with crystalloids. She was monitored for any signs of deterioration such as neurologic changes, chest pain, dyspnea, fever, and recurrence of hypotension.
Table 1: Laboratory results from initial assessment

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On her 2nd hospital day, the patient was stable; however, she still complained of headache, anorexia, myalgia, and pruritus of both palms which were erythematous and desquamative. There were no episodes of fever, hypotension, chest pain, dyspnea, or any neurologic changes. Cultures were released; her urine and blood culture of both arms showed no growth after 48 h of incubation. Vaginal discharge Gram stain revealed the presence of 2+ Gram-positive cells and Gram-positive bacilli 3+. Vaginal discharge culture sensitivity isolated heavy growth of methicillin-resistant S. aureus, resistant to oxacillin and penicillin, and sensitive to clindamycin, cotrimoxazole, erythromycin, linezolid, tetracycline, and vancomycin. She was referred to dermatology service for the comanagement of skin lesions whose initial impression was cutaneous manifestation of menstrual TSS. Supportive management was advised with hypoallergenic diet and started on levocetirizine 5 mg/tablet once at bedtime and beclomethasone ointment twice daily. Infectious disease specialist advised to complete 5 days of IV antibiotics. Meticulous monitoring and supportive management were continued.

On the 5th hospital day, the patient was well, stable, and improved. She was discharged with the following home medications; co-amoxiclav 625 mg/tablet 2× a day and clindamycin 300 mg/capsule 4× daily for 1 week, naproxen 550 mg/tablet twice daily for joint pains, desloratadine 5 mg/tablet once daily, betamethasone dipropionate ointment twice daily, and calcium + Vitamin D3 once daily. The patient was counseled well on perineal hygiene care and discontinuation of menstrual cup use. She was advised strict monthly follow-up and to observe for any signs of recurrence.


  Case Discussion Top


Menstrual TSS is a severe, acute, superantigen, toxin-mediated disease, characterized by rapid-onset hypotension, fever, and rash, which rapidly progresses to multiple-organ system failure and lethal shock. It is frequently preceded by a prodromal period involving fever, chills, myalgia, and gastrointestinal upset.[8] The overall rates of TSS in the United States have remained relatively stable since the late 1980s and are now presently in the range of 0.8–3.4 per 100,000 adults, with 74% attributed to menstrual TSS and mortality rates ranging from 1.8% to 12%. The peak of incidence, morbidity, and mortality of Menstrual TSS was associated mainly to the increased usage of highly absorbent tampons. However, after thorough patient education and withdrawal of ultra-absorbent tampons from the market, a significant decline in incidence and case fatality rates was observed from 5.5% in 1979–1980 to 1.8% in 1987–1996.[4]

Menstrual cups are menstrual hygiene products that are made of silicone, rubber, or latex, which are thermoplastic isomer that are increasingly used as a popular alternative to tampons. They are advertised to be ergonomic, convenient, and most of all safer than tampons.[9] In a study by Juma et al. in 2017,[6] they assessed women using female barrier methods, which similarly uses medical grade silicone or latex products, and the incidence of menstrual TSS is very low and was determined to be 2.25 cases per 100,000 users per year.[6]

Central to the pathophysiology of menstrual TSS is in association with the use of highly absorbent tampons. Tierno et al.[10] explained that, as menstrual blood accumulates in the polyester foam, it chips of carboxymethylcellulose, which in turn would produce carbon dioxide and increase vaginal pH during menses from the usual 4.2 to approximately 7.4. These factors provide a conducive environment for S. aureus growth, which is the organism responsible for the production of an exotoxin within the super antigen family, the TSS toxin 1. Toxin-mediated superantigen may induce clonal T-cell proliferation, resulting in massive cytokine release, subsequently causing cytokine storm that leads to fever, rash, capillary leakage, subsequent hypotension, and organ damage.[11] On the other hand, pathogenesis of TSS associated with menstrual cups is still unclear and may be multifactorial, since silicone itself does not promote microbial growth. It is hypothesized that the accumulation of blood provides a good medium for bacterial growth. In detail, an in vitro trial by Nonfoux et al. in 2018 postulated that air inserted into the vagina along with the cup favors S. aureus growth and subsequent TSS toxin 1 production in the menstrual blood collected in the cup. When the amount of menstrual blood exceeds that of the menstrual cup, the TSS toxin 1 produced in the fluid is in contact with the vaginal mucosa, resulting in toxin transcytosis into the blood which leads to menstrual TSS. It was also noted in their study that women may reinsert a contaminated cup following the manufacturer's instructions that the cup may be removed, emptied, and rinsed with tap water before reinserting. It was found that a significant amount of biofilm of S. aureus remained in the menstrual cup after 8 h and 3 washes regardless of model and composition; thus, this study recommends the use of small menstrual cups to avoid entry of air, and it emphasized boiling of menstrual cups in between uses.[12]

Diagnosis of TSS as defined by the Centers for Disease Control is divided into two categories: the probable case, where the laboratory criteria and four of the five clinical criteria described below are present, and the confirmed case, which is a case that meets the laboratory criteria and all five of the clinical criteria described are present, including desquamation, unless the patient dies before desquamation occurs[13] [Table 2].
Table 2: Centers for Disease Control and Prevention, 2011, Georgia, USA, case definition for toxic shock syndrome (other than Streptococcus)

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At present, there is no stern clinical guideline with regard to the treatment of menstrual TSS; however, to prevent the fatal consequence, once with clinical suspicion, it is mandated to start empiric antibiotics while awaiting culture results. The recommended treatment regimen is a combination of clindamycin 600–900 mg IV every 8 h together with a penicillin plus beta-lactamase inhibitor or carbapenem in the form of meropenem 1 g IV every 8 h or imipenem 1 g IV every 6 h.[14]

Clindamycin is an antibiotic used in the treatment of serious anaerobic, Staphylococcus, and Streptococcus infections, whose main mode of action is suppression of bacterial protein synthesis. More importantly, it is central to the treatment of menstrual TSS for its ability to suppress synthesis of TSS toxin 1.[15]

There is no clinical study to dictate treatment duration for menstrual TSS; the present recommendation is to continue treatment at least 48–72 h until patients are clinically and hemodynamically stable.[16]


  Summary Top


As correlated with our index patient, she is a known silicone menstrual cup user for 6 years with an unintentional usage of more than 12 h on day 3 of menses. She presented with the classic signs and symptoms of menstrual TSS, and her diagnosis was confirmed through case definition criteria set by the Centers for Disease Control 2011 where she fulfilled five of the clinical criteria: fever, rash, desquamation, and hypotension, with the presence of multisystem involvement - vomiting, myalgia, and vaginal and oropharyngeal hyperemia. This was further supported by the heavy growth of methicillin-resistant S. aureus in her vaginal discharge culture. She was treated with clindamycin 600 mg and meropenem 1 g IV every 8 h for 5 days, which led to successful treatment outcomes. There was no recurrence of hypotension or any signs of shock upon timely initiation of empiric antibiotic regimen. On discharge, all symptoms have been resolved. The patient was advised to discontinue use of menstrual cups; furthermore, clindamycin 300 mg/capsule every 6 h was continued and she was started on co-amoxiclav 625 mg/tablet every 12 h for 1 week as step down for meropenem. Continuation of antibiotics was done as preventive measure to avoid recurrence of menstrual TSS. At present, on her 4th month follow-up, there are no signs of recurrence. A study by Davis et al. stated that recurrence may occur in the absence of tampon use. Recurrence of menstrual TSS was observed in 5 out of 30 women despite discontinued tampon use during menses in a 5-month period after the initial episode.[14] Despite an established high recurrence rate, few recommendations regarding antibiotic prophylaxis in women previously treated for menstrual TSS have been proposed including clindamycin, rifampicin and penicillin plus beta-lactamase inhibitor. Another possible approach is the use of oral contraceptives, which prevents menstruation, thus reducing the risk of recurrence.[17] It is emphasized to discontinue menstrual cups and to administer additional oral antibiotics, to remove the carrier state of S. aureus which were all employed in our index patient.

At this day and age, when women empowerment is of utmost importance, we give women the freedom to choose what is right and appropriate for them, including the choice of sanitary menstrual hygiene products. It is a personal decision often influenced by cultural acceptability, user preferences, and affordable options. It is our responsibility as obstetricians and gynecologists to lead the way of awareness to promote wellness among Filipino women. To our knowledge, at present, there are only three international journals under the specialty of Internal Medicine who have published case reports of menstrual cup-associated TSS. In the local setting, there is rare if not limited knowledge in terms of incidence, morbidity, and mortality rate of this disease and our index patient may be the first Philippine reported case of menstrual cup-associated TSS treated with clindamycin and meropenem, which led to successful treatment outcomes. This paper is written to raise awareness, promote knowledge, and support women with their choice of menstrual hygiene products at the same time advocating wellness and safety.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Todd J, Fishaut M, Kapral F, Welch T. Toxic-shock syndrome associated with phage-group-I Staphylococci. Lancet 1978;2:1116-8.  Back to cited text no. 1
    
2.
Reingold AL, Hargrett NT, Shands KN, Dan BB, Schmid GP, Strickland BY, et al. Toxic shock syndrome surveillance in the United States, 1980 to 1981. Ann Intern Med 1982;96:875-80.  Back to cited text no. 2
    
3.
Parsonnet J, Hansmann MA, Delaney ML, Modern PA, Dubois AM, Wieland-Alter W, et al. Prevalence of toxic shock syndrome toxin 1-producing Staphylococcus aureus and the presence of antibodies to this superantigen in menstruating women. J Clin Microbiol 2005;43:4628-34.  Back to cited text no. 3
    
4.
Schmitz M, Roux X, Huttner B, Pugin J. Streptococcal toxic shock syndrome in the Intensive Care Unit. Ann Intensive Care 2018;8:88.  Back to cited text no. 4
    
5.
Available from: https://en.wikipedia.org/wiki/Menstrual_cup. [Last accessed on 2021 Aug 11].  Back to cited text no. 5
    
6.
Juma J, Nyothach E, Laserson KF, Oduor C, Arita L, Ouma C, et al. Examining the safety of menstrual cups among rural primary school girls in western Kenya: Observational studies nested in a randomised controlled feasibility study. BMJ Open 2017;7:e015429.  Back to cited text no. 6
    
7.
Lappin E, Ferguson AJ. Gram-positive toxic shock syndromes. Lancet Infect Dis 2009;9:281-90.  Back to cited text no. 7
    
8.
Mitchell MA, Bisch S, Arntfield S, Hosseini-Moghaddam SM. A confirmed case of toxic shock syndrome associated with the use of a menstrual cup. Can J Infect Dis Med Microbiol 2015;26:218-20.  Back to cited text no. 8
    
9.
Howard C, Rose CL, Trouton K, Stamm H, Marentette D, Kirkpatrick N, et al. FLOW (finding lasting options for women): Multicentre randomized controlled trial comparing tampons with menstrual cups. Can Fam Physician 2011;57:e208-15.  Back to cited text no. 9
    
10.
Tierno PM, Hanna BA. Propensity of tampons and barrier contraceptives to amplify Staphylococcus aureus Toxic shock syndrome toxin-I. Infect Dis Obstet Gynecol 1994;2:140-5.  Back to cited text no. 10
    
11.
Neumann C, Kaiser R, Bauer J. Menstrual cup-associated toxic shock syndrome. Eur J Case Rep Intern Med 2020;7:001825.  Back to cited text no. 11
    
12.
Nonfoux L, Chiaruzzi M, Badiou C, Baude J, Tristan A, Thioulouse J, et al. Impact of currently marketed tampons and menstrual cups on Staphylococcus aureus growth and toxic shock syndrome toxin 1 production in vitro. Appl Environ Microbiol 2018;84:e00351-18.  Back to cited text no. 12
    
13.
Center for Disease Control and Prevention. Toxic Shock Syndrome (Other than Streptococcal) (TSS) 2011 Case Definition; 2021. Available from: https://wwwn.cdc.gov/nndss/conditions/toxic-shock-syndrome-other-than-streptococcal/case-definition/2011/. [Last accessed on 2021 Sep 19].  Back to cited text no. 13
    
14.
Davis JP, Chesney PJ, Wand PJ, LaVenture M. Toxic-shock syndrome: Epidemiologic features, recurrence, risk factors, and prevention. N Engl J Med 1980;303:1429-35.  Back to cited text no. 14
    
15.
Sriskandan S, McKee A, Hall L, Cohen J. Comparative effects of clindamycin and ampicillin on superantigenic activity of Streptococcus pyogenes. J Antimicrob Chemother 1997;40:275-7.  Back to cited text no. 15
    
16.
Davis JP, Osterholm MT, Helms CM, Vergeront JM, Wintermeyer LA, Forfang JC, et al. Tri-state toxic-shock syndrome study. II. Clinical and laboratory findings. J Infect Dis 1982;145:441-8.  Back to cited text no. 16
    
17.
Dixit S, Fischer G, Wittekind C. Recurrent menstrual toxic shock syndrome despite discontinuation of tampon use: Is menstrual toxic shock syndrome really caused by tampons? Australas J Dermatol 2013;54:283-6.  Back to cited text no. 17
    


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